International Review of Research in Mental Retardation, Volume 38

V O L U M E T H I R T Y E I G H T INTERNATIONAL REVIEW OF RESEARCH IN MENTAL RETARDATION Board of Associate Edit...

Author: Robert M. Hodapp

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V O L U M E

T H I R T Y

E I G H T

INTERNATIONAL REVIEW

OF

RESEARCH IN MENTAL RETARDATION

Board of Associate Editors

PHILIP DAVIDSON University of Rochester School of Medicine and Dentistry

ELISABETH DYKENS Vanderbilt University

MICHAEL GURALNICK University of Washington

RICHARD HASTINGS University of Wales, Bangor

LINDA HICKSON Columbia University

CONNIE KASARI University of California, Los Angeles

WILLIAM McILVANE E. K. Shriver Center

GLYNIS MURPHY University of Kent

TED NETTELBECK Adelaide University

MARSHA MAILICK SELTZER University of Wisconsin-Madison

JAN WALLANDER Sociometrics Corporation

V O L U M E

T H I R T Y

E I G H T

INTERNATIONAL REVIEW

OF

RESEARCH IN MENTAL RETARDATION Edited by

Robert M. Hodapp Vanderbilt Kennedy Center for Research on Human Development Department of Special Education, Peabody College Vanderbilt University Nashville, TN 37205

AMSTERDAM • BOSTON • HEIDELBERG • LONDON NEW YORK • OXFORD • PARIS • SAN DIEGO SAN FRANCISCO • SINGAPORE • SYDNEY • TOKYO Academic Press is an imprint of Elsevier

Academic Press is an imprint of Elsevier 525 B Street, Suite 1900, San Diego, CA 92101-4495, USA 30 Corporate Drive, Suite 400, Burlington, MA 01803, USA 84 Theobald’s Road, London WC1X 8RR, UK Radarweg 29, PO Box 211, 1000 AE Amsterdam, The Netherlands First edition 2009 Copyright Ó 2009, Elsevier Inc. All rights reserved No part of this publication may be reproduced, stored in a retrieval system or transmitted in any form or by any means electronic, mechanical, photocopying, recording or otherwise without the prior written permission of the publisher Permissions may be sought directly from Elsevier’s Science & Technology Rights Department in Oxford, UK: phone (+44) (0) 1865 843830; fax (+44) (0) 1865 853333; email: [email protected]. Alternatively you can submit your request online by visiting the Elsevier web site at http://elsevier.com/locate/permissions, and selecting Obtaining permission to use Elsevier material Notice No responsibility is assumed by the publisher for any injury and/or damage to persons or property as a matter of products liability, negligence or otherwise, or from any use or operation of any methods, products, instructions or ideas contained in the material herein. Because of rapid advances in the medical sciences, in particular, independent verification of diagnoses and drug dosages should be made British Library Cataloguing in Publication Data A catalogue record for this book is available from the British Library Library of Congress Cataloging-in-Publication Data A catalog record for this book is available from the Library of Congress ISBN: 978-0-12-374467-8 ISSN: 0074-7750 For information on all Academic Press publications visit our website at elsevierdirect.com Printed and bound in the United States of America 08 09 10 10 9 8 7 6 5 4 3 2 1

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CONTENTS

Contributors Preface

ix xi

1. The Transition Out of High School and into Adulthood for Individuals with Autism and for Their Families

1

Julie Lounds Taylor 1. Introduction 2. Definitional Issues 3. Theories on the Transition to Adulthood: How Typical Development Can Inform the Study of Families of Adolescents and Young Adults with ASD 4. What Do We know About the Transition to Adulthood for Individuals with ASD and for Their Families? Review of the Extant Research 5. Directions for Future Research 6. Conclusions References

2. Inclusive Health and People with Intellectual Disabilities

2 4

5 10 24 28 28

33

Patricia Noonan Walsh and Roy McConkey 1. Introduction 2. Personal Development and Health 3. Health and Intellectual Disabilities: A Contextual Approach 4. Health Gain 5. Proposals for More Inclusiveness References

3. Persevering in the Face of Hardship: Families of Individuals with Developmental Disabilities in the People’s Republic of China

34 36 40 46 58 61

69

Peishi Wang 1. Introduction 2. Background 3. Educational Systems 4. Chinese Families of Individuals with Disabilities 5. Conclusion References

70 70 73 77 88 89 v

vi

Contents

4. Families of Children with Intellectual Disabilities in Vietnam: Emerging Themes

93

Emily D’Antonio and Jin Y. Shin 1. 2. 3. 4.

Introduction Context of Disability-Family Studies in Vietnam Major Themes Arising From Vietnamese Family Studies Development of Psychometrically Valid Scales in a Vietnamese Population 5. Conclusion References

5. Special Education and Social Services in Korea: Past, Present, and Future

94 95 99 110 115 119

125

Sun Young Ryu 1. Introduction 2. An Overview of Korea 3. The History of Special Education and Family Services 4. Current Status of Special Education and Family Services 5. Discussion and Future Directions 6. Conclusion References

6. Perception, Cognition, and Action: New Perspectives on Down Syndrome

125 126 128 131 139 143 144

147

N. Virji-Babul and D. Weeks 1. 2. 3. 4. 5.

Introduction Action and Object Recognition Interpersonal Interactions Imitation and Gestures The Neurobiology of Down Syndrome: Implications for the Action, Perception, and Cognition Framework 6. Neural Mechanisms Underlying Perception, Cognition, and Action 7. Perception–Action Coupling in DS 8. The Influence of Action Experience on Perception and Cognition 9. Impact on Development and Intervention 10. Conclusion References

148 149 151 152 155 157 158 160 162 165 166

Contents

7. Agenesis of the Corpus Callosum: A Literature Review

vii

171

Shihfen Tu, Donna Doherty, Kathryn J. Schilmoeller, and Gary L. Schilmoeller 1. Introduction 2. Physical/Health-Related Issues 3. Social/Emotional/Behavioral Traits 4. Language/Communication/Cognitive Characteristics 5. Therapeutic Interventions for Individuals with ACC 6. Conclusion and Future Directions References

8. Autism Intervention Research: From the Reviews to Implications for Practice

172 176 180 181 185 187 190

195

Stephanny F. N. Freeman, Tanya Paparella, and Kelly Stickles 1. Introduction 2. Synthesizing the Reviews of Autism Treatments 3. UCLA’s Early Childhood Partial Hospitalization Program: A Pilot Study 4. Integrated Summary References

9. Epidemiology of Mental Illness and Maladaptive Behavior in Intellectual Disabilities

196 197 214 233 235

239

Johannes Rojahn and Lisa J. Meier 1. Introduction 2. Intellectual Disability 3. Psychopathology In Intellectual Disability 4. Epidemiology 5. Risk Factors and Correlates 6. Conclusions References Index Contents of Previous Volumes

240 241 241 243 264 276 280 289 297

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CONTRIBUTORS

Numbers in parentheses indicate the pages on which the authors’ contributions begin.

Emily D’Antonio (93) Department of Psychology, Hofstra University, Hempstead, New York 11549, USA Donna R. Doherty (171) College of Education and Human Development, University of Maine, Orono, Maine 04469-5749, USA Stephanny F. N. Freeman (195) Department of Child Psychiatry, Semel Institute for Neuroscience and Human Behavior, University of California, Los Angeles, California 900241759, USA Roy McConkey (33) Institute of Nursing Research, School of Nursing, University of Ulster, Newtownabbey, N Ireland BT37 0QB Lisa J. Meier (239) Center for Psychological Services, Department of Psychology, George Mason University, Fairfax, Virginia 22030, USA Tanya Paparella (195) Department of Child Psychiatry, Semel Institute for Neuroscience and Human Behavior, University of California, Los Angeles, California 900241759, USA Johannes Rojahn (239) Center for Psychological Services, Professor in the Department of Psychology and Director, School Psychology Program, George Mason University, Fairfax, Virginia 22030, USA Sun Young Ryu (125) Executive Director, Community Advocacy Network, Inc., Elmhurst, New York 11373, USA Gary Schilmoeller (171) College of Education and Human Development, Child Development and Family Relations, University of Maine, Orono, Maine 04469-5749, USA Kathryn J. Schilmoeller (171) College of Education and Human Development, University of Maine, Orono, Maine 04469-5749, USA ix

x

Contributors

Jin Y. Shin (93) Department of Psychology, Hofstra University, Hempstead, New York 11549, USA Kelly A. Stickles (195) Graduate School of Education and Information Studies, Semel Institute for Neuroscience and Human Behavior, University of California, Los Angeles, California 90024-1759, USA Julie Lounds Taylor (1) Vanderbilt Kennedy Center for Research on Human Development and Department of Pediatrics, Vanderbilt University Medical School, Vanderbilt University, Nashville, Tennessee 37210, USA Shihfen Tu (171) College of Education and Human Development, University of Maine, Orono, Maine 04469-5766, USA Naznin Virji-Babul (147) Down Syndrome Research Foundation and Simon Fraser University, Burnaby, BC, V5B 4J8, Canada Patricia Noonan Walsh (33) Centre for Disability Studies, School of Psychology, UCD, Belfield, Dublin 4, Ireland Peishi Wang (69) Department of Education and Community Programs, Queens College, City University of New York, Flushing, New York 11367, USA Daniel J. Weeks (147) Department of Psychology, Simon Fraser University, Burnaby, BC V5A 1S6, Canada

PREFACE

With this volume, Volume 38, I assume the job of general series editor of the International Review of Research in Mental Retardation. Ably edited since Volume 20 by Laraine Masters Glidden, the International Review has continued its role as the intellectual disability field’s most important, prestigious publication devoted to state-of-the-art research reviews. It is today stronger than at any time since its founding in 1966. Now is a great time to accept the editorial baton. Although accepting any baton evokes visceral reactions from my track days many years ago, the baton metaphor also serves to highlight several field-related issues. At the first, most basic level, the passing of the editorial baton highlights continuity with the field’s past. For 43 years, the International Review has published the best of scientific work related to intellectual and developmental disabilities. Over 300 articles have been published in 37 volumes. Virtually every major worker in the intellectual disabilities field has published in its pages, under the direction of three series editors. These editors—Norman Ellis (Volumes 1–11; 1966–1982), Norman Bray (with Ellis for Volumes 12–14, separately for Volumes 15–19; 1984–1997), and Laraine Masters Glidden (Volumes 20–37; 1999–2009)—have carefully, tactfully run the good race. I am beholden to each of them for their record of achievement and for the high esteem in which the International Review is universally held. Accepting the editorial baton also engenders thoughts of the future. Indeed, at various points over the past decade, researchers have questioned the viability of the research field that examines intellectual disabilities. As participants at the annual Gatlinburg or other conferences quickly notice, the field of research specifically devoted to intellectual disabilities is very small, with active researchers probably numbering no more than 300. Issues of tenuous grant support (Baumeister, 1997), an aging corps of researchers, and the need to train and nurture the next generation have all preoccupied researchers both old and new. As the title of a recent article asks, ‘‘Who will lead the mental retardation field in the year 2020?’’ (Havercamp, Tasse, Lunsky, & Garcin, 2003). But as this and other International Review volumes illustrate, the future may not be quite so bleak. Over the past several decades, this small but hardy band of researchers has expanded the nature and scope of disability work. As noted below, this group has become increasingly international and, over time, larger in number. To this more established group, one can xi

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now add a growing cadre of younger researchers. These younger researchers, now knee-deep in balancing new jobs, marriages, children, courses, students, studies, grants, and tenure, are also opening areas that until now have received little attention. Working together, then, the more established and younger researchers are expanding the boundaries of the field itself. Although many measures of such expansion are possible, one simple measure concerns the numbers of issues of established journals and the founding of new journals. As of 2005, the Journal of Intellectual Disabilities Research, the flagship journal of the International Association for the Scientific Study of Intellectual Disabilities (IASSID), expanded from 8 to 12 issues each year and new journals have been founded that connect intellectual disabilities to policy-related research (Journal of Policy and Practice in Intellectual Disabilities, begun in 2004), origins and development of disorders (Journal of Neurodevelopmental Disorders, 2008), psychopathology (Journal of Mental Health and Intellectual Disabilities, 2008), and health (Disabilities and Health Journal, 2008). In becoming series editor, then, I am reminded of Norman Ellis’ (1966) declaration, in the Preface to the International Review’s first volume, that ‘‘Behavioral research pertaining to mental retardation has suddenly burgeoned. Indeed, it seems reasonable to speculate that more research has occurred in this field in the past 10 years than in all previous years’’ (p. vii). So what, exactly, does Volume 38 portend? Although the easy answer is a little of everything, I prefer to characterize this issue as representing a continuum, from the latest expansions of more established areas to initial forays into unchartered territories. In a provocative expansion, Julie Lounds Taylor applies life span development theories to the many changes that families experience as their adolescents with disabilities transition into adulthood. Although transition has existed as a subfield within Special Education for decades, few studies exist on the families of these young adults, even as the transition from school-to-adult services is among the most important changes faced by these young adults and their families. Returning to Erikson and to attachment theory, Taylor explores the ways that understanding normative changes might help us to identify similarities and differences that occur in families when their adolescent with disabilities enters adulthood. Similar expansions—the latest word on more established topics—can be seen as well in other contributions. In their review of intervention approaches to young children with autism, Stephanny Freeman, Tanya Paparella, and Kelly Stickles make the case for more individualized approaches, using as a case example their own Early Child Partial Hospitalization Program (ECPHP) at UCLA. In many ways, Freeman and colleagues are presenting the next generation of intervention approaches in this area, going beyond comprehensive or targeted treatments, intervention- or method-specific approaches to instead advance a more eclectic

Preface

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approach. Featuring their own type of response-to-treatment model, Freeman et al. individualize instruction based on each child’s most recent progress, correcting and changing as they go along. Their initial findings seem especially promising. Other chapters also present the latest extension on more established topics. Naznin Virji-Babul and Dan Weeks go beyond the distinctions among cognition, perception, and action to join these three for children and adults with Down syndrome. Reviewing the latest in brain technology, Virji-Babul and Weeks present findings that may lead to more effective, tailored interventions for these individuals. So too does one see an intervention focus in Johannes Rojahn and Lisa Meyers’ update on dual diagnosis. As we increasingly appreciate that many individuals with intellectual disabilities have both intellectual impairments and psychiatric disorders, an entire subdiscipline—highlighted by Rojahn’s 2008 founding of the Journal of Mental Health and Intellectual Disabilities—has now sprung up to address these issues. If such articles can be considered the latest word on more established topics, so too does this volume include beginning forays into unchartered lands. At its most concrete, the term ‘‘unchartered lands’’ refers to such actual countries as China, Vietnam, and Korea. Presented by younger researchers who are creating a field of disability research where none existed, these three contributions highlight both the differences and similarities of the status of children with disabilities and their families within the context of each society. To choose but one example per chapter, Peishi Wang shows how China’s single-child policy affects children with disabilities, sibling and parent attitudes, and how children with disabilities are perceived in the society; Emily D’Antonio and Jin Shin how the aftereffects of the Vietnam war continue to affect those with disabilities and their families; and Sun Young Ryu how the Korean history of schools has led to relatively advanced services for children with physical disabilities, even as services are more delayed for children with intellectual disabilities. Other examples could be cited as well, highlighting the ways in which the status of persons with disabilities is partly dependent on the specific country and culture. At the same time, however, similarities also exist, both across these three countries and from these to other lands. In all countries, then, mothers of children with (vs. without) disabilities experience higher levels of stress, and issues of poverty and of rural versus urban living enter in. Difficulties in developing sufficient amounts of service (by trained personnel) seem common to all three countries, as is the universal problem in accessing and receiving such appropriate, high-level services by children (or adults) and their families. It is instructive to juxtapose those problems common to only one society and those that seem universal. If indeed 85% of all children with disabilities live outside of Western, industrialized countries—the countries

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in which most research has taken place on children with disabilities and their families—we do well to ‘‘charter’’ these unchartered lands. Less concretely, so too do other contributors venture into unchartered lands. Patricia Noonan Walsh and Roy McConkey, for example, examine the entire issue of the health of individuals with disabilities. Bringing to this issue an international, human rights perspective, Walsh and McConkey describe the nature of health disparities, why they might occur, and how a research agenda is critical to examining and correcting such health inequalities. Although the entire topic of health care seems both complicated and controversial, Walsh and McConkey succeed in joining today’s news to the disability community, both here and across the world. In another foray into unchartered lands, Shihfen Tu, Donna Doherty, Kathryn Schilmoeller, and Gary Schilmoeller examine individuals with agenesis of the corpus callosum (ACC). Although a topic rarely if ever considered within research journals devoted to intellectual disabilities, many individuals with ACC have intellectual and other disabilities. Beyond the basics, Tu and her colleagues describe the many areas of functioning affected by ACC, as well as promising intervention approaches. There you have it: nine excellent contributions, ranging widely in topics and approaches, by established and newer researchers. But before ending, let me add again a few notes of thanks. Having completed one volume as series editor, I have an even greater respect for Laraine Glidden’s decade of hard work for the International Review, her perseverance, and her good humor. To me personally, Laraine has been exceptionally generous with her time, information, and advice. Her major bit of advice—that I will enjoy getting to know and working with contributors who I would never have known otherwise—has already come true. Such relationships, alone, seem worth the price of admission. More subtly, though, I value Laraine’s implicit assertion—to me and to so many others in the field—that I can succeed at this and at other endeavors, and that it is all definitely worth the try. Thanks, too, to Tara Hoey, Mica Haley, Gayathri Venkatasamy, and all of the other seasoned, savvy professionals at Elsevier Press, who have made this rookie editor’s life so much easier. I would as well like to thank reviewers for this issue, especially Nancy Miodrag, Rick Urbano, and Elisabeth Dykens. Last (but in no way least), I thank the nine sets of contributors to this volume. Squeezed into incredibly busy lives, each has completed (and revised) thoughtful, excellent contributions. If it takes a village to raise a child, it takes an international village—of diverse professionals from subdisciplines within and outside of the disability field—to raise a volume. And. . .we’re off. Time to lean into that first turn. . . ROBERT M. HODAPP

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REFERENCES Baumeister, A.A. (1997). Behavioral research: Boom or bust? In W. E. MacLean (Ed.), Ellis’ handbook of mental deficiency, psychological theory and research (3rd ed., pp. 3–45). Mahwah, NJ: Erlbaum. Havercamp, S.M., Tasse, M.J., Lunsky, Y., & Garcin, N. (2003). Who will lead the field beyond 2020? Mental Retardation, 41, 473–476.


C H A P T E R O N E

The Transition Out of High School and into Adulthood for Individuals with Autism and for Their Families Julie Lounds Taylor* Contents 1. Introduction 2. Definitional Issues 3. Theories on the Transition to Adulthood: How Typical Development Can Inform the Study of Families of Adolescents and Young Adults with ASD 3.1. Erikson’s psychosocial theory 3.2. Attachment theory 3.3. Summary 4. What Do We know About the Transition to Adulthood for Individuals with ASD and for Their Families? Review of the Extant Research 4.1. How do individuals with ASD experience the transition to adulthood? 4.2. How do families of individuals with ASD experience the transition to adulthood? 5. Directions for Future Research 5.1. Research grounded in theory 5.2. More systematic investigation of the transition time 5.3. Integration of school, family, and outcome research 6. Conclusions References

2 4

5 5 7 10 10 11 19 24 24 26 27 28 28

Abstract This chapter focuses on the impact of exiting high school for individuals with autism spectrum disorder (ASD) and their families. First, general developmental theories that can assist in guiding research on the transition to adulthood are explained and applied to families of individuals with ASD. Next, extant research on the impact of transition out of high school and into adulthood for individuals *

Vanderbilt Kennedy Center, Department of Pediatrics, Vanderbilt University School of Medicine and the Monroe Carell Jr. Children’s Hospital at Vanderbilt, Nashville, TN 37203, USA

International Review of Research in Mental Retardation, Volume 38 ISSN 0074-7750, DOI 10.1016/S0074-7750(08)38001-X

Ó 2009 Elsevier Inc. All rights reserved.

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with ASD and for their families is summarized. Finally, a number of directions for future research are discussed.

1. Introduction “He lost everything then (when he exited high school). All the training was gone. They had worked on stimming, now it’s back. He had worked part time with a job coach. Now he can’t do that. There is no funding.” - Mother of a 24-year-old son with ASD “I worry about him, where he’ll be in life down the road. Worry about him financially; him gaining friendships and not being lonely.” - Mother of a 19-year-old son with ASD In 2007, the United States Department of Education reported that there were nearly 257,000 children and adolescents in the primary and secondary school systems with a diagnosis of autism spectrum disorder (ASD), a neurodevelopmental disorder beginning before the age of 3 and characterized by impairments in communication and social interaction, and the presence of restricted and repetitive behaviors (IDEAdata.org, 2009). The rapid rise in the number of children diagnosed with ASD began in the early 1990s (Gurney et al., 2003) and children from that generation are now exiting the school system, putting an extreme burden on an already over-taxed adult service system (Howlin, Alcock, & Burkin, 2005). Although this increase in the prevalence of ASD may be due in large part to diagnostic substitution (Shattuck, 2006), nevertheless many of these adolescents and young adults are in need of formal and informal supports during and after transition. As they exit high school and transition to adulthood, individuals with ASD lose the entitlement to many (and sometimes all) of the services that they received while in the school system. As demonstrated by the previous quotes, in most cases these young adults enter a world of adult services plagued by waiting lists and a dearth of appropriate opportunities to achieve a maximum level of adult independence. When services are inadequate, parents often assume primary responsibility for meeting their son or daughter’s needs (Howlin, 2005). As more and more individuals with ASD leave the school system and transition into adulthood and the adult service world, it is critical to understand the factors that promote an optimal transition process. Furthermore, as service providers rely more heavily on parents to coordinate services, it becomes increasingly important to understand how mothers and families are affected by this transition. There is little empirical research addressing how the transition to adulthood might affect families of individuals with ASD; however, the clinical and anecdotal evidence indicates a range of possible reactions.

Transition to Adulthood for Individuals with Autism

3

Some researchers have suggested that the transition out of high school and into adulthood would be associated with considerable stress and anxiety for mothers who have a son or daughter with ASD, due to the loss of services received through the school system (Howlin, 2005). However, my experience speaking to mothers of adolescents with ASD indicates that this may not always be the case. Some of these mothers were looking forward to their son or daughter’s exit out of the school system and into adulthood. Most mothers give two primary reasons for this more positive outlook: (1) their son or daughter felt socially alienated and even rejected in high school, which put a considerable strain on both the individual and the family; and/ or (2) they expected to find a post-high school educational or occupational placement for their son or daughter that might be more appropriate to his or her areas of strengths and difficulties. Regardless of whether these parents are looking forward with anticipation or with dread to the transition to adulthood, it is a salient and important event in the family life cycle. Unfortunately, little empirical evidence can be used to inform these families about what to expect during the transition to adulthood and beyond. In this chapter, I identify theoretical models developed from research on typically developing adolescents and adults that have the potential to inform research on the transition to adulthood for individuals with ASD and for their families, and then interpret the extant literature in light of these theories. I begin by defining the transition to adulthood in terms of typically developing adolescents, and explain the difficulties in applying these definitions to young adults with ASD. Then, I discuss two theoretical models that have the potential to be useful in guiding research related to the transition to adulthood and the ways in which families are affected: Erikson’s psychosocial theory and attachment theory. Further, I summarize the research on the transition to adulthood, first for individuals with disabilities and second for their families. As the vast majority of research on the transition to adulthood for people with ASD has focused on the individual with the disability him or herself, the bulk of this review will focus on the person with the disability. Specifically, I will summarize how individuals with ASD and other intellectual and developmental disabilities tend to experience the transition out of high school and their occupational and educational activities in the years following their high school exit. I will next discuss the more limited research addressing how mothers and families experience this transition. Finally, I will conclude by offering a number of directions for future research. This chapter focuses on families of adolescents and young adults with ASD; however, many of the studies included in the literature review collected data from individuals with other intellectual and developmental disabilities and their families. Although ASD provides some unique obstacles in the transition to adulthood, challenges faced by families of young adults with ASD often resemble those faced by families of young adults

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with other intellectual and developmental disabilities. Therefore, many of the conclusions and recommendations made in this chapter will be relevant to families of individuals with disabilities other than ASD.

2. Definitional Issues Before discussing theories, it is first important to clarify what exactly is meant by “transition to adulthood.” The developmental tasks that indicate a successful transition to adulthood for adolescents without disabilities have been well identified, stemming from Freud’s notion of “love and work” (cited by Hazan & Shaver, 1990). These tasks most often include leaving the parental home, finishing school and starting employment, and marrying and having children (Fussell & Furstenberg, 2005). In recent years, however, the entry into adulthood for typically developing individuals has become much more ambiguous and prolonged and these markers are often achieved in a gradual, disordered fashion (Furstenberg, Rumbaut, & Settersten, 2005). It is increasingly common, for example, for youth to move out of the parental home and into a residential university setting, but then move back into the parental home for a time after the commencement of their university studies. Additionally, it is often the case that men and women are living independently, have finished their schooling, and are engaged in full-time work activities but have not yet married or had children. In the mid-1990s, Arnett (2000) proposed the concept of emerging adulthood as a way to account for this heterogeneity in how individuals transition from adolescence to adulthood. The emerging adulthood period, Arnett (2000) explained, is distinct from adolescence and early adulthood because of its relative freedom from social roles and societal expectations. He suggested that the transition to adulthood is no longer best represented by demographic transitions, such as ending formal schooling, getting married, or having children. Instead, the criteria for the transition to adulthood are individualistic, encompassing concepts such as independence in decision making, being responsible for one’s own person, and financial independence (Arnett, 2000). When the youth in transition has ASD, the complexity implicit in defining the transition to adulthood is multiplied. Some developmental tasks of adulthood are obtained by most individuals with ASD, such as finishing school. Other tasks, such as moving out of the parental home or finding regular employment, are only achieved by a fraction of individuals with ASD in their stated forms, but can be modified to be attainable by many more (e.g., structured or semi-structured living arrangements, supported employment). Finally, some tasks are attained by few individuals with ASD, such as getting married or having children. The criteria


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proposed by the emerging adulthood literature are similarly complicated; many individuals with ASD may never have complete independence in decision making nor have financial independence from both their families and federal or state agencies. Because of these difficulties in definition, many researchers have forgone theory-based ideas of transition and instead defined the transition period for those with intellectual and developmental disabilities using specific ages (such as ages 18–26 in Blacher, 2001). In the next section, I discuss two theories that may be useful in developing a more holistic picture of the transition to adulthood for individuals with ASD; these theories also have the potential to inform research on how parents and families are affected by this transition.

3. Theories on the Transition to Adulthood: How Typical Development Can Inform the Study of Families of Adolescents and Young Adults with ASD 3.1. Erikson’s psychosocial theory Erik Erikson’s psychosocial theory (1950) is one example of a theory of typical development that has the potential to aid in generating hypotheses and interpreting research findings relating to the transition to adulthood for individuals with ASD and their families. In this theory of personality, Erikson proposed eight stages of human development starting in infancy and extending into old age. Each stage focuses on becoming competent in an area of life and is characterized by a developmental “crisis” or conflict. Before the individual moves on to the next stage of development, the previous conflict must be resolved on a spectrum ranging from positive to negative. A positive resolution indicates that the person has acquired the skill or competency of that stage, whereas a negative resolution results when the individual fails to develop the competency. The way that a crisis is resolved at each developmental stage is not only important to that stage, but also to all subsequent stages, as each competency builds on the competencies from earlier stages. That is, a negative resolution to one stage will make it much more difficult to resolve the crisis of a subsequent stage in a positive way. For the current discussion, two of Erikson’s psychosocial stages are most relevant. The first is “identity versus identity confusion,” which takes place during adolescence. As adolescents are exploring their independence and developing their sense of self, they begin to think about such questions as “Who am I, and what is my place in the world?” When this personal exploration is appropriately encouraged and reinforced, adolescents are able

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to choose their sets of values and their vocational goals and develop a lasting personal identity. Having developed a set of personal values and an independent identity allows adolescents to know where they fit into society and to confidently move into adulthood and the next stage of development. According to Erikson (1968), failure to develop an identity leads to confusion and insecurity about future adult roles and results in difficulty with successful resolution of future adult stages. The second of the relevant psychosocial stages is “intimacy versus isolation,” which Erikson (1964) proposed as the major stage of early adulthood. Erikson believed that early adulthood was the time when people began exploring intimate relationships with those outside of their families of origin, and that the development of close, committed relationships with such others was vital to optimal psychosocial development. Those who are able to successfully resolve this phase develop committed and secure relationships, whereas those who are unsuccessful will experience isolation and loneliness. Keeping in mind that the resolution of each stage depends on the previous stage, Erikson believed that young adults who failed to develop a personal identity during the adolescent stage would have a difficult time finding intimacy with others in young adulthood. Researchers have yet to empirically apply Erikson’s stages to those with intellectual and developmental disabilities; however, the concepts inherent in the adolescent and early adulthood stages have been shown to be salient for individuals with ASD. Finding an identity in terms of solidifying adult roles and gaining independence can be difficult for adolescents and young adults with ASD, who seldom live independently and often have difficulty finding and maintaining meaningful employment (Ballaban-Gil, Rapin, Tuchman, & Shinnar, 1996; Billstedt, Gillberg, & Gillberg, 2005; Eaves & Ho, 2008; Howlin, Goode, Hutton, & Rutter, 2004). Furthermore, multiple studies have suggested that these adolescents and young adults tend to have limited friendships and relationships with people outside of their families, with the relationships they do have often lacking in intimacy (Howlin, Mawhood, & Rutter, 2000; Koning & Magill-Evans, 2001; Orsmond, Krauss, & Seltzer, 2004); this leads to greater feelings of loneliness among adolescents and young adults with ASD than among typically developing individuals (Bauminger & Kasari, 2000). Although the pace at which individuals with ASD move through Erikson’s stages may be slower when more severe intellectual disabilities are present, there is no theory or research to suggest that these concepts do not remain important. Using Erikson’s theory as a framework for the transition to adulthood implies that identity and intimacy seeking will be two major components of this transition. If the adolescent with ASD is able to form an identity that is independent from his or her family, particularly in terms of future adult roles and occupations, then he or she will be more likely to develop intimate relationships with others during the early adulthood stage


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(intimacy vs. isolation). According to Erikson, failing to encourage an independent identity may make it more difficult for adolescents and young adults with ASD to develop intimacy with others outside of the family and subsequently may contribute to their future social isolation and to family distress. However, families of individuals with ASD—who are used to protecting their son or daughter throughout childhood—may have a more difficult time than families of typically developing adolescents encouraging independence, especially if the son or daughter has significant functional limitations (Cooney, 2002). Parents of all children use protective strategies to influence the stressful events to which their children are exposed. As children mature, parents move from protective strategies that directly guide or limit their children’s activities, such as participating in activities or direct prevention, to more indirect protective strategies such as being available or soliciting information (Power, 2004). It can be difficult for parents of typically developing children and adolescents to discern how much to protect their son or daughter; parents of adolescents with ASD have the additional complication of weighing their son or daughter’s functional abilities and safety when discerning how much to protect him or her. Parental overprotection can lead to problems adapting to difficult situations for typically developing children (for a review see Power, 2004), and it is logical to think that overprotecting adolescents with ASD—thereby limiting their autonomy—could also lead to problems in adaptation.

3.2. Attachment theory Attachment theory, specifically as it applies to adolescence and early adulthood, can also provide a useful framework from which to consider the transition to adulthood for individuals with ASD and their families. Attachment theory has become one of the most prominent ways to describe parent–child relationships, particularly during infancy and early childhood. According to this theory, the child develops a bond with his or her primary attachment figure (usually a mother) and is able to use that person as a source of comfort and reassurance during times of stress and distress (Ainsworth, Blehar, Waters, & Wall, 1978). In the words of attachment theorists, the child is able to use the attachment figure as a “secure base” from which to explore his or her environment. This bond can vary in quality, contingent on the quality of care that the child has received (Ainsworth et al., 1978; Egeland & Farber, 1984; Pederson et al., 1990). The type of attachment relationships that children have with their primary caregiver can generally be classified into one of four types; this classification is reflective of how readily children can derive comfort from the caregiver when stressed (Ainsworth et al., 1978). Securely attached children use the primary caregiver as a base from which to explore and

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learn from their environments. When distressed, these infants seek comfort from, and are calmed by, their attachment figure (Ainsworth et al., 1978). Children classified as insecure-avoidant actively ignore or avoid the caregiver when they are distressed, whereas insecure-ambivalent children show anger toward and an inability to be soothed by their primary caregiver (Main & Weston, 1981). Finally, infants who display disorganized attachment appear to possess no coherent mechanisms for coping with stress. When they are distressed, these infants either blend contradictory features of several of the above strategies, such as strong proximity seeking followed by strong avoidance, or they appear dazed and disoriented (Carlson, Cicchetti, Barnett, & Braunwald, 1989). Although the concept of attachment was originally conceptualized as explaining the bond between young infants and their primary attachment figures, it has been applied to relationships throughout the life span (Ainsworth, 1985). As opposed to young children who use their attachment figure as a secure base in both a physical and emotional sense (seeking both physical proximity and emotional comfort during times of distress), adolescents tend to use their attachment figure as more of an emotional secure base, not necessarily seeking physical proximity but more emotional comfort during times of trouble. The type of relationship that children and adolescents have with their attachment figures guides their expectations and feelings about significant others during adulthood, referred to by attachment theorists as “internal working models” (Bowlby, 1973). This idea of internal working models has been used to explain adult romantic relationships (Hazan & Shaver, 1987), extending the application of attachment from birth until death. As adults develop new relationships, they rely in part on expectations about how others will treat them based on their past experiences—the internal working model—and they use this model to interpret the behavior of others (Fraley & Shaver, 2000). Therefore, children and adolescents who have a secure relationship with their attachment figure during childhood and adolescence develop the expectation that their caregiver can be counted on when needed and subsequently expect the same consistency and reliability from close adult relationships. Alternatively, if a child learns that caregivers cannot be counted on for support and comfort, he or she will develop an insecure working model of attachment and will expect that same behavior from significant others in adulthood. According to attachment theorists (Allen & Land, 1999), adolescence and early adulthood can best be described as a time of transition; at the start, adolescents are by and large dependent on their primary attachment figures (typically parents) for caregiving. Through a process of autonomy seeking and gradual separation from the family of origin during adolescence and early adulthood, individuals eventually develop attachment relationships with other adults (romantic relationships), as well as the capacity to be attachment figures for their own offspring. Although it may appear that the


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autonomy seeking common to typically developing adolescents implies a breaking of the attachment bond with the parent, in fact, the opposite is true. Adolescents are best able to develop their own autonomy in the context of close, supportive attachment relationships with their parents (Allen et al., 2003; Baltes & Silverberg, 1994). In turn, close, supportive parent–child relationships in adolescence can be predicted from the quality of the parent–child relationship during childhood (Fraley, 2002). Therefore, when considering how individuals experience the transition to adulthood from an attachment perspective, it is important to consider their early attachment relationships, as well as their success in strivings for autonomy. Attachment theory and related issues of autonomy seeking are not only relevant to the transition-aged individual him or herself, but also to the ways that parents experience their son or daughter’s transition to adulthood. The development of autonomy and separating from the family of origin was long thought to be extremely difficult for parents, often described as a period of “storm and stress” (Adelson & Doehrman, 1980; Steinberg, 1990). Current thinking, however, is that “storm and stress” do not necessarily typify adolescence. Although there are certainly increased arguments between adolescents and their parents compared to at younger ages, most parents report a continuity in the relationship quality with their son or daughter throughout adolescence (Steinberg, 1990). Attachment theory would suggest that an important component of how parents experience the transition to adulthood is their early relationship with the son or daughter; a secure attachment bond during infancy and early childhood should be related to a more positive parent– child relationships during adolescence and more productive autonomy seeking by the son or daughter, which in turn leads to a less stressful transition experience for parents. Although there are some complexities when applying attachment theory to adolescents and young adults with ASD and their families, it can still provide a useful framework to think about how these families experience the transition to adulthood. Researchers initially thought that young children with ASD were unable to form attachment bonds with their caregivers at all, even going so far as to describe the lack of a normal attachment relationship as a primary characteristic of autism in the early DSM taxonomy (American Psychiatric Association, 1980; Rutgers, Bakermans-Kranenburg, van IJzendoorn, & Berckelaer-Onnes, 2004). Since then, a number of empirical studies have refuted this idea (Carlson, Sampson, & Sroufe, 2003; Rogers, Ozonoff, & Maslin-Cole, 1993; Rutgers et al., 2004; Shapiro, Sherman, Calamari, & Koch, 1987), finding that very few children with ASD have no relational bond with their caregiver. In fact, it is now generally accepted that children with ASD form bonds with their primary caregiver at the same rates as typically developing children (Rutgers et al., 2004).

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There is a difference, however, in the quality of attachment relationships they tend to have. A meta-analysis of attachment security across studies of children with ASD found that these children were less securely attached than typically developing populations (Rutgers et al., 2004). The estimate from this meta-analysis was that 50% of children with ASD have a secure relationship with their primary caregiver, as opposed to about 70% in a typically developing population (Ainsworth et al., 1978; Crockenberg, 1981; Rutgers et al., 2004). From an attachment perspective, the higher proportion of insecure attachment relationships between parents and children with ASD during childhood may lead to more difficulties in their relationships with their parents and in autonomy seeking during the transition to adulthood. Alternatively, those children with ASD who have secure attachment relationships during childhood will be more likely to have closer parent–child relationships during the transition to adulthood and experience more ease in developing their own autonomy, resulting in a smoother transition experience for parents.

3.3. Summary In sum, theories from a developmental psychology perspective have great promise for guiding research on the transition to adulthood for individuals with ASD and their families. When considering these theories, two main ideas emerge. First, an important aspect of the transition to adulthood for adolescents and young adults with ASD is to develop an identity separate from the family of origin and to develop intimate relationships with those outside of the family. Second, the quality of early and late parent–child relationships can be a key determinant of the degree of success and ease in which individuals with ASD transition to adulthood, as well as how stressful this transition is for families. The next section will review relevant research related to the transition to adulthood for individuals with ASD, focusing on the transition out of high school, and offer suggestions for how to interpret this research in light of the previously discussed theories.

4. WHAT DO W E KNOW A BOUT THE T RANSITION TO A DULTHOOD FOR I NDIVIDUALS WITH ASD AND FOR T HEIR F AMILIES ? REVIEW OF THE E XTANT R ESEARCH Although the transition to adulthood for individuals with ASD is a complex process, the following literature review focuses only on the transition out of high school. I chose high school exit as a key indicator of the transition to adulthood for two reasons: (1) of all developmental tasks of adulthood, it is the most commonly attained by individuals with ASD;


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and (2) nearly all transition studies on individuals with intellectual and developmental disabilities center around high school exit. In contrast to typically developing adolescents in the United States—who exit high school at a prescribed time (at the end of 12th grade)—considerable variability exists in the age at which adolescents and young adults with ASD exit the school system. Some exit with their same-aged, non-disabled peers, while others take advantage of the Individuals with Disabilities Education Act (IDEA) and remain in secondary school until their 22nd birthday. Although it may be simplistic to only consider high school exit as a marker of the transition to adulthood, this milestone provides a focused lens through which to examine the research related to the transition to adulthood for people with ASD or other intellectual and developmental disabilities. The following literature review is divided into two major sections. The first section discusses how individuals with autism experience the transition to adulthood. The majority of this section focuses on objective markers of transition success, such as residential independence, finding employment, and developing friendships with others outside of the family. The second section focuses on families’ reactions to their son or daughter’s transition to adulthood. The preponderance of this research has been done with families whose son or daughter has a developmental disability other than ASD. Therefore, this section of the review will focus on families of individuals with other intellectual and developmental disabilities, ending with a discussion of how this research relates to ASD.

4.1. How do individuals with ASD experience the transition to adulthood? 4.1.1. Objective indices of successful transition: Attainment of developmental tasks of adulthood Nearly all of the research on the transition to adulthood for individuals with ASD has prescribed to the traditional view of transition, focusing on the attainment of developmental tasks associated with adulthood. These studies focus on the degree of independence he or she has in terms of living arrangements, the extent to which the individual with ASD is able to gain and maintain competitive employment, and the presence of intimacy with those outside of the family (Ballaban-Gil et al., 1996; Billstedt et al., 2005; Eaves & Ho, 2008; Howlin et al., 2004). Four major studies, summarized in Table 1.1, have followed children diagnosed with ASD into young adulthood in the United Kingdom, United States, Canada, and Sweden, examining their success in obtaining the developmental tasks of adulthood. Across studies, a uniform picture of the difficulties that individuals with ASD have in attaining these tasks has emerged. Three of the four studies characterized the residential independence of the adults with ASD in their samples. The vast majority of adults were

Table 1.1 Summary of four studies focused on objective markers of the transition to adulthood for people with ASD

Studies

Sample description

Residential independence

Employment

Friendships

Overall success

Ballaban-Gil et al. (1996)

45 individuals with ASD who had been diagnosed in New York, followed up an average of 11 years after diagnosis. Ages 18 to 29 years at follow-up 68 individuals with ASD diagnosed in London between 1959 and 1979. Ages 21 to 48 years at follow-up

2% living independently; 53% living in a residential placement; 44% living with parents

11% competitively employed; 16% in supported employment or sheltered workshop; 73% were not employed

N/A

N/A

4% living independently; 6% in semisheltered placement; 51% in residential placement; 39% living with parents

13% competitively employed; 18% in supported employment or sheltered workshop; 69% were not employment

25% had at least one friendship; 51% had no friends with whom they shared activities; 24% were indeterminate

Types of outcome: 12% very good; 10% good; 19% fair; 46% poor ; 12% very poor

Howlin et al. (2004)

Eaves & Ho (2008)

Billstedt et al. (2005) and Billstedt, Gillberg, and Gillberg (2007)

48 individuals with ASD who had been diagnosed in Canada between 1974 and 1984. Average age 24 years at follow-up Populationbased sample of 120 individuals with ASD diagnosed in the 1980s and living in Sweden. Average age 25 years at follow-up

8% living independently; 35% living in a residential placement; 56% living with parents

56% had ever been employed; 4% competitive employed at interview

33% had at least one friendship; 10% had ever had a romantic relationship

Types of outcome: 4% very good; 17% good; 32% fair; 46% poor; 0% very poor

N/A

N/A

N/A

Types of outcome: 0% good; 8% fair; 13% restricted but acceptable; 21% poor; 57% very poor

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living in some type of supported arrangement. Almost 30% of young adults with ASD in Ballaban-Gil et al.’s (1996) New York sample were living in residential placements outside of the family home, compared to 51% in the United Kingdom sample (Howlin et al., 2004), and 35% in the Canadian sample (Eaves & Ho, 2008). Another common placement was living with families of origin at the time of follow-up, with 39% (Howlin et al., 2004) to 56% (Eaves & Ho, 2008) of young adults having this arrangement. Finally, in all samples, less than 10% of the adults with ASD were living independently, without supports. A particularly pessimistic picture emerges when examining the work activities of individuals with ASD after exiting high school. In the Howlin et al. (2004) sample, just under one-third of the adults with ASD had some type of employment. About 13% were in a competitive employment situation, and another 18% were in a sheltered or supported setting. Similar patterns of employment were found for the Ballaban-Gil et al. (1996) study; 27% of adults had some sort of work activity. About one-half of those were competitively employed, and the other half was employed in supported positions or sheltered workshops. The remaining individuals with ASD had no employment activities at the time of the interview. However, even those who were able to find employment tended to have jobs that were poorly paid and did not provide a living wage. Finally, Eaves and Ho (2008) found that nearly 45% of their sample of young adults with ASD had never been employed. At the time of their follow-up interview, only 4% were competitively employed, with one person in their sample able to financially support himself. Another developmental task of adulthood, friendships and intimacy with people outside of the family, was examined for individuals with ASD who had exited the school system. Eaves and Ho (2008) found that about one-third of the young adults had at least one friendship that involved mutual enjoyment and sharing, and 10% reported having ever had a romantic relationship (although none of the young adults in their sample were currently romantically involved). Parents of over one-half of the adults with ASD in the Howlin et al. (2004) study said that their son or daughter had no friends with whom they shared activities, and 20% said that their son or daughter had at least one relationship that involved some degree of mutual enjoyment and sharing. Finally, three men in this sample (out of 68) were currently or had been married. It is important to note that the Howlin et al. (2004) sample only included those individuals who had an IQ of 50 or above and, therefore, does not represent those who have the lowest levels of intellectual functioning. Perhaps most striking about these three studies is that the difficulties attaining the developmental tasks of adulthood faced by individuals with ASD were consistent both across time (the time between the studies ranged 12 years) and geographic boundaries (the studies took place in


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the United Kingdom, United States, and Canada). In sum, it appears that about 30% of young adults with ASD were employed at any given time, although the proportion approached 50% when considering whether individuals had ever been employed. For those who were able to find employment, jobs tended to be in menial, low-paying jobs. The vast majority of individuals with ASD were not living independently, and most did not have an intimate relationship (friendship or romantic) with another outside of their family of origin. It is important to note that these problems were not limited to those individuals who had severe cognitive limitations; even those adults with average or above-average intellectual functioning found it difficult to attain the developmental tasks of adulthood (Ballaban-Gil et al., 1996). 4.1.2. Multidimensional evaluation of transition success These same investigators have attempted to construct a more holistic representation of how individuals with ASD negotiate the developmental tasks of adulthood, referred to as “adult outcome” in these studies. Eaves and Ho (2008) and Howlin et al. (2004) used identical criteria for adult outcome and classified young adults with ASD according to five dimensions, ranging from “Very Good” to “Very Poor.” The three components that defined a “Very Good” outcome for adults with ASD were regular, unsupported employment, friendships with others, and independent living. A “Good” outcome was defined by having some sort of work activity but requiring a degree of support in daily living and having some friends. A “Fair” outcome was characterized by living at home and/or having some degree of autonomy, but also requiring considerable support in daily living with no close friends but some acquaintances. A “Poor” outcome included those who were in residential placements outside of the home with very little autonomy and no friends outside of their residence, and individuals with a “Very Poor” outcome tended to be living in long-stay hospitals with no friends and no autonomy. Although the samples were quite different, both of these studies reported similar degrees of transition success. About 20% of both samples of adults with ASD achieved good or very good outcomes, 30% had fair outcomes, and about 50% had poor or very poor outcomes. One limitation of the aforementioned studies is that they were based on clinic samples or samples of convenience; it is difficult to determine whether the experiences of these individuals mirror those of the general population with ASD. Billstedt et al. (2005, 2007) addressed this issue by following three population-based samples of children with ASD born in Sweden during the 1980s. They not only used similar domains in their definition of adult outcome, but also included as additional criteria the absence of any other major psychiatric disorder and clear verbal or

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non-verbal communication skills. Overall, their results confirmed the previous pattern of findings. They found that 78% of adults with ASD had poor or very poor outcomes, 13% had restricted but acceptable outcomes, 8% had fair outcomes, and none had good outcomes. These studies, along with a number of others (for a review see Howlin, 2005), suggest that there is great variability in transition success, with the majority of adults with ASD remaining dependent on parents or service providers for assistance in their activities of daily living. 4.1.3. Comparing ASD to other intellectual and developmental disabilities Difficulties in achieving the developmental tasks of adulthood are not limited to individuals with ASD; young adults with other intellectual and developmental disabilities also have trouble obtaining these milestones. For example, Keogh, Bernheimer, and Guthrie (2004) examined the transition to adulthood and attainment of developmental tasks for 20 young adults who had IQ scores of 75 or below, including those with mild, moderate, and severe intellectual disabilities. At follow-up, the mean age of the young adults in the sample was about 22 years. In terms of living arrangements, one-half of their sample was living at home with one person (5%) living independently; the remaining young adults were in group homes or some other supported living arrangement. Two of these individuals (10%) were working without supports, 30% had no employment activities, and the remainder either did volunteer work or worked in an assisted capacity. Kraemer and Blacher (2001) reported comparable employment findings in their study that examined 26 post-high school-aged, young adults with severe intellectual disabilities: 19% were in supported employment, 54% were in sheltered workshops or day activity centers, and 27% had no work activities. Although Keogh et al. (2004) and Kraemer and Blacher (2001) involved small samples that were measured at one point in time, longitudinal study of the transition to adulthood for young adults with disabilities reported a similar pattern of results. Blackorby and Wagner (1996) examined the residential and employment outcomes of 1,990 youth identified with a disability through the school system using the National Longitudinal Transition Study (NLTS). The most prevalent diagnosis in the sample was learning disability (n = 343) although 276 of these youth had a diagnosis of intellectual disability and will be the focus of this discussion. The NLTS collected data at two time points; the first time point occurred 2 months to 2 years after high school exit, and the second 3–5 years after exit. In terms of employment, 25% of the young adults with intellectual disabilities were competitively employed at the first follow-up; this percentage increased to 37% at the second follow-up, when the young adults had been out of


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school 3–5 years. For those who were employed, young adults with intellectual disabilities were much less likely to see an increase in their wages over time compared to those with other types of disabilities (learning disability, emotional disturbance, visual impairment, etc.). A more optimistic picture emerged when examining change in their residential independence over time. At the first time point, only 4% of young adults with intellectual disabilities were living independently; this number increased sixfold at the second follow-up to 24% living independently. In sum, this study suggested that success in attaining adult milestones for individuals with intellectual disabilities was improving over time; however, they continued to lag behind their peers with other types of disabilities and behind typically developing peers. 4.1.4. Predicting objective indices of adult success Another key aspect of studying the transition to adulthood for individuals with ASD is examining the factors that promote a smooth and successful transition. A handful of studies have predicted success in achieving the developmental milestones of adulthood for individuals with ASD from variables measured earlier in the life course. The emphasis of these studies has been exclusively on characteristics of the individuals with ASD themselves. Their results point to more independent adult outcomes for individuals with higher IQ scores and better early language skills (Billstedt et al., 2007; Eaves & Ho, 2008; Gillberg & Steffenburg, 1987; Howlin et al., 2000, 2004; Lord & Bailey, 2002). Knowing the IQ and early language abilities of individuals with ASD may be informative when predicting their adult independence, but this information is less helpful when considering ways to improve the adult success of these individuals. Both IQ and early language tend to be fairly static and difficult to intervene in, especially once individuals with ASD become adolescents and adults. On the other hand, contextual or environmental factors that promote or impede adult success may provide better avenues for intervention, but have been virtually ignored by researchers to date. Frank and Sitlington (2000) conducted one of the only examples of a study that examined contextual factors associated with the successful achievement of adult milestones. These researchers examined the transition out of high school and into adulthood for two groups of individuals with intellectual disabilities: those who graduated in 1985, prior to the passage of the IDEA which mandated transition planning, and those who graduated in 1993, well after the passage of the IDEA. Approximately 1 year after graduation, the class of 1993—compared to the 1985 graduates—was more likely to be employed or meaningfully engaged in activities such as further education or job training. For those who were employed, the class of 1993 was more likely than the class of 1985 to have wages above the poverty level and to report that they had gotten a wage increase in the past year.

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Transition planning within schools is only one of many possible contextual factors that could influence how easily individuals with intellectual and developmental disabilities, including ASD, transition to adulthood. Another factor might be the early parent–child relationship; as already discussed, attachment theory suggests that positive early relationships lead to positive parent–child relationships during adolescence, and in turn a smoother process of separating from the family of origin during the transition to adulthood. It is possible that successful adult outcomes may depend as much or more on environmental characteristics and supports, which are more malleable, as on basic intelligence and language skills (Seltzer, Shattuck, Abbeduto, & Greenberg, 2004). 4.1.5. Subjective indices of success in the transition to adulthood Conspicuously missing from much of the discussion of the transition to adulthood for individuals with ASD and other intellectual or developmental disabilities are subjective indices of success. Although using the attainment of developmental tasks is valuable in that it allows for comparisons across samples over time, this approach ignores many subjective indicators of transition success that are salient to all individuals, including those with disabilities. A small number of studies have measured quality of life and life satisfaction among young adults with intellectual disabilities; these analyses present a more positive view of the transition to adulthood than what would be suggested by the attainment of developmental tasks alone. The Keogh et al. (2004) study added a component of life satisfaction to their data collection, asking the young adults with intellectual disabilities how they perceived their relationships at home, school, work, and other recreational settings. They found that very few expressed any dissatisfaction with their lives, even though they were living fairly dependently. Furthermore, Kraemer, McIntyre, and Blacher (2003) found that young adults with intellectual disabilities who had exited high school felt more empowered in their lives than those still in the school system. These findings point to the importance not only of incorporating a subjective measure of life satisfaction when determining whether or not individuals with ASD have successfully transitioned to adulthood, but also of considering autonomy and empowerment separate from the developmental tasks that are typically used to represent it (residential independence or unsupported employment). 4.1.6. Summary The extant research exploring how individuals with ASD experience the transition to adulthood has focused almost exclusively on the attainment of the developmental tasks of adulthood as indicators of a successful transition. Although some variation exists, according to these criteria most young


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adults with ASD (and with other intellectual and developmental disabilities) are not transitioning effectively. Some of the variation in transition success for individuals with ASD can be attributed to their early IQ scores and language ability, and transition success is likely also related to the quality of services they received within the school system. Using the attainment of developmental tasks as indicators of success may present a pessimistic picture of the transition to adulthood, as those individuals with ASD who have more severe cognitive and functional limitations are unlikely to ever gain the amount of independence needed to meet these milestones. However, even young adults with ASD who have typical intellectual functioning tend to struggle to achieve these developmental tasks. The lack of success that individuals with ASD have in transitioning to adulthood when operationalized in this way is concerning and points to the need to consider alternate approaches when defining this transition. Definitions of the transition to adulthood from a psychosocial or attachment perspective, such as taking responsibility for oneself, having some autonomy in decision making, and developing intimate relations with those outside of the family might better represent whether individuals with ASD have successfully transitioned.

4.2. How do families of individuals with ASD experience the transition to adulthood? 4.2.1. Families’ reaction to the transition to adulthood To date, only one study has examined how parents experience the transition to adulthood for individuals with ASD; therefore, the bulk of this review will focus on families who have a son or daughter with other intellectual and developmental disabilities. The transition to adulthood is a particularly stressful time in the family life cycle for families of individuals with intellectual disabilities. This was demonstrated by Wikler (1986), who used a cohort-sequential design to determine which points in the family life cycle were most stressful for parents of individuals with intellectual disabilities. She found that families whose son or daughter was entering into adolescence showed increased amounts of stress compared to families of younger children, and that stress increased once again when the son or daughter was transitioning out of high school and into adulthood (age 20–21). Furthermore, research suggests that, compared to parents of typically developing children, the stress surrounding the transition to adulthood is heightened when the son or daughter has an intellectual or developmental disability. For example, Whitney-Thomas and Hanley-Maxwell (1996) found that parents of students with intellectual disabilities reported greater discomfort with the transition process and had less optimistic views about their son or daughter’s future compared to parents of typically developing

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young adults. This theme was confirmed in a more recent study by Glidden and Jobe (2007), who examined the transition-related rewards and worries for parents of adolescents and young adults who had been in honors classes, regular classes, and special education classes. Parents of adolescents and young adults in the latter group reported more transition-related worries and fewer transition-related rewards. 4.2.2. Specific stressors and worries related to transition Moving beyond the discussion of whether the transition out of secondary school and into adulthood is difficult for families of individuals with intellectual disabilities, a number of researchers have attempted to identify what, exactly, is the most stressful aspect of this transition. One theme that emerges from this work is the concern about finding appropriate residential and vocational placements. Thorin and Irvin (1992) conducted a study with 42 family members of adolescents and young adults with severe intellectual disabilities who had not yet exited the secondary school system, asking them which transition-related concerns they found most stressful. Family concerns tended to center around residential and vocational issues, with residential concerns such as finding a quality out-of-home placement rated the most stressful, and work life concerns, such as finding and maintaining a quality job placement, rated as somewhat stressful. In addition, families of transition-aged young adults reported being stressed because of financial concerns and problems within the family. Chambers, Hughes, and Carter (2004) mirrored these findings, asking parents of eight students with intellectual disabilities which life domain (employment, residential and daily living, postsecondary education, social relationships, and leisure and recreation) they felt would be most important to the son or daughter with the disability after he or she exited high school. Five out of the eight parents (63%) said that living independently or in a supported placement was most important, whereas 25% (two out of eight) said that finding a job was most important. Small-sample, qualitative studies by Hanley-Maxwell, Whitney-Thomas, and Pogoloff (1995) and Cooney (2002) also revealed the primacy of concerns related to employment and residential services, both before and after high school exit, for families of transition-aged individuals with intellectual disabilities. Cooney (2002) interviewed parents of children with intellectual disabilities who were in their last year of high school. Parents discussed their unfamiliarity with and uncertainty about the adult service world and reported feeling uninformed about post-high school employment and residential options. Results from the Hanley-Maxwell et al. (1995) study suggested that these concerns were warranted. They interviewed mothers of young adults who had already exited the school system, who reported not only an absence of desirable services but often no services at all. Indeed, many of


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these families were on long waiting lists for residential placements, and they cited their lack of options as a significant source of stress. A second theme that emerges from this work is parents’ concern about their son or daughter’s ability to live an autonomous life. Baine, McDonald, Wilgosh, and Mellon (1993) sent a questionnaire of 42 categories of stressors to families of transition-aged individuals with severe disabilities. They asked families to rate the stressfulness of each category of stressor and found that the most severe stressor was dependency of the son or daughter with the disability, operationalized as self-help skills, mobility, and independent functioning. The category rated the second highest in terms of stressfulness was autonomy, such as independent decision making and delayed independence. The concerns found in the Thorin and Irvin (1992) and Chambers et al. (2004) studies were also mentioned by families in the Baine et al. (1993) sample, but tended to be rated as less stressful than more general concerns related to dependency and autonomy. Hanley-Maxwell et al. (1995) and Cooney (2002) explored why concerns about the autonomy of the son or daughter with an intellectual disability cause stress for families. Overall, families described ambivalence about encouraging their son or daughter’s autonomy. They were very concerned with increasing the amount of independence that their son or daughter had, but at the same time were reluctant to relinquish control due to fears about the perceived dangers inherent in adult life for their son or daughter. This concern was echoed in a recent study of 108 parents of adolescents and young adults with intellectual disabilities (Griffin, McMillan, & Hodapp, in press). These parents reported on their concerns regarding postsecondary education programs for their son or daughter with disabilities; 88% of parents reported their primary concern was the safety of the student. In fact, parents were more concerned about the student’s safety than the cost of the program, the focus on employment after program completion, or the similarity of the program to a typical college experience. Overall, these studies suggested that parents of adolescents and young adults with intellectual disabilities who were preparing to exit the school system were highly concerned about finding appropriate residential and employment services for their son or daughter. After exit, adult services remained a source of worry and concern. Parents were also concerned about developing autonomy in their son or daughter, but cited concerns about safety as a barrier. Uncertainty about and dissatisfaction with the adult service world is apt to make it difficult for parents to relinquish control over the activities of the individual with the disability, thereby creating another barrier to the development of his or her autonomy. Interventions aimed at alleviating stress and concern surrounding the transition to adulthood for families of young adults with intellectual disabilities (or ASD) might consider focusing on familiarizing parents with the adult services available to

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them and to the individual with the disability, as well as developing the autonomy of the son or daughter. 4.2.3. Individual differences in families’ reaction to transition Although the transition out of high school and into adulthood appears to be stressful for most families of adolescents and young adults with intellectual disabilities, a recent study by Neece, Kraemer, and Blacher (2009) suggests that there is great variability in how families experience this transition. They interviewed 128 parents of young adults with severe intellectual disabilities who had exited the public school system. The respondents were nearly all biological mothers and the son or daughter averaged 23 years of age at the time of the interview. Neece et al. (2009) asked a number of open-ended questions about the transition process, and then classified mothers as either being satisfied or dissatisfied with the transition process. Approximately 52% of mothers described being satisfied with their son or daughter’s transition out of high school, indicated by parents’ approval of the services their son or daughter received during and post-transition, satisfaction with service providers, mentioning the effectiveness of the transition plan, or expressing general feelings of satisfaction with the process. The remaining 43% (5% could not be classified) was classified as being dissatisfied with the transition process. Mothers who were dissatisfied tended to report poor quality of services, non-existent or insufficient transition planning through the school, inability of the son or daughter to maintain post-high school vocational placements, or general feelings of dissatisfaction related to transition. Parents’ satisfaction with the transition process is important not only in and of itself, but because of the ways in which it affects family and parental well-being. Neece et al. (2009) found that mothers who were satisfied with their son or daughter’s transition to adulthood tended to report high family well-being, whereas those who were unsatisfied with the transition experience reported low family well-being. Similarly, Glidden and Jobe (2007) found that mothers of young adults with intellectual disabilities who reported higher transition-related rewards and lower transition-related worries had higher levels of family well-being and lower depression scores. From these studies, it is impossible to implicate a satisfactory transition out of high school as the causative agent in higher levels of family wellbeing; longitudinal studies that measure family and maternal well-being from before to after high school exit could tease out the direction of effects. From an intervention perspective, it may be important to know whether those families who are functioning better prior to transition will have a better transition experience, or if a better transition experience predicts better family functioning after transition.


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4.2.4. What about ASD? Although researchers have examined the transition to adulthood from the perspective of the developmental tasks attained by individuals with ASD, the empirical literature is nearly silent about how families may be affected by this process. Many of the challenges faced by families of young adults with ASD are similar to those faced by families of young adults with other intellectual and developmental disabilities, such as finding employment opportunities and residential placements that provide a level of support consistent with the individual’s functional limitations. ASD, however, provides some unique challenges in the transition to adulthood, mainly due to the social impairment inherent in the diagnosis. Finding appropriate job placements for somebody who has restricted interests or is not socially motivated may make the transition to adulthood even more stressful for families of individuals with ASD. Furthermore, adults with ASD without intellectual disabilities have difficulty fitting into an adult service system designed to accommodate intellectual disabilities, which may increase the burden on families to find appropriate services and placements. The only study to examine the ways in which families of young adults with ASD react to the son or daughter’s exit from the school system was conducted by Lounds, Seltzer, Greenberg, and Shattuck (2007). This study examined how change in the characteristics of 140 adolescents and young adults with ASD over a 3-year period—half of whom exited the school system over the course of the study—affected subsequent change in maternal well-being. They found that, relative to families in which the individual with ASD remained in the school system over the study period, maternal depression declined and the relationship between the mother and the son or daughter with ASD improved after adolescents and young adults with ASD left the school system. These findings, which ran counter to the authors’ expectations, might reflect the unique social difficulties individuals with ASD have in the secondary school system, relative to those with other developmental disorders such as Down syndrome. Parents may experience relief when a young adult with ASD exits the school system, particularly if he or she is socially isolated or ostracized by classmates. Alternatively, the improvement in well-being might reflect the fact that mothers anticipated this transition with great trepidation, whereas for the most part the young adults in the sample had transitioned successfully. Indeed, one-quarter of those who left high school in the Lounds et al. (2007) sample continued in postsecondary education, nearly one-third had competitive or supported employment, 29% received sheltered vocational services, and only six individuals were without formal day activities. 4.2.5. Summary Not surprisingly, this research suggests that the transition to adulthood for individuals with intellectual disabilities does not happen in a vacuum.

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Instead, beyond the stress at other points in the family life cycle, families experience the transition to adulthood as a particularly stressful time. Such stress is above and beyond that experienced by families of typically developing individuals transitioning to adulthood. The increased stress is mainly due to concerns about finding quality residential and vocational placements, as well as concerns about dependence and autonomy for the son or daughter with the disability. There is also a fair amount of variability in how families experience the transition to adulthood, with some families reporting a smooth, successful transition and others an unsuccessful process. The ease of the transition process is important not only in and of itself but also because it is related to more general measures of parental and family well-being. It is important to note that nearly all of the literature on the family impacts of the transition to adulthood for individuals with disabilities has focused on those with intellectual disabilities, not ASD. The one empirical study in an ASD sample revealed a pattern of findings that were somewhat divergent compared to what would be expected from qualitative and clinical observations. More research is needed to determine whether and how the family impact of transition is different for those who have a son or daughter with ASD compared to other intellectual or developmental disabilities.

5. Directions for Future Research 5.1. Research grounded in theory Future research on the transition to adulthood for individuals with ASD should incorporate the testing of theories developed from populations of typically developing individuals. The vast majority of research in ASD and intellectual disabilities tends not to make use of these theories and, as such, we end up with a fragmented body of knowledge that is difficult to integrate, both within the disability field and also with research on the transition to adulthood among typically developing individuals. Our knowledge about the transition to adulthood for individuals with ASD would be greatly enhanced by the systematic testing of theories broader than one researcher’s specific sample and research questions, so that we can discover under which conditions these theories hold true and under what conditions they do not. By doing research in this way, we can develop a coherent body of findings that has the greatest potential to benefit transition-aged individuals with ASD and their families. The theories discussed in this chapter—Erikson’s psychosocial development and attachment—are two possible examples of theories that could be helpful in our understanding of the transition to adulthood for youths with


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ASD. Erikson’s theory suggests indicators of a successful transition other than the developmental tasks typically used in research with individuals with ASD, such as identity development and intimacy with others. Borrowing from attachment theory, the development of autonomy might be another indicator of a successful transition, and early parent–child relationships could be a mechanism that promotes a successful transition to adulthood. These two theories are by no means an exhaustive list, but instead are a few examples of developmental theories that could be helpful when studying the transition to adulthood for individuals with ASD. Although the attainment of developmental tasks as indicator of the transition to adulthood is informative from both a descriptive and a service provision standpoint, it is inadequate from a theoretical perspective. Living independently, being competitively employed, and involvement in romantic relationships are tasks that are related to the ideas of identity, autonomy, and intimacy discussed by Erikson and attachment theorists; however, these theoretical ideas are much broader than the developmental tasks used to index them. Developing an identity separate from the family of origin and the pursuit and procurement of employment that is consistent with the chosen identity are possible regardless of whether the young adult with ASD is living independently or working in the community without supports. Furthermore, it is possible for young adults with ASD to live quite autonomously even if they co-reside with their parents. In order to truly understand how individuals with disabilities are transitioning to adulthood, it may be necessary to borrow and adapt measures of identity, autonomy, and intimacy intended for typically developing adolescents and young adults, and in some cases to develop new measures that could be used as a complement to developmental tasks. For example, multiple measures of identity development have been validated for use among typically developing adolescents, such as the Extended Objective Measure of Ego Identity Status II (Bennion & Adams, 1986) and the Dimensions of Identity Development Scale (Luyckx et al., 2008). These measures may provide a window into the extent to which higher functioning adolescents and young adults with ASD explore and commit to the development of their identity, as well as how their identity development relates to the attainment of the developmental tasks of adulthood. Similarly, measures of adolescent and young adult autonomy, such as the Emotional Autonomy Scale (Steinberg & Silverberg, 1986), have long been used in developmental psychology. A measure such as this likely could be used without alteration to assess the degree to which higher functioning adolescents and adults with ASD rely on their parents, and could potentially be modified to be applicable to all individuals with ASD, regardless of functional ability. Finally, measures of intimacy should be developed that take into account the social difficulties experienced by nearly all individuals with ASD.

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A broader approach may reveal a more optimistic and perhaps more accurate picture of the transition to adulthood. Based on attaining developmental tasks, it is not difficult to come up with examples of young adults with ASD who appear to be transitioning “unsuccessfully,” but in actuality may be doing quite well in adulthood. A young adult who is living in a group home, working in a sheltered situation, but who thoroughly enjoys the work that he or she is doing, is connected with those around him or her, and is content with his or her life would be judged as having an unsuccessful transition by the developmental tasks. A more holistic view of the transition to adulthood would be garnered by including measures of life satisfaction, identity development, and connectedness with others. In this example we would paint a more optimistic picture of adult outcomes. Measuring constructs broader than attaining the developmental tasks of adulthood when examining an individuals’ transition success may also alleviate some of the bias against a successful transition for those young adults who have more functional limitations. Young adults with ASD who need a substantial amount of assistance in their daily activities are likely not able to be completely autonomous in their living or work arrangements, but may have the ability to develop an autonomous, independent identity. Advocating the inclusion of measures of life satisfaction or identity does not mean to imply that the difficulties faced by individuals with ASD in attaining the developmental tasks of adulthood are not concerning, only that it does not represent the entirety of the transition to adulthood.

5.2. More systematic investigation of the transition time So far, much of the research on transition among families of adolescents and young adults with intellectual disabilities has been qualitative, using very small samples. These studies have been informative, providing a detailed picture of the transition process for the individuals in their samples. A few studies have examined families’ response to transition from a more quantitative perspective; however, all but one of these studies focused on families who had a son or daughter with intellectual disabilities and not ASD. From our current knowledge, it is unclear whether the quantitative studies on the impact of transition among families of adolescents and young adults with other intellectual and developmental disabilities (of which there are few) are relevant to families of individuals with ASD. The social difficulties unique to ASD may make the transition time particularly difficult for parents, especially if they have trouble finding appropriate services and placements for their son or daughter after high school. Alternatively, these same social difficulties and subsequent isolation from peers during high school may make it easier for parents of adolescents and young adults with ASD to let go of services received in the school system, in order to find a vocational placement more appropriate for their son or daughter. Future


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research should take the qualitative findings and quantitative findings from other disability groups and use them to develop generalizable studies of the transition to adulthood for individuals with ASD and their families. Inspired by the extant literature, a number of specific topics could be explored using quantitative methodologies among transition-aged adults with ASD and their families. Qualitative studies found that parents were concerned about the autonomy and dependence of the individual with the disability; studies could measure these factors in young adults with ASD to determine whether, apart from the attainment of the developmental tasks of adulthood, autonomy is important to families. The extant literature also suggests that success in developing adult independence is related to the individual with ASD’s early IQ scores and language ability and is likely related to the types of services he or she received within the school system. There are likely many other, more malleable factors that could be explored as possible mechanisms of promoting a successful transition to adulthood for individuals with ASD and their families, such as the quality of the early parent–child relationship or parents’ encouragement of their son or daughter’s autonomy. These examples are just a few of the possible influences on the transition to adulthood that could be explored using empirical methodology. We know little about how individuals with ASD experience the transition to adulthood or about how families are impacted by this transition. Quantitative, generalizable research has the potential to be of great benefit to these families.

5.3. Integration of school, family, and outcome research To uncover a more holistic picture of how individuals with ASD and their families experience the transition to adulthood, future research must also begin to integrate questions and knowledge from different disciplines. Although there is little ASD-specific research on this transition to date, the studies focusing on families of adults with intellectual disabilities suggest that researchers tend to stay inside of their disciplinary boundaries. Thus, research on transition-related services and school programs is separate from studies of adult outcomes of individuals with disabilities, which tend to be separate from studies examining the families’ experiences of transition. In contrast to how transition is studied, we know that in real life these topics work in tandem. The effectiveness of the transition program in the school may have a major impact on the way that an individual with ASD transitions to adulthood as well as how his or her family experiences the transition. Alternatively, it is impossible to tell the real worth of a transition program without measuring whether it makes a real and lasting difference for individuals with ASD and their families. Similarly, it is important to consider the bi-directional manner in which individuals and families influence each other. The way that an individual

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with ASD transitions to adulthood may have a significant impact on family functioning; conversely, family functioning may be a key determinant of the success with which individuals with ASD are able to transition. We cannot truly understand the effect of transition on families without paying attention to the situations within the schools and the experience of the transition for the individuals with ASD themselves.

6. Conclusions In the past 20 years, much of the research on adolescence in nondisabled individuals and their families has focused on both the transitions that signal the start of adolescence and the transitions that indicate a shift from adolescence to adulthood. Mothers of non-disabled adolescents often find the transition to adulthood to be difficult (Kidwell, Fischer, Dunham, & Baranowski, 1983; Montemayor, Eberly, & Flannery, 1993). Families of individuals with ASD must contend with these normative challenges, while at the same time assuming the role of service coordinator for their son or daughter after he or she leaves school (Howlin, 2005). The situation becomes potentially more serious when considering that, compared to families of children with other developmental disorders, all of this change is occurring among families that tend to be more stressed throughout the life course (Bouma & Schweitzer, 1990; Donovan, 1988; Dumas, Wolf, Fisman, & Culligan, 1991; Holroyd & McArthur, 1976; Rodrigue, Morgan, & Geffken, 1990; Wolf, Noh, Fisman, & Speechley, 1989). Despite the importance of this time in the family life cycle, we know very little about what a successful transition to adulthood looks like for individuals with ASD. We have a limited number of identified factors that can help families predict what to expect for their son or daughter with ASD beyond high school, and we have little to no understanding of how families are affected during this time. Similarly, we are only beginning to identify the factors that will help the transition process go as smoothly as possible for parents of young adults with ASD. Systematic, theory-driven research will help us to understand the characteristics of individuals with ASD and their families that are able to successfully negotiate this transition in the family life course. Such findings, in turn, will help us design more effective interventions and adult services.

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Rodrigue, J. R., Morgan, S. B., & Geffken, G. (1990). Families of autistic children: Psychological functioning of mothers. Journal of Clinical Child Psychology, 19, 371–379. Rogers, S. J., Ozonoff, S., & Maslin-Cole, C. (1993). Developmental aspects of attachment behavior in your children with pervasive developmental disorders. Journal of the American Academy of Child and Adolescent Psychiatry, 32, 1274–1282. Rutgers, A. H., Bakermans-Kranenburg, M. J., van IJzendoorn, M. H., & van BerckelaerOnnes, I. A. (2004). Autism and attachment: A meta-analytic review. Journal of Child Psychology and Psychiatry, 45, 1123–1134. Seltzer, M. M., Shattuck, P., Abbeduto, L., & Greenberg, J. S. (2004). Trajectory of development in adolescents and adults with autism. Mental Retardation and Developmental Disabilities Research Reviews, 10, 234–237. Shapiro, T., Sherman, M., Calamari, G., & Koch, D. (1987). Attachment in autism and other developmental disorders. Journal of the American Academy of Child and Adolescent Psychiatry, 27, 480–484. Shattuck, P. T. (2006). The contribution of diagnostic substitution to the growing administrative prevalence of autism in US special education. Pediatrics, 117, 1028–1037. Steinberg, L. (1990). Autonomy, conflict, and harmony in the family relationship. In S. Feldman, & G. Elliot (Eds.), At the threshold: The developing adolescent (pp. 255–276). Cambridge, MA: Harvard University Press. Steinberg, L. D., & Silverberg, S. B. (1986). The vicissitudes of autonomy on early adolescents. Child Development, 57, 841–851. Thorin, E. J., & Irvin, L. K. (1992). Family stress associated with transition to adulthood of young people with severe disabilities. Journal of the Association for Persons with Severe Handicaps, 17, 31–39. Whitney-Thomas, J., & Hanley-Maxwell, C. (1996). Packing the parachute: Parents’ experiences as their children prepare to leave high school. Exceptional Children, 63, 75–87. Wikler, L. M. (1986). Periodic stresses of families of older mentally retarded children: An exploratory study. American Journal of Mental Deficiency, 90, 703–706. Wolf, L. C., Noh, S., Fisman, S. N., & Speechley, M. (1989). Psychological effects of parenting stress on parents of autistic children. Journal of Autism and Developmental Disorders, 19, 157–166.

C H A P T E R T W O

Inclusive Health and People with Intellectual Disabilities Patricia Noonan Walsh* and Roy McConkey† Contents 1. Introduction 2. Personal Development and Health 2.1. Health and disability 2.2. For good and ill 2.3. Personal fulfillment 3. Health and Intellectual Disabilities: A Contextual Approach 3.1. Health disparities 3.2. Social determinants 3.3. Access to health care 3.4. Building on the evidence base 4. Health Gain 4.1. Changing perceptions 4.2. Inclusive health research 4.3. Person-centered strategies 4.4. Core questions 5. Proposals for More Inclusiveness 5.1. Practical initiatives 5.2. Public health and intellectual disabilities 5.3. Research strategies References

34 36 36 38 38 40 40 41 43 44 46 48 48 50 52 58 59 59 60 61

Abstract Our core argument is that an inclusive approach is required to maximise the health and well-being of persons with intellectual disabilities. For the individual, this means redefining health holistically and not as the absence of illness. Family carers and support staff need to work proactively with empowered individuals in pursuit of healthier lifestyles. Likewise at a national level, public health policy and health promotion strategies need to include the particular challenges faced by persons with disabilities if the latter are to fully share in achieving health gains. * †

Professor Emerita in Disability Studies, UCD, Dublin, Ireland Professor of Learning Disability, Institute of Nursing Research, University of Ulster, N. Ireland

International Review of Research in Mental Retardation, Volume 38 ISSN 0074-7750, DOI 10.1016/S0074-7750(08)38002-1


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Patricia Noonan Walsh and Roy McConkey

The chapter has four main sections. We consider: (1) how health is central to personal development and fulfillment; (2) recent evidence about health and intellectual disabilities, noting specifically findings from studies that have moved away from an over-reliance on biological health; and (3) the emerging interest in finding optimal ways to produce health gain. Finally, (4) we note promising strategies to pursue in the three domains of practice, policy, and research related to health and lifelong development of persons with intellectual disabilities. We conclude that the gains of an inclusive focus on health are for everyone.

1. Introduction Health lies at the core of global policies, and as a topic of research interest, it absorbs many millions of dollars annually (United Nations, 2000). In each country, it fuels national debates on how best to plan and pay for citizens’ health care. Above all, health is prized by most people as fundamental to their personal well-being. Arguably, for all men and women, health is at the heart of efforts to enhance human development itself (Sen, 2002). Health is no less important for people with intellectual disabilities, and perhaps even more vital and full of promise as a critical pathway in attaining a better quality of life. And yet people in this segment of the population are poorer and less healthy than their peers, and further removed from opportunities to develop their capabilities. Historically, disability and illness were often equated, with the result that health among people with disabilities meant remedies for deficits. An emerging view focuses on building their positive health. Recent debates and policies about health inequalities, especially when these are avoidable and thus unfair, mirror changed paradigms in thinking about health and disability. In his conceptual review, Mont challenges the usefulness of a single summary indicator to capture disability. It is not a condition borne by an individual: rather, disability is . . . “complex and multifaceted, with its roots in culture” (Mont, 2007, p. 1662). Disability, then, should not be equated with illness or pathology: health and disability are related but distinct domains. Good health is the rightful life condition of each individual with disabilities, according to the United Nations Convention on the Rights of Persons with Disabilities (2007). And crucially, it is recognized that each person’s health is influenced by environmental factors, according to the new International Classification of Functioning, Disability and Health or ICF, a framework on disability and health articulated by World Health Organization (2001). Arguably, these two dynamic paradigms—one grounded in human rights and the other in a more full appreciation of lifelong functioning—have brought fresh attention to bear from the public health community on the health needs of those who already experience disabilities or who acquire limitations during their lives (Lollar, 2002).

Inclusive Health and People with Intellectual Disabilities

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These radical shifts in thinking about the complex life experiences of people with disabilities within swiftly changing physical, social, and political environments bring in their wake a host of uncomfortable questions. If health must nurture human capabilities, are we obliged to seek optimal ways to promote human development both at a personal level and also globally, so that developing countries can reach for better health outcomes for all their people? What actions most surely target health inequalities—those focused only on persons with disabilities, or more widely dispersed? How might two realms—public health and personal autonomy—find harmony so that the same standards of health and health care are applied to people with intellectual disabilities and the general population? Can health researchers balance the presumed vulnerability of prospective research participants with forceful calls for extensive health research? If economies falter, must governments ration health care—and if so, what currency will people with disabilities spend in an environment charged with tough-minded budgetary decisions? Questions so charged, so fundamental, so compelling, are not the offspring of a single academic discipline, nor are they solely the concern of disability scholars or advocates. Our main purpose in this chapter is to chart avenues for promoting health and well-being among people with intellectual disabilities that will lead to their taking greater responsibility for their health and thus their quality of life. We have adopted the term “inclusive,” even though it refers to a process more commonly applied in other domains where the experiences of people with disabilities have been separate and unequal: for example, education (UNESCO, 2003), employment (Sutton & Walsh, 1999), or community service (Corporation for National and Community Service, 2004). In this chapter, we take it to express the United Nations Convention’s statement on the health of people with disabilities, which affirms that while they may have particular health needs related to disability, they . . . have the right to the enjoyment of the highest attainable standard of health without discrimination on the basis of disability and that they must be provided with . . . the same range, quality and standard of free or affordable health care and programmes as provided to other persons (United Nations, 2007, art. 25). We have adopted the dynamic framework embedded in the ICF (World Health Organization, 2001), which defined disability so as to avoid two fallacies—that it is solely a medical problem or, at the other extreme, the result of social construction alone (Leonardi, Bickenbach, Ustun, Kostanjsek, & Chatterjee, 2006). If the ICF is truly universal, then this structure will create enough space to consider how health is related to the lifelong development and functional participation in society of persons with intellectual disabilities. In the sections that follow we consider: (1) how health is central to personal development and fulfillment; (2) recent evidence about health and intellectual disabilities, noting specifically studies that have moved away from an overreliance on biological health; and (3) optimal efforts to

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produce health gain. Finally, (4) we suggest strategies to pursue in the three domains of practice, policy, and research related to health and lifelong development of persons with intellectual disabilities.

2. Personal Development and Health More than 60 years ago, the Universal Declaration of Universal Human Rights proclaimed that . . . Everyone has the right to a standard of living adequate for the health and well-being of himself and of his family, including food, clothing, housing and medical care and necessary social services (United Nations, 1948, art. 25). Grounded in human rights, the emerging capability approach to human development takes as its pertinent goal the possibility for people to function in areas of central importance, among these bodily health, or . . . being able to have good health, including reproductive health; to be adequately nourished; to have adequate shelter (Nussbaum, 2007, p. 23). Is this approach to development as pertinent for persons with intellectual disabilities? To explore this question, it is important to consider how health and disability are related, whether health policies should embrace good as well as ill-health and how individuals with intellectual disabilities themselves appraise their own health as an element of personal well-being.

2.1. Health and disability Despite a wealth of research evidence about the health of people with intellectual disabilities—for example, systematic inequalities in their health and access to health care (Krahn, Hammond, & Turner, 2006)—recent findings represents a new body of knowledge in the wider domain of public health (Drum, Krahn, & Bersani, 2009). It is sobering to recall that health and disability were de-coupled conceptually only in the recent past. Heretofore, the presence of disability was equated with disease, both burdens so undesirable that thinking of the good or poor health experiences of people with disabilities was simply not developed (Rock, 2000). A (bio) medical model previously defined disability as an observable deviation from biomedical norms, arising from disease, trauma, or other health condition (Bickenbach, Chatterji, € un, 1999). Nosek conjured the stereotypical image of . . . sick Badley, & Ust€ people in wheelchairs covered with blankets . . . to depict this blurred notion (1992). In order to measure health and disability at both individual and population levels, the World Health Organization over many years of consultation developed its ICF (2001). The framework is dynamic, as it . . .. takes into account the social aspects of disability and does not see disability only as a ‘medical’ or ‘biological’ dysfunction. The impact of the environment on a person’s functioning may also be recorded, as the ICF includes contextual factors that . . . represent the complete background of an individual’s life and living. They include two components: Environmental Factors and Personal Factors, which may


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have an impact on the individual with a health condition and that individual’s health and health-related states (World Health Organization, 2007b, p. 15). Refining the ICF definition in the interests of greater clarity, Leonardi et al. (2006) proposed a definition of disability that highlights its dynamic, interactive nature and gives equal importance to the individual’s health as well as environmental factors in mediating the experience of disability: Disability is a difficulty in functioning at the body, person, or societal levels, in one or more life domains, as experienced by an individual with a health condition in interaction with contextual factors (p. 1220). When this view of disability is coupled with the force of the United Nations Convention on the Rights of People with Disabilities, then promoting optimal physical and mental health of people with disabilities throughout their lives is a priority in order to minimize their disabilities. The interactive framework of ICF leaves space for individual differences to emerge—for example, in responses to adversity that are manifested in personal resilience (Goodley, 2005). Nor is health a black hole defined by the absence of—not light, but—illness. The long-standing World Health Organization (1948) definition of health remains highly pertinent: . . . health is a state of complete physical, mental and social well-being and not merely the absence of disease or infirmity (World Health Organization, 1946). There is ample evidence that people with intellectual are nonetheless likely to incur secondary conditions and experience poorer mental and physical health, as do individuals with other disabilities. Secondary conditions are defined in Healthy People 2010 as . . . medical, social, emotional, family, or community problems that a person with a primary disabling condition likely experiences (U. S. Department of Health and Human Services 2000, chap. 6). Many conditions are preventable if appropriate, timely action is taken. The inclusion of disability in a major statement of national health policy, one of whose aims is to eliminate health disparities among segments of the population, indicates that the health of individuals with disabilities has become an explicit focus of public health attention (Wilber et al., 2002). Arguably, a focus on the biology of disability has hitherto limited our understanding of social and environmental health determinants for people with intellectual disabilities. Yet there is much we still have to learn about enhancing the physical and mental well-being of people who may be biologically vulnerable to ill-health, who do not readily promote their own health, and whose health care may falter due to poor communication, unreliable access to health services, or inexpert responses from health professionals (Krahn et al., 2006). The framework outlined in the ICF (World Health Organization, 2001) indicates that multiple, complex factors

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are at work at all levels—personal, social, and environmental—to yield poorer health outcomes for people with disabilities. Understanding the interplay of these elements is likely to demand a public health approach—that is, one that aims to enhance the well-being of all members of society through the improved health of a whole population, irrespective of its diversity.

2.2. For good and ill Public health policies, however, must embrace both health and illness. Measuring and targeting secondary conditions on behalf of persons with disabilities also falls within the remit of public health, as . . .. traditional public health functions (assessment, policy development, and assurance) are applicable to preventing and reducing secondary conditions associated with unnecessary activity limitations, health costs, lost wages, reduced participation and quality of life (Centers for Disease Control, Factsheet). Arguably, the positive or negative valence of an individual’s state may be distributed on two axes—being healthy (or ill) and being well (or unwell). Good mental or physical health may or may not accompany being “well,” or being ‘ill’. For example, medical interventions that focus on the removal or prevention of illnesses may not in themselves address the broader health issues that beset people with intellectual disabilities. Keyes (2002) coined the term “flourishing” to describe the positive mental health status of individuals; the opposite of which he labeled “languishing.” People in the latter group did not have a mental illness per se but they had low “well-being”; their life seemed empty and stagnant. In a large-scale survey of over 3,000 adult Americans, he found that languishers were six times more likely to have major depressive episode than those who were flourishing. Furthermore, Keyes stresses the social dimension to positive health as in a person’s social integration, social actualization, social acceptance, and social contribution. This conceptualization could be applied to people with intellectual disabilities, although as far as we are aware this has not been done in any formal way. It is our guess that proportionately more people with an intellectual disability would be rated or may rate themselves as “languishing” compared with their nondisabled peers and that this group would show a heightened risk for episodes of ill-health compared to those who were deemed to be flourishing. But whether or not this is so, the distinction between health and illness remains pertinent especially when it comes to investigating strategies for health promotion and health gain for persons with intellectual disabilities.

2.3. Personal fulfillment The United Nations (2007) Convention rests on core principles, one of which is each person’s dignity and autonomy, the freedom to make her or his own choices. Thus, if signatory governments indeed . . . recognize that


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persons with disabilities have the right to the enjoyment of the highest attainable standard of health without discrimination on the basis of disability (United Nations, 2007, art. 25), it follows that people with intellectual disabilities will become recognized as experts in giving a personal account of their own health and its potential impact on their capabilities. Ideally, their narratives should complement the evidence gained from large-scale, systematic studies. Relying on individual experience is by no means a threat to gaining coherent, broad-based health knowledge on how health care should be refined and made available at the population level. Krahn and Drum (2007) reviewed a decade of literature on policy-based practice to improve access to health care for people with intellectual disabilities. They concluded that better surveillance and measurement—as well as direct participation by people with intellectual disabilities—are necessary to advance knowledge in this area. However, while direct involvement by individuals is laudable, it may not suffice in improving health care, according to a systematic review of 42 studies about involving patients in the general population in their own care. The authors of this review concluded that the initiatives contributed to changes in service provision, but the effects of these on quality of care have not been reported (Crawford et al., 2002). A distinction between self-reported health and presence of functional difficulties was apparent in wide-ranging interviews with 29 older women with intellectual disabilities in Ireland and the United States (LeRoy, Walsh, Kulik, & Rooney, 2004). While most of the women indicated that their health was good, and good when compared with that of other women their age, slightly over half of the respondents said that problems with their health prevented them from doing some activities on a daily basis. Their stories shed light, too, on how living environments may exert a blanket influence on healthy behaviors day after day, with little room for individual choice. As one older woman living in a group home commented on the food she typically ate . . . I eat what my housemother cooks and when she says to eat it (LeRoy et al., 2004, p. 435). Confidence in relying on people with intellectual disabilities themselves as reliable informants has been shored up elsewhere, as Havercamp, Scandlin, and Roth (2004) comment, and cite they previous studies in which health data gathered for large-scale surveys were self-reported. But although health is a cardinal element in measures of quality of life, relatively few studies have examined self-reports of health among adults with intellectual disabilities. Lunsky, Emery, et al. (2002) compared self-reports of 77 adults with mild intellectual disabilities from supported living settings in the United States and Canada on health behaviors, somatic complaints, and medication usage with reports from residential staff. The two groups of informants agreed about many health behaviors and major medical disorders, but staff tended to report fewer minor symptoms of illness. These authors proposed that information from both individuals and staff can help to identify health issues as well as areas for health promotion.

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Brown and Brown (2003) applauded efforts to heighten the importance of considering quality of life among people in health and medical treatment, as this approach moves away from eradication of disease and focuses instead on what individuals wish and the impact of health care on their lives. Although a laudable target, there is as yet no well-founded association that links personal satisfaction with health, health outcomes, and personal fulfillment among people with intellectual disabilities. In the next section we consider evidence that is not overreliant on biological health, and sensitive to the wider context in which people with intellectual disabilities pursue healthy lives.

3. Health and Intellectual Disabilities: A Contextual Approach To date, research in health and intellectual disabilities has been dominated by three main themes, albeit within an illness rather than a health focus. These are health disparities, social determinants of health, and access to health care. Previous literature reviews and journal articles noted in this section build a convincing picture of the health disadvantages of people with an intellectual disability.

3.1. Health disparities Disparities—chiefly negative—are evident when people with intellectual disabilities are compared with their peers in the general population (Fisher, 2004; Krahn et al., 2006; Linehan, Walsh, van Schrojenstein Lantman-de Valk, Kerr, & Dawson, 2009; Ouellette-Kuntz, 2005; Scheepers, Kerr, et al., 2005; van Schrojenstein Lantman-de Valk & Walsh, 2008; Walsh, 2008). Overall different patterns of health need in the two groups contribute to this imbalance. People with intellectual disabilities have a different patterns of health need. For example, epilepsy, gastro-oesophageal reflux disorder, sensory impairments, osteoporosis, schizophrenia, dementia, dysphagia, dental disease, musculoskeletal problems, accidents, and nutritional problems are all much more commonly experienced. Conversely, health problems related to smoking, alcohol, and use of illegal drugs are uncommon (Cooper, Melville, & Morrison, 2004, p. 414). Some reservations need to be entered: direct comparisons are often not made between samples drawn from the same population. Usually, the incidence rates for populations of those with intellectual disabilities are contrasted with rates available in published literature. This makes confounding variables harder to detect such as lower socioeconomic status and access to health


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services (van Schrojenstein Lantman de Valk, 2005). In their review of comparative studies that identify health problems among people with intellectual disabilities, Jansen, Krol, Groothoff, and Post (2004) observed that most are based on comparisons with reported prevalence rates of general health surveys: only a few studies include a control group of individuals without intellectual disabilities. But in the Netherlands, van Schrojenstein Lantman-de Valk, Metsemakers, Haveman, and Crebolder (2000) were able to compare patients with and without intellectual disabilities who were recorded on the same general practice database: those with intellectual disabilities were found to have 2.5 times more health problems. Large-population studies have distinctive value and surpass samples based on biomedical causes alone in developing public health plans. For example, Chapman, Scott, and Stanton-Chapman (2008) analyzed a large statewide-linked birth and public school record database (N = 327,831) in Florida, USA. They found that low maternal education was associated with the largest effects at the population level for mild and moderate/severe mental retardation (intellectual disability). It was concluded that a broader perspective recognizing the interactive complexity of risk factors is needed, and that good surveillance data will lead to more effective prevention and improved targeting for screening and intervention.

3.2. Social determinants Recently, the social determinants of ill-health have loomed even larger in weight within prosperous as well as low- and medium-income countries. These factors—early life experiences, social support, work, and food, for example—are encountered by all humans (Fig. 2.1). However, their relevance to the health of people with intellectual disabilities has yet to be fully understood (Emerson, Felce, McConkey, & Walsh, 2008). Although socioeconomic position is the core determinant of health, a priority for researchers is to unify evidence in this domain with knowledge about the health of people with intellectual disabilities (Graham, 2005). For these men and women, their greater propensity to ill-health has often been attributed to biological differences associated with an intellectual disability, such as genetic influences or additional impairments. This bias has reinforced the emphasis on medical interventions, largely drug treatments. Only latterly has attention shifted toward social and economic determinants of health, with confirmation that the same factors are likely to impact on people with intellectual disabilities as for the nondisabled population. In a recent review, Emerson (2007) concluded that the association between poverty and intellectual disabilities accounts in part for the health and social inequalities experienced by people with intellectual disabilities and their families. Indeed, social circumstances generate a double jeopardy—they increase both the likelihood of the disability and the likelihood of ill-health.

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Social gradient Stress

Transport

Food

Early life

Health Addiction

Social exclusion

Social support

Work Unemployment

Figure 2.1 Poor social and economic circumstances affect health throughout life. Adapted from Wilkinson, R., & Marmot, M. (2003). Social determinants of health. The solid facts. Copenhagen: WHO Regional Office for Europe.

Emerson and Hatton (2007a) investigated the social determinants of health status among people with intellectual disabilities by carrying out a crosssectional survey of self-reported health among 1,273 English adults with mild or moderate intellectual disabilities. They found that indicators of socioeconomic disadvantage accounted for a significant proportion of variation in health status—apart from any variation that might be attributed to participants’ characteristics or living circumstances. A measure of hardship, an indicator of socioeconomic disadvantage, was associated with self-reported health. Elsewhere, Emerson and Hatton (2007b) reported the findings of secondary analysis of data on a nationally representative sample of 12,160 British children aged under 17 years. It is one of few such studies that attempt to investigate the impact of socioeconomic position on health— presented in Fig. 2.1 for the general population (Wilkinson & Marmot, 2003)—and gave evidence for health inequalities experienced by people in the segment of the population with intellectual disabilities. Social context, specifically geographical area, potentially heightens understanding of the emotional and behavioral needs of children and adolescents with intellectual disabilities. Emerson, Robertson, and Wood (2007) completed a cross-sectional survey of teacher-reported needs among 386 South Asian children and 118 Black children in a deprived urban conurbation in the United Kingdom. Teachers reported higher rates of emotional and behavioral needs among Black (when compared with South Asian) children. Increased rates of emotional and behavioral needs were


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generally associated with older child age, lower child ability, the absence of sensory impairments and male gender. Neighborhood deprivation was associated with variation in emotional and behavioral need, although patterns differed for the two ethnic groups of children.

3.3. Access to health care A third current theme is access to health care, and a number of factors have been identified as potential barriers for people with intellectual disabilities in achieving equity of access to health-care services. These include the characteristics, living circumstances, and previous experiences of the person with intellectual disabilities, the abilities of professionals, the structure of health service systems, and negative attitudes toward people with a learning disability (Baxter & Kerr, 2002; Cumella & Martin, 2004; Meehan, Moore, & Barr, 1995). Poorer access means that patients do not get the health care that they require. Comparison to the general practitioner consultation rates of patients with other chronic conditions would seem to indicate that, among people with intellectual disabilities, contact with primary care doctors may not be commensurate with need, concluded Felce, Boxter et al. (2008b). Available evidence suggests that, to date, mainstream health services have failed to meet the needs of people with intellectual disabilities (Balogh, Ouellette-Kuntz, Bourne, Lunsky, & Colantonio, 2008). Unsurprisingly, the reasons for poor access to health care arise from many tributaries—individual, environmental, and systemic. 3.3.1. Individual differences • A person with intellectual disabilities may not be able to communicate needs and symptoms, such as pain and discomfort which may be compounded by sensory impairments. For example, a person who experiences somatic symptoms may manifest physical problems as challenging behavior or a loss of daily functioning, thus impeding identification of underlying health problems (van Schrojenstein Lantman-de Valk & Walsh, 2008). • People with intellectual disabilities supported by paid carers who are recently employed may not be able to recount a medical history. • Fear of unknown places and investigations, or past negative experiences leading to noncooperation on the part of the person with intellectual disabilities. This may be exacerbated by challenging behaviors, such as hyperactivity, shouting, or self-injurious behavior. • Difficulties in undertaking full physical examinations because of physical impairments of people being examined or treated. Overall, access to health care is especially complex for persons with intellectual disabilities, with individual, social, situational, and environmental factors playing their part in how fully or reliably information is shared.

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3.3.2. Social environment • Failure of family carers or paid carers (e. g., service staff) to recognize when health care is needed and maybe a reluctance to seek medical or nursing attention when required. • Difficulties of health-care professionals in assessing a person’s needs owing to insufficient training, equipment, or motivation. • Medical terminology not explained in simple terms to people and their carers. 3.3.3. Health systems • Uncoordinated systems of health care which people often have to navigate on their own. • Lack of specialist health screening services for people with intellectual disabilities that permit more thorough investigations.

3.4. Building on the evidence base A robust evidence base supports the conclusion that there is more than one pathway which results in poor access to health care. Added complexity comes from three sets of factors—individual differences, features of the social environment, or the design of health systems—that may be expressed singly or in combination with others. Authors of the 2007 National Healthcare Disparities Report defined people with disabilities—one of the priority populations identified—as individuals having physical, sensory, and/or mental health conditions associated with a decrease in functioning in daily activities. It was striking that the presence of disability interacted with membership in other groups, to yield poorer access to health information. The authors of the Report have charged a special group to advise on measures of disabilities from existent data that could track disparities in quality and access to care for individuals with disabilities for the National Healthcare Disparities Report (NHDR) and that would be comparable across national surveys (U. S. Department of Health and Human Services, 2008, p. 227). A particular challenge to strengthening the evidence base is that many data collections either do not capture disability or do not do so in the same way. Arguably, persons with intellectual disabilities are even more likely to experience such disparities, and yet knowledge of how myriad factors interweave and thus influence access to health care remains limited. Lack of optimal measures may contribute to the limitations in present knowledge. Commenting on the specific health problems specific to particular populations: the NHDR authors note that . . . people with disabilities may face barriers in getting access to care and experience differences in quality of care that are not captured by data because of the limitations in the survey instruments (p. 146).


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A case might be made to gather further evidence on these three sets of factors at individual, societal, and health systems levels in relation to health care among persons with intellectual disabilities, and also among persons in particular subgroups (such as older persons or those with particular syndromes) or to examine variations across different settings (e. g., people living independently). We contend, however, that future research would be better directed at exploring the changes required in support services to address these issues, consolidated at individual, regional, and transnational levels (Krahn & Drum, 2007). 3.4.1. Implications for individuals One priority is the development of reliable mechanisms for ongoing identification of health needs and changes over time, through screens and health checks. Screening may be especially valuable at times of transition. Lennox, Rey-Conde, and Cooling (2006) found that the Comprehensive Health Assessment Program (CHAP) health review (see Section 4.4.4) was effective in identifying a number of health issues in adults in Australia who were moving from institutional care into the general community, where they would then seek ongoing health care from a general practitioner. Documenting the availability and uptake of health services for the individual will help to avoid fragmented information. The development of person-held health action plans holds promise. Curtice and Long (2002) in the United Kingdom describe a “health log” for people with intellectual disabilities that is designed to provide continuity in their health care when they move across systems or when support staff change. 3.4.2. The implications for service systems within localities and regions Here the focus needs to be on identifying the factors that account for inequitable access to services and health prevention/ and health promotions initiatives as a means of reducing intracountry variations. 3.4.3. Transnational comparisons There is a continuing need to facilitate learning across different service systems and economies with a greater emphasis on cultural variations in health outcomes. These comparisons will also illuminate the difficulties in obtaining reliable and valid measures within and across countries, and also the gaps in data gathering that have become apparent in past studies—for example, the need to include measures of social deprivation.

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Albeit arduous, the search for reliable methods to gather comparable health information is worthwhile, if it permits comparisons between people with intellectual disabilities and their peers in the general population. Partners in two European Union (EU)-funded public health projects, Pomona (2002–2003) and Pomona II (2004–2008), developed an evidence-based set of health indicators pertinent to people with intellectual disabilities but devised to permit comparisons between data about this segment of the population with data gathered in health surveys—for example, about the presence of epilepsy, prescribed medications, or body mass index (BMI)—of the general population (Linehan et al., 2009). The group operationalized these indicators in a health surveillance tool, translated the survey tool into 13 European languages, and gathered health data about 1,200 individuals in 14 countries (Walsh, on behalf of the Pomona group, 2008). Findings demonstrated that indicators are useful in gathering comparable health data across countries, and also in making it possible for national governments to compare adults with intellectual disabilities with their nondisabled peers. But, well-founded and timely as it may be, health information is at best necessary, if not sufficient, in yielding meaningful health gain.

4. Health Gain A compelling question has emerged from recent research that deserves much greater attention than it has received thus far, namely what is needed to produce significant health gains for people with intellectual disabilities? Admittedly, research undertaken on the three themes reviewed above has contributed some answers to this question through identifying factors that are associated with poor health; the presumption being that the removal of negative factors produces better health. In broad terms, there is supporting evidence that this does happen. For instance, the dramatic increase in the life expectancy of people with Down Syndrome in affluent countries over recent decades, as detailed by Janicki, Dalton, Henderson, and Davidson (1999), is usually attributed to better access to modern medicines or surgical procedures. Indeed, the extra years lived by people in this segment of the population of some countries over the past generation is striking. Roizen and Patterson (2003) report findings of a large-scale study of people with Down syndrome in the United States that showed an increase in median age of death from 25 years in 1983 to 49 years in 1997. But medical treatment alone may not be the sole reason for a longer life span. There are likely to have been other factors at work, some of which could have had an even more crucial role. An instance is the stark reduction


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since the 1950s in the numbers of children with Down syndrome who were institutionalized from birth and who were spared the increased risks of infections as well as other threats to their well-being which were a feature of institutional care (Fryers, 1986). The opportunity to grow up in a loving family home is certainly a well-known contributor to healthy lives. This social change may indeed provide a fuller explanation for people with Down syndrome having longer lives, in that it was their parents who advocated for them to access life-saving medical treatments which did not often occur during the institutional era. Variations in longevity trends are likely to depend on complex factors, including available forms of support— or their lack—and living conditions for individuals and their families across countries. The serious deficiency of services and resources allocated to the care of persons with intellectual disabilities globally, especially in most lowand middle-income countries, well illustrates this phenomenon (World Health Organization, 2007a). Two lessons are worth drawing out from this analysis. First, investigations into health gain must move beyond testing simple associations—if X happens does health improve? Rather a range of potential influences have to be considered using multivariate and/or multiphase research designs so that the probability of competing explanations can be determined but more crucially so that the impact on people’s health of combinations of factors can be examined. In the Down syndrome example, increased longevity likely results from a combination of family care and access to modern medicine— plus other factors as well! The second lesson is the need to look beyond the obvious if we are to fully understand the complexity of improving people’s health. The link between a reduction in obesity levels with better nutrition and more exercise is well established for the general adult population (Martinez, Kearney, Kafatos, Paquet, & Martıńez-Gonzelez, 1999) with children (Janssen et al., 2005), and with people who have an intellectual disability (Rimmer & Yamaki, 2006). Further research that simply replicates these findings is not a priority. Rather the research agenda needs to center around the prevention of poor nutrition and lazy routines, and the promotion of healthier lifestyles with those at risk. And we need a better understanding of why certain subgroups within the rubric of intellectual disability face particular health challenges, such as reducing obesity among people with Prader–Willi syndrome (Holland, Treasure, Coskeran, & Dallow, 1995). Viewed in this way, there may indeed be specific factors, over and above those that apply to everyone, that require attention in order to produce health gains for people with intellectual disabilities. In summary then, we have limited understanding of how to produce sustained and significant health gains among persons with intellectual disabilities. We propose a two-pronged research strategy for better health. One approach is to embed intellectual disability research within health

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research initiatives targeting the general population and wider service practices. The second is based around a more focused, person-centered strategy. Ideally, both are required, as they are complementary. But the adoption of either strategy needs to be underpinned by changed perceptions around health.

4.1. Changing perceptions We have already stressed the need to promote health as more than the absence of illness but there are two further underlying presumptions that may need to be challenged when it comes to producing health gains. In themselves, they provide fruitful research topics. First, a passive acceptance of illness and chronic conditions in persons with intellectual disabilities persists. By contrast, proactive, preventative approaches based around individual empowerment and self-determination are central to most modern endeavours in health promotion (Marks, Sisirak, & Heller, 2009; Minkler, 1994). Such a change presages a profound shift in perceptions and beliefs about persons with an intellectual impairment. Arguably, a rights-based perspective that emphasizes equality of access to health care is slowly taking hold within modern disability services, although our understanding of the processes that nurture changes in the perceptions of influential people is limited by the lack of empirical enquiry around their perceptions of disability. We suspect that in the arena of health promotion such research is of particular significance, especially with health personnel in mainstream services but also with people who have been labeled as having the disability and their frontline carers. One outcome of changed perceptions is that people with intellectual disabilities gain equitable access to all public health initiatives and are not sidelined from them. Second, health gain cannot be seen as the responsibility of designated specialists. Instead, it must be owned as a shared responsibility across a range of stakeholders who are prepared to work together on common goals. While medical, nursing, and therapeutic personnel may be seen as having a particular contribution to make to health gain in terms of their specialist knowledge and skills, we suspect that the efficacy and the sustainability of their interventions is dependent on the involvement and commitment of other supporters and service personnel and of course, the individual person. Yet research to date has rarely explored the means for bringing about cooperative working.

4.2. Inclusive health research To date, there has been a paucity of research that considers the health needs of people with intellectual disabilities within the broader context of improving the health and well-being of all citizens. This dearth perhaps


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reflects the relative “invisibility” of people with intellectual disabilities in the public health domain. Typically, population-based health surveys in the United States (Hodapp, Urbano, & So, 2006) as well as in countries of the EU (Linehan, Walsh, et al., 2009) may not readily identify members of this segment of the population, despite growing evidence about the health inequalities experienced by people with intellectual disabilities. In particular, there has been little comparative health research, notably with other vulnerable and marginalized groups in society. Although this may identify specific differences among the ID population, common needs and impediments to health improvements are likely to be highlighted through the multivariate analyses that larger sample sizes permit. Such information will strengthen the case for interventions that might have been dismissed as only applying to a specific population, such as people with intellectual disabilities. A second research strand is to investigate whether strategies that have been successful in producing health gain for the general population are applicable to groups within that population who have additional needs, albeit with some adaptation. Such research would help to establish the robustness of the intervention and better inform improvements to its efficiency and efficacy which will benefit all beneficiaries. Moreover, this research should result in more comprehensive theoretical models and explanatory frameworks. The smoking cessation program adapted and evaluated by Tracy and Hosken (1997) is an example of this type of research. An even more ambitious research agenda involves longitudinal studies of people’s health and well-being across the life span. Such research is wellestablished for the mainstream population, notably children, but we have little comparable data for those with intellectual disabilities across the life span. Longitudinal studies would provide valuable insights into predisposing and protective factors related to good health that people with intellectual disabilities share with others but also those that are especially pertinent to them. Despite ample evidence that socioeconomic position is the fundamental determinant of health, Graham (2005) commented that researchers have to date maintained boundaries between the intellectual disability and public health domains. But exceptional studies have emerged to heighten understanding of how population-wide determinants impact on the lives, particularly on their physical and mental health, of people with intellectual disabilities. For example, Emerson and Hatton (2008) examined the contribution of socioeconomic position to the health and mental health status of children and adolescents with intellectual disabilities in a sample of 10,438 British children. Intellectual disability was a significant risk factor for poorer general health, emotional disorders, and conduct disorders. Between-group differences in socioeconomic position and household composition, however, accounted for 24% of the increased risk of poor health, 23% of the increased risk for conduct disorder, and 37% of the increased risk for emotional disorder. These authors concluded that

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socioeconomic disadvantage may account for a significant proportion of the increased risk for poorer health and mental health of children and adolescents with intellectual disabilities. Perhaps the future will see a move toward alliances and consortia that bring together researchers from different disciplines and interests. A shift in focus would facilitate shared expertise on the specific type of health gain that is sought—such as cancer prevention, diabetes management, or social connectedness—rather than discrete populations. Collaboration would also provide greater opportunities to investigate the intersection between specific populations and health improvements, which is a large gap in our present evidence base. Overall, sampling strategies to include participants with intellectual disabilities require more planning and greater effort that is often not required of health researchers studying the general population. Sadly, this seems to result in their preference to omit these “abnormal” participants because of the difficulties they pose for sampling. Equally much of our present research with persons who have intellectual disabilities is done with relatively small convenience samples that may not represent the totality of persons subsumed under the label of “intellectual disability.” This risk is particularly great in comparative studies of people with intellectual disability and nondisabled or other disabled groups. Unrepresentative sampling can lead to spurious conclusions. All of which is a reminder that scientific study of population health is still in its infancy.

4.3. Person-centered strategies Another approach to understanding people’s health is to focus on individuals rather than group characteristics. This is the second strategy to studying health gain among persons with intellectual disabilities, with a focus on three interrelated core questions: • How might individuals take more responsibility for their own health and well-being? • How might the people’s supporters become active health promoters? • How might public health services adjust to meet the needs of people and their supporters? In this respect, generating health gain has much in common with creating development in other aspects of people’s lives, notably in their personal care, education, and obtaining employment. It is likely therefore that the core strategies that have been effective in these areas can be applied to the field of health. These provide a useful starting point but they should be validated through evidence-gathering and critical appraisal. We can identify five strategies for which there is a prima facie case for their adoption.


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4.3.1. Identification of individual need This applies not only to the person’s particular health needs which the various screening instruments described earlier can quickly identify but the assessment must extend to their individual living circumstances, their lifestyles, and access to health services. Moreover, this needs to be a ongoing process as new health needs invariably arise (Felce et al., 2008) 4.3.2. Person-centered approaches and personalized supports Any interventions to promote health gain need to be personalized to the individual and crucially the necessary supports should be identified and provided. It is the latter that is the most commonly given reasons for plans not achieving their goals (Robertson, Emerson, et al., 2006). 4.3.3. Information must be accessible With people who have pronounced literacy and communication difficulties, extra efforts have to be made to ensure information relating to health promotion and access to health services is easily obtained and understood by the person’s carers but also by individuals. As we will see, advances in information technology may help here. 4.3.4. Health promotion must be integrated into people’s lifestyles This familiar adage for promoting better health is more difficult to apply given the lifestyles of many people with intellectual disabilities. But unless health promotion is woven into the fabric of ordinary living, the impact of extraordinary interventions will not be sustained. A particular instance is the sedentary life that most people with intellectual disabilities lead and its detrimental health correlates (Varo et al., 2003). Both personal and environmental characteristics are likely to contribute to a lifestyle marked by relative inactivity. Data gathered from 500 adults with intellectual disabilities in various residential settings in the United Kingdom suggest that physical inactivity was associated with lower ability and more restrictive settings (Robertson, Emerson, et al., 2000) 4.3.5. People’s social, emotional, and spiritual well-being needs attention as well as their physical health This truism, too, often expresses the holistic goals of modern service provision. But a quality of life focus is a reminder that improved physical health per se is only one element in health gain and indeed better physical health and less illness may result from improvements on other life domains (Felce, 1997).

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4.4. Core questions This listing reinforces the importance of promoting people’s health as part of their everyday supports and not relegating health improvement to a distinct service or set of health personnel. However, these broad strategies need to be refined in terms of the three core questions posed earlier in this chapter: developing greater personal responsibility for one’s own health and well-being; enlisting personal supporters as active health promoters; and adjusting public health services to meet the needs of people with intellectual disabilities and their supporters. 4.4.1. Promoting personal responsibility for health Given common perceptions of people with intellectual disabilities and their dependence on others to aid their understanding and decision-making, this aspect of future research enquiry may seem less plausible than others. However this is another example of the need for changed perceptions. The wider research literature on self-determination, advocacy, and selfefficacy for persons with intellectual disabilities contains important insights to inform health-related research (Algozzine, Browder, Karvonen, Test, & Wood, 2001). Promising lines for future research include the following. 4.4.1.1. Understanding people’s perceptions of health and illness We have a dearth of research that describes how people with intellectual disabilities view health and illness. As the U. S. Surgeon General’s Closing the Gap report (2002) noted: As a society we have not really been listening and paying attention to them. We have been too likely to expect others to speak their needs . . . we have not found ways to empower them to improve and protect their own health (p. v). Fender, Marsden, and Starr (2007) used focus groups to produce a userled health assessment that was feasible and acceptable to implement and had a greater emphasis on physical fitness than traditional disease-based measures. Kiefer (2008) argues for the need to identify what individuals perceive to be the major facilitators and barriers they perceive to their individual health and wellness. These insights can make an important contribution to our understanding about personal motivation to change health-related behaviors. 4.4.1.2. Person-held health records In their review of research into person-held health records, Kaelber et al. (2008) were confident that the greater deployment of these systems will


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beneficially impact health-care costs, quality, and efficiency. When introduced for patients with long-term mental illness, people became more involved in their care and the communication among professionals increased (Stafford & Laugharne, 1997). Electronic recording systems with visual and auditory facilities will make such plans more accessible to people with poor literacy skills. 4.4.1.3. The development and implementation of the educational interventions designed to increase people’s understanding and to promote behavioral change There are a growing number of such programs. For example, Heller, Hsieh, and Rimmer (2004) organized a 12-week, 3 days per week, exercise and health education program for 32 adults with Down syndrome, with a “no intervention” contrast group. Compared to controls, the training group showed significant changes in attitudes toward exercise, including increased exercise self-efficacy, more positive expected outcomes, fewer cognitive–emotional barriers, improved life satisfaction, and marginally lower depression. Similarly Lunsky, Straiko, and Armstrong (2003) described an 8week curriculum, “Women Be Healthy,” developed for women with intellectual disabilities to help them become more equal partners in their health care. The curriculum included psychoeducation, coping skills training, exposure to the medical setting, and assertiveness training. Twenty-two women completed the program and they showed significant improvements in health knowledge, health behavior beliefs, and coping strategies following intervention, and maintained some of these treatment gains (health knowledge and health behavior beliefs) at a 10-week follow-up. These small-scale studies and others like them could be replicated within support services once the program content and leadership are made available. Adaptations that increase their efficacy with particular individuals would be worth documenting and sharing. 4.4.1.4. Developments in information technology The Internet, coupled with mobile phone technology, can now be combined to provide individualized training and support programs for persons on health issues. O’Hara, Seagriff-Curtin, Levitz, Davies, and Stock (2008) have demonstrated the use of personal digital assistants to improve the oral health of adults with mild-to-moderate intellectual disabilities notwithstanding some technical difficulties encountered; notably keeping the batteries charged. Similar programs could be used to assist people to devise nutritious menus and maintain an exercise regime that is tailored to their particular needs. These strategies will become more economical with

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technological advance and users’ growing familiarity with computers and mobile phones. 4.4.1.5. Use of peer models and advisers Stock et al. (2007) used older children (Grade 7) to teach younger ones (from kindergarten upward) about nutrition, physical activity, and healthy body image. Both groups showed significant gains in knowledge, behavior, and attitude scores; along with lower increases in BMI and weight with age. Likewise, a review of peer-led, school-based health programs suggested they were at least as effective, if not more so, than adult-led lessons (Melanby, Rees, & Tripp, 2000). Similar cotutoring approaches have been undertaken with people who have an intellectual disability but await evaluation (e. g., Marchand-Martella et al., 1992). 4.4.2. Health literacy All of these approaches combine to promote greater health literacy of people with intellectual disabilities. Nutbeam (2000) stresses the importance of health literacy as an outcome of health promotion. The World Health Organization (1986) defines it thus: Health literacy represents the cognitive and social skills which determine the motivation and ability of individuals to gain access to, understand and use information in ways which promote and maintain good health. Nutbeam (2000) notes that transmitting health information is a fundamental task, more is required to improve health literacy in a population. In addition, people need to be helped to develop confidence to act on the knowledge they are given as well as the ability to work with and support others. He argues that health literacy will best be achieved through more personal forms of communication and through community-based educational outreach. The latter observations are especially pertinent to people with intellectual disabilities and other socially disadvantaged groups. But reliance on mass media may also explain the apparent failure of many health promotion campaigns to impact on such groups. Dedicated health promotion efforts seem optimal for people with intellectual disabilities. These should focus on . . . physical fitness, weight control, smoking cessation, emotional support, and disease prevention. Empirical research is needed to find educational approaches that are effective for individuals with developmental disabilities, many of whom may not understand traditional strategies such as television or radio public service announcements and printed materials (Havercamp et al., 2004, p. 425). A health handbook for people with intellectual disabilities presents a well-rounded


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curriculum and invites learners to play an active part (Marks, Sisirak and Heller, 2009). Other supporters, such as care staff and family members, can promote their health actively. In the future, greater research efforts need to be directed at improving the health literacy of people with intellectual disabilities so that they can become more responsible decision-makers with respect to their own health and well-being. 4.4.3. Supporters and carers as active health promoters Given the reliance of people with intellectual disabilities on others for assistance and support across many aspects of their life, the relative lack of research on the impact that family carers and paid supporters can have on people’s health is surprising and requires attention. Certainly, reviews of obesity interventions have identified the motivation of carers as a critical feature in the effectiveness of weight loss interventions (Hamilton, Hankey, Miller, Boyle, & Melville, 2007). Some of the topics mentioned in relation to people with an intellectual disability will be applicable to their carers, and should be done with their full cooperation and engagement. Our goal should be to make carers feel empowered and competent in the role as health promoters. But there are three issues that deserve greater attention. 4.4.3.1. The lifestyles of carers There is abundant literature that describes the poorer health and increased stress experienced by mothers particularly when their son or daughter displays challenging behavior (Eisenhower, Baker, & Blacher, 2009) which seems to be compounded by economic hardship (Olsson & Hwang, 2008). McGrother, Hauck, Bhaumik, Thorp, and Taub (1996) reported that these carers had 40% more limiting health disorders compared to the general population, with depression almost four times more common among female carers. More recently, Singer (2006) concluded that while a meta-analysis of 18 studies found that most mothers of young children with developmental disabilities were not depressed and that depression is not an inevitable consequence of parenting children in this group, there is nonetheless a serious problem to address. Paid support staff, too, seem to experience greater stress than other health-care workers and the low status of their work is a potential stressor (Hatton et al., 1999). To date, no research seems to directly explored the impact of care-givers’ well-being on the people for whom they care, or more pertinently, their capacity to become health promoters when their own well-being is compromised by their caring role. Might health promotion endeavors directed at carers’ health lead to concomitant improvements in the people for whom they care? This is a plausible hypothesis worth testing. Of course, improvements to carers’ health could include

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interventions designed to reduce the burden experienced by carers and to improve their economic position (Krahn, Hammond, & Turner, 2007). Interventions are more likely to take place if planned on an individual basis within the limits of existing policies, although ultimately reforms of national health and social services may be needed. 4.4.3.2. Meeting carers’ needs A second theme is around the education and training that is made available to carers. Possible topics could include undertaking simple health checks, determining when referrals are necessary, and monitoring compliance with recommended health interventions. There is evidence that carers often fail to take these simple actions although the reasons for disregarding them are not clear. The accessibility of written information provided to carers is an issue that affects them as well as the persons they support, especially as carers too may have poor literacy skills. Certainly the lack of accessible information about services does account for the lower uptake among certain family carers. Porterfield and McBride (2007) used data from the 2001 National Survey of nearly 40,000 children with special health-care needs to demonstrate that lower-income, less-educated parents were less likely to report that their children needed special services and these factors, coupled with insurance coverage, determined their use of them. They surmised that family-centered support and better education of carers would be an appropriate policy response, but further research could usefully validate the form these interventions need to take. Family caregivers are often marginalized by health-care professionals and denied access to information that they may need to do a good job. The unmet needs of family caregivers are of wide importance, and have been explored by bioethicists, as well as service providers and researchers from the intellectual disability field. Levine (2008) argues that ... family caregivers’ willingness to help does not remove all responsibility from policymakers, nor from health professionals, community organizations, and society in general. Family caregivers are not resources to be used until exhausted; they are true partners in care. Their mental and physical health and well-being are legitimate causes for concern for bioethicists, public health officials, and medical professionals (p. 65). 4.4.3.3. Creating community action There may be lessons to be learnt from attempts to promote the health of ethnic communities. Taylor (1998) identified the importance to increased physical activity of using community advisory panels, community need assessments, and community members delivering the intervention. Likewise, group-based approaches could prove effective as they help to build a common ownership, sense of solidarity, and shared purpose across carers and people with intellectual disabilities.


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4.4.4. Increased access to public health services The third theme related to improved health gains for people with intellectual disabilities is better access to public health services, notably primary care doctors and acute hospitals. There is growing evidence that the identification of health problems through regular health checks undertaken by primary care nurses and doctors does result in improved physical health, although mental health problems may remain unrecognized and untreated (Cooper, Morrison, et al., 2006; Martin, Roy, & Wells, 1997). The latter study offered a health screen intervention to one group of adults with intellectual disabilities to determine whether health gains would be detected after 1 year: they concluded that their intervention yielded sustained benefits—for example, a higher level of meeting health promotion needs—in outcomes compared with standard treatment alone that had been offered to control participants. Challenges for providers are likely to arise if health checks are transformed into sustainable programs of health promotion. In their study, Felce, Baxter et al., (2008a) suggested that these interventions need to be conducted regularly as new morbidity was identified through subsequent checks. Further, lessons learned in studies of the general population may inform those providing health promotion services to people with intellectual disabilities. Park et al. (2009) found in their analysis of health screening data in South Korea that persons with severe disabilities (physical, neurological, or sensory) were less likely to take part in mass screening programs. The barriers to accessing appropriate medical attention in acute hospitals have also been documented, particularly in the United Kingdom. Such barriers include long waiting times, the unfriendliness of others; communication difficulties and lack of opportunity to be consulted about examinations and treatments conducted on them (Sowney & Barr, 2006). Some key issues for future research include the following. 4.4.4.1. Training for mainstream health-care providers Krahn and Drum (2007) note the dearth of training in the United States of health-care providers in relation to people with intellectual disabilities. Melville, Cooper et al. (2006) developed a training intervention aimed at primary care staff (mostly practice nurses) consisting of an information pack and a face-to-face training session. Participation in the two components of the training intervention was associated with significantly greater change in participants’ ratings of their knowledge and self-efficacy than those receiving the training pack alone. Lennox et al. (2007) in Australia demonstrated the efficacy of a CHAP that guided general practitioners to work with adults who had an intellectual disability and their carers in gathering a systematic medical history, a guided health review and the development of a health action plan. Compared with a control group, there were marked increases in

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the detection of hearing and visual impairments, in uptake of immunizations, and health screening for women. It remains to be seen if these training interventions aimed at primary care personnel result in sustained health gains for people with intellectual disabilities. Put positively, how might this form of training be designed and implemented so that this outcome is achieved? A related question is how these initiatives can move from demonstration projects at a local level to being implemented nationally. A further question is, can successful interventions be embedded in training curricula? A survey carried out by partners in 14 European countries who led the Pomona II project (2005–2008; www.pomonaproject.org) suggested that training of medical doctors and other health professionals varies very widely in substance and extent across Europe. Only a few reported well-established specialist curriculum content, and some respondents indicated that in their countries medical students were unlikely to have any substantive instruction in how to work with patients with intellectual disabilities. 4.4.4.2. Creating seamless health services and supports The more complex health needs of people with an intellectual disability can bring them into contact with a greater range of health professionals who work in diverse health settings. Among the problems encountered are communication breakdowns, duplication of assessments, and uncoordinated interventions (Abbot, Watson, & Townsley, 2006), all of which have implications for the cost-effectiveness of health services. To date, little research has been undertaken into organizational issues that affect the delivery of health services to vulnerable groups. However, it is likely that local partnerships provide a promising way forward given the intractable problems associated with implementing national changes. The wider engagement of people with intellectual disabilities with public health initiatives and health-related research opens new territory, full of promise for personal development and better quality of life. A fresher, ecological perspective on health outcomes recognizes that interventions must be finely tuned as well as timely. While gains in individuals’ health status are primary, short-term goals, changing the lifestyles, and living conditions which determine health status are considered to be intermediate health outcomes (Nutbeam, 1998).

5. Proposals for More Inclusiveness A deeper understanding of how people with intellectual disabilities interact with family and friends, society, formal service systems, and the wider environment in pursuit of good health is a first step in devising


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effective interventions. In this final section, we give examples of how researchers, practitioners, and policymakers stand to gain from sharing knowledge and developing collaborative efforts and propose further steps toward greater inclusiveness of people with intellectual disabilities in health-related initiatives.

5.1. Practical initiatives How feasible or effective is it for specialists from both the health and intellectual disability sectors to work alongside each other, as has been proposed? In recent years, Special Olympics has played a unique role in pairing direct support to athletes with research endeavors, building on their success in providing local opportunities for people with intellectual disabilities in many countries to take part in sport in their communities. The organization has also promoted training for health professionals. But while providing a high-quality service to individuals and families, and promoting training for health professionals, it also acts as a catalyst for transnational research in low-income as well as high-income countries. Efforts to promote healthy athletes have also supported valuable research initiatives that have yielded, for example, health screening data about thousands of participants (Special Olympics, 2005). A related achievement has been marked progress in developing reliable systems to gather health information on large numbers of participants. The knowledge accrued in this way can help to determine whether health status and standards of health care are the same for people with intellectual disabilities and their peers in the general population. Is it possible that similar initiatives can be routinely become part of other forms of services, such as supported employment?

5.2. Public health and intellectual disabilities Public health and disability have long been separate policy streams. Guidelines for the general population may recognize disability as a health-related demographic characteristic of some individuals in the target group, or perhaps a risk factor for shorter life expectancy. It is less common for whole-population statements to embed the health needs of people with intellectual or other lifelong disabilities across all themes and issues—nutrition, for example. A recent national policy document on men’s health in Ireland (Department of Health and Children, 2008) illustrates a persistent practice. It states that Irish men comprise a diverse population, noting also that men are prone to accidents or sports injuries that may give rise to disability. But it does not specifically refer to the distinctive physical and mental health needs of men with intellectual disabilities. Yet evidence suggests that gender differences in health problems and access to health care within this segment

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of the population are important. In a UK study of gender differences among adults referred to specialist mental health services in the United Kingdom, personality disorder was more common among men, who were more likely than women to be referred through generic mental health services (Tsakanikos, Bouras, Sturmey, & Holt, 2006). In the light of the ICF’s (World Health Organization, 2001) interactive framework it is timely to ask if a newly established vision expressed in inclusive initiatives such as Healthy People 2010 in the United States, might be implemented more widely so as to embrace people with intellectual disabilities from the start. One way is to capture the expertise and energy of those in the public health domain and respond to the fresh challenges posed by promoting health among people with intellectual and other disabilities (Lollar, 2002). Efforts to promote health among people with intellectual disabilities need not supplant, but rather can complement, traditional approaches focused on illness: Rimmer (1999) charted a paradigm that replaces disease prevention with the active prevention of secondary health conditions among people with disabilities. On the other side of this coin, experts in the intellectual disability field may share their store of knowledge about appraising readiness to change, curriculum design, and program evaluation with those charged with advancing health among diverse populations (Marks et al., 2009).

5.3. Research strategies Two topics may be especially pertinent to gather knowledge about whether public health policies can increase health gain among people with intellectual disabilities. First, health economics policies form part of the socioeconomic environment. In volatile economic period, challenges are bound to recur: will budget cuts lead to rationing health services? Will it be perceived that longevity has a price too high when allocating costly care to older people with intellectual disabilities? Research around social and political decision-making and the impact on the health of people with intellectual disabilities has scarcely begun. Second, in widening public health initiatives at a local level, health promotion activities may be extended to include people with intellectual disabilities. Research that compares the outcomes for all participants in the host region or neighborhood would be particularly informative. To carry out research studies in these and related areas, a variety of expertise will be required. The health agenda is broader than intellectual disability specialist agenda, and it is likely that we must apply methods that are not only traditional psychological or empirical methods, but cross-national and longitudinal. Greater inclusiveness in studies of the health of people with intellectual disabilities may be achieved by applying methods already available. For instance, in reviewing evidence about presumed acquiescence among


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people with intellectual disabilities in interview situations, Finlay and Lyons (2002) stated that their purpose was to increase .... both the validity and inclusiveness of self report scales and interviews with people who have mental retardation. These authors suggest that familiar tactics—using simple sentences, permitting the interviewee to express uncertainty and checking answers—can help to reduce acquiescence. Would-be researchers seeking funding for studies must confront two major challenges. The first is to combat the widespread tightening of funds earmarked for health and human services, the first claimants on national budgets. The second is to find a way to carry out inclusive health-related research in a funding climate well matched to that purpose. The recent emergence of academic centers naming both health and disability as their focal areas is a positive sign of collaborative intent. Each of these strategies may serve a single health research priority for all persons, whether or not they experience intellectual or other disabilities: health protection. In the United States, Gerberding (2005) names the new research imperative as protecting health, and urges bold steps to . . .. rebalance the current investment portfolio. Health protection must be prioritized ... (p. 1403). This author specifically proposes multidisciplinary, collaborative research that considers multiple individual and contextual determinants and their interactions. Its ultimate aim is to provide evidence that will guide complex decisionmaking and improve people’s health. A more unified approach to building knowledge and protecting the health of all can potentially help people with intellectual disabilities to gain health and develop their capabilities. In conclusion, we have argued throughout this chapter that adopting more inclusive approaches to health policies and research can help to lessen the negative inequalities that people with intellectual disabilities experience. Many of the tools to promote more seamless strategies are already in our grasp but they await more systematic implementation by professionals, by carers and with people labeled as disabled. Evaluating the comparative outcomes in health gain achieved by people with intellectual disabilities and the general population may serve not only as measures of health status, or health protection, but also as a means of advancing human development. The gains of an inclusive focus on health are for everyone.

REFERENCES Abbot, D., Watson, D., & Townsley, R. (2006). The proof of the pudding: What difference does multi-agency working make to families with disabled children with complex health care needs? Child and Family Social Work, 10, 229–238. Algozzine, B., Browder, D., Karvonen, M., Test, D. W., & Wood, W. M. (2001). Effects of interventions to promote self-determination for individuals with disabilities. Review of Educational Research, 71(2), 219–277.

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Persevering in the Face of Hardship: Families of Individuals with Developmental Disabilities in the People’s Republic of China Peishi Wang* Contents 1. Introduction 2. Background 2.1. Current statistics on individuals with disabilities in China 3. Educational Systems 3.1. Disability legislation 3.2. Special education services in China 3.3. Learning in regular classrooms 4. Chinese Families of Individuals with Disabilities 4.1. The concept of family in the Chinese context 4.2. Stigma 4.3. Literature on Chinese families of individuals with disabilities 4.4. Siblings of individuals with disability 5. Conclusion References

70 70 70 73 73 74 74 77 77 78 80 87 88 89

Abstract In 2006, it was estimated that China has approximately 83 million individuals with disabilities, which is about 6% of the general population. However, little is known about the families of these individuals. This chapter provides an overview of literature on family research in the People’s Republic of China. Only 16 peer-reviewed studies in both English and Chinese were found for this review. The paramount need of Chinese families of children with developmental disabilities is access to free and public education. More empirical studies are

*

Department of Educational and Community Programs, Queens College, City University of New York, Flushing, New York 11367



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needed to assist policy makers in developing educational and social service infrastructure to effectively serve these individuals with disabilities.

1. Introduction One new child is born with a serious disability every 40 s in China—that is, 2,000 new cases every day and 700,000–800,000 new cases every year (Stratford & Ng, 2000). The Second National Sample Survey on Disability, which took place in 2006, indicated that there are approximately 83 million persons with different types of disabilities in China (China Disabled Persons’ Federation, 2008a). As the largest developing country in the world with an estimated population of 1.3 billion (CIA, 2009), China is facing a monumental task in rehabilitation services and education for people with disabilities. The aim of this chapter is to familiarize readers with research on families’ experiences raising a child with developmental disabilities in the People’s Republic of China. In order to help readers understand and appreciate the experiences of these families, some background information on the current conditions of individuals with disabilities in China is provided at the beginning of the chapter. This is followed by a brief discussion about the current educational system for students with disabilities and their impact on families. Next, a review of literature on family research in both Chinese and English language is presented. Finally, implications and recommendations are discussed for future research.

2. Background Before the early 1980s, discriminatory terms such as “can fei,” which means “the handicapped and useless,” were used to refer to individuals with disabilities. During the last two decades, with China’s rapid economic growth and social development, and the active advocacy of the disability community, today “can ji ren” (“persons with disabilities”) is commonly used both in official documents and by the general public.

2.1. Current statistics on individuals with disabilities in China The Second China National Sample Survey on Disability took place from April 1 to May 31, 2006 (National Bureau of Statistics of China, 2006). The survey covered all of China’s provinces (with the exception of Taiwan), autonomous regions (except Hong Kong and Macao), and municipalities. It is estimated that China has 83 million persons with different types of

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Chinese Families of Individuals with Disabilities

Table 3.1 Categories and percentages of individuals with disabilities in 2006 in China

Disability categories

Estimated total (million)

Percentages

Physical disability Hearing disability Multiple disability Visual impairment Mental illness Intellectual disability Speech disability

24.12 20.04 13.52 12.33 6.14 5.54 1.27

29.07 24.16 16.30 14.86 7.40 6.68 1.53

Source: China Disabled Persons’ Federation (2008a). Communique on Major Statistics of the Second China National Sample Survey on Disability. Retrieved January 14, 2009 from http://www.cdpf.org.cn/english/contactus/content/ 2008-04/14/content_84989.htm

disabilities, which represents 6% of the total national population. Table 3.1 displays categories and percentages of all individuals with disabilities as reported by the survey. Seventy-five percent of the population with disabilities (or 62,250,000) lives in rural areas while the other 25% (or 20,710,000) in urban areas. This mirrors the distribution pattern of the general population. In China, urban living is generally preferred and desired due to the availability of resources in urban areas. One needs to obtain a residential permit in order to live in a city or town. Therefore, the geographic locations of people with disabilities have a significant impact on whether they have access to rehabilitation services. Across China, 70,500,000 households have one or more members with disabilities. Twelve percent of these households have two or more members with disabilities. The average size of such household is 3.51 persons. About 30% of all persons with disabilities have severe disabilities, while 70% have mild or moderate disabilities. The survey also reported that, in 2005, although approximately 3 million people with disabilities were employed, 5 million were unemployed. In urban areas, the average annual income for individuals with disabilities was 4,864 yuan (or the equivalent of 712 U.S. dollars) per capita, only a third of what their nondisabled counterparts make a year. According to fiscal figures released by the National Bureau of Statistics of China, the average annual income of workers in urban areas across the nation was 18,405 yuan (or 2,694 U.S. dollars) in 2005. In rural areas, the average annual income is 2,260 yuan (or 330 U.S. dollars), which is below poverty line (World Bank, 2008). About 13% of all rural residents with disabilities are making less than 683 yuan (or 98 U.S. dollars) per person per year. The Second National Survey’s finding that 6% of the general population has disabilities reflected a lower prevalence of disabilities in China than in

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some other countries. At present, it is commonly accepted by the international community that people with disabilities make up around 10% of the world population (UN Millennium Project, 2005). This discrepancy may be due to the fact that some disability categories that have been extensively identified and served in the United States are not recognized in China. Currently, there are no instruments to identify learning disabilities or emotional and behavioral disorders (McLoughlin, Zhou, & Clark, 2005; Pang & Richey, 2006). The term “developmental disabilities” [i.e. severe chronic conditions that are due to mental and/or physical impairments (Centers for Diseases Control and Prevention, 2004)] is not recognized or used in official documents or by the general public in China. The term intellectual disability (ID) was first adopted in China in 1984 (Tao, 1988). According to the Chinese Classification of Mental Disorders, ID is defined as “a group of conditions of arrested development of mind which is especially characterized by subnormality of intelligence and impairments in social adaptive ability.” Criterion for age of onset is not mentioned. But in China, onset has traditionally been restricted to the childhood period at the latest [rather than the age of 18 as in the United States (Tao, 1988)]. However, there is no empirical evidence available on the employment of adaptive behavioral measures in the diagnostic procedures (McLoughlin et al., 2005; Sonnander & Claesson, 1997). In other words, determination of ID in some cases is based solely on intelligence quotient scores (Deng, PoonMcBrayer, & Farnsworth, 2001). Autism was first recognized in China only in 1982 (Tao, 2000). In 2002, Autism Diagnostic Interview-Revised (ADIR), which is considered the “gold standard” in Europe and North America, was validated with 82 participants in China (Guo, Yang, Liu, & Jia, 2002). However, only a very small number of doctors in China were trained on how to use the ADIR. The vast majority of doctors and other professionals do not know what autism is (McCabe, 2007). Even with the disabilities that have been officially recognized in China, critics are concerned about the quality of the diagnostic procedures in China. Contrary to common Western practices, the responsibility of diagnosing and classifying disabilities is generally carried out by medical doctors, particularly pediatricians and psychiatrists (McLoughlin et al., 2005). Although there is a Chinese version of Diagnostic and Statistical Manual of Mental Disorders-IV (American Psychiatric Association, 1994), that is, the Chinese Classification of Mental Disorders, “the diagnosis is largely based on history taking and psychological testing administered by psychiatrists who have little or no formal training in psychometrics.” (McLoughlin et al., 2005, p. 278). Since the 1980s, Western screening methods and assessment instruments, such as the Wechsler tests and the Denver Developmental Screening


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Test, have been employed in China for epidemiological studies. Although some of these instruments have been standardized and modified to be applicable to the Chinese culture (Sonnander & Claesson, 1997), accurately interpreting data and test findings remains a challenge due in large part to a severe shortage of trained professionals qualified to administer these tests (McLoughlin et al., 2005). To gain an accurate census of people with disability is an enormous task because it requires tremendous amount of resources and trained personnel. For the last decade, China has made significant strides in its economic development, yet trained professionals who work with people with disabilities continue to be scarce. Hence, some critics are rather cautious regarding statistics on disability in China (Deng et al., 2001; McLoughlin et al., 2005; Pang & Richey, 2006; Sonnander & Claesson, 1997).

3. Educational Systems In the late 1970s, because of economic and social reforms under Deng Xiaoping’s Reform and Opening policy, the equal rights of individuals with disabilities began to be addressed, and special schools for children with mild ID, visual impairments, and hearing impairments were opened in major cities (Chen, 1996).

3.1. Disability legislation The Compulsory Education Law of the People’s Republic of China, which took effect on July 1, 1986, is the first compulsory education law in China (National People’s Congress, 1986). Compulsory education in China includes 9 years of elementary and middle school. According to the law, local people’s governments at various levels shall establish primary schools and middle schools at such locations that children and adolescents can attend schools near their homes. Also, the local governments are required to establish special schools (or classes) for children and adolescents who are blind, deaf, or with mild ID (Ministry of Education of the People’s Republic of China, 1989). Pupils with disabilities would be allowed 12 years to complete the national curriculum (Piao, Gargiulo, & Xu, 1995). The law’s stated policy goal was to gradually make this education requirement applicable to all children (including those with disabilities) by the year 2000. However, the law does not mandate that education be provided to all students, especially those with severe disabilities and multiple disabilities. Currently, the responsibilities of educating children with severe disabilities are largely shouldered by families and parents (Deng et al., 2001; McCabe & Tian, 2002; Wang, 2008).

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The most comprehensive disability law, the Law of the People’s Republic of China on the Protection of Persons with Disabilities, stipulates that all persons with disabilities should have equal rights, including the right to receive education with their peers without disabilities (National People’s Congress, 1990). The 1994 Regulations on Education for Persons with Disabilities (State Council of the People’s Republic of China, 1994) also calls for compulsory 9 years of education to be provided to children with disabilities. The responsibility is given to schools, social groups, families, and all fields of society (McCabe, 2003). Education for children with severe disabilities is not mentioned directly in these two milestone laws, despite the fact that many of the laws’ articles call for the education of students with disabilities in general. Some advocates for children with severe disabilities have pointed out that under these laws children with severe disabilities are not explicitly included, and thus maybe implicitly included. Conversely, others have pointed out that the lack of specific legal protection creates a barrier to educational opportunities for children with severe disabilities (McCabe, 2003).

3.2. Special education services in China In the Law of the People’s Republic of China on the Protection of Persons with Disabilities, different types of educational programs for children with disabilities were encouraged, including special schools, special classes, and general education for those children with mild disabilities (National People’s Congress, 1990). Special schools used to be considered as the backbone of special education in China (Piao et al., 1995). For over a century, these schools were the only means of providing special education services (Yang & Wang, 1994). Since the early 1980s, special education classes within regular schools have played an important role in the education of children with disabilities. Recent efforts have more directly addressed learning in regular classrooms (LRCs) as an option for students with disabilities (Deng & Manset, 2000; Deng et al., 2001; He, 2004; McCabe, 2003). In 1988, the Chinese Ministry of Education suggested that special classes and LRCs should serve as the main venues of the special education system (Deng & Manset, 2000).

3.3. Learning in regular classrooms LRC is aimed at offering government-supported arrangements for children with disabilities to be educated in neighborhood schools, in classrooms with their peers without disabilities (Deng & Manset, 2000; McCabe, 2003). This strategy does not necessarily reflect allegiance to the concept of inclusion; rather, it more accurately reflects a shortage of personnel, limited fiscal resources and facilities in addition to geographical considerations (Piao et al., 1995). It should be noted that in 1994, the Ministry of Education of the People’s


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Republic of China issued regulation number 16: Methods of Launching Learning in Regular Classrooms for Children and Youth with Disabilities. This regulation stipulated that the participants of LRC should mainly be those children and youth with low vision or blindness, deafness or hard of hearing, and mild mental retardation. In other words, children with more significant disabilities are not placed in these LRCs. Since the 1980s, LRC has become a preferred venue of teaching students with mild disabilities in China. Its cost-effectiveness and immediate results are the two most commonly cited advantages of LRC over the traditional special education methodology (He, 2004). However LRC is not without problems (Deng & Manset, 2000; Deng et al., 2001; He, 2004; McCabe, 2003). First, the emphasis of LRC is on teaching students with disabilities in regular classrooms, which has more to do with providing access to public education rather than ensuring the right to be educated in general education. Second, because teachers typically have 40 or more students in their classrooms, individualization is difficult if not impossible to implement. All instruction in China is highly teacher-directed and focuses on large group activities. Third, regular classroom teachers do not have enough time, resources, or the professional knowledge to help students with disabilities in their classrooms. Fourth, the achievements of students with disabilities have not been required to be included in official program evaluations, and specific evaluation procedures have not been developed. In some LRCs, students with disabilities have been observed sitting alone, isolated from classroom activities; others have remained at home despite the fact that their names appear on “official” class registration list. This unfortunately common practice has been dubbed “drifting in the regular classroom” (Deng & Manset, 2000). Without close, systematic supervision, students with disabilities can be easily neglected in general education classrooms (Chen, 1993; Deng, 1996; Piao, 1991). Moreover, children with severe and multiple disabilities and many children with moderate disabilities are still excluded from LRC initiatives (McCabe, 2003, 2007; Wang, 2008). Many do not get the opportunity to attend school at all, especially those living in rural areas (Deng et al., 2001; McLoughlin et al., 2005). According to a recent report on the status of people with disabilities in China (China Disabled Persons’ Federation, Beijing University, & National Bureau of Statistics of China, 2009), 64% of all children with disabilities between the ages of 6 and 15 were enrolled in 9-year compulsory education schools, with 72% of them enrolled in regular elementary schools, 18% in regular middle schools, and 6.2% in special schools (see Table 3.2). Although the school entrance rates for students with disabilities have increased dramatically, there are still serious concerns regarding LRC’s effectiveness, as illustrated in a survey conducted in Zhuzhou, Hunan province (He, 2004). The researcher found that half of the regular classroom teachers responded on the survey that they spent most of their instructional time with the regular students

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Table 3.2 Percentages of enrollments of children with disabilities (ages 6–18) during 2008

Regular elementary schools Regular middle schools Special education schools Special education classrooms within regular schools Regular high schools Vocational training schools

National

Urban areas

Rural areas

72.0 18.1 6.2 0.5

63.4 23.9 8.5 1.4

73.2 17.2 5.9 0.4

2.1 1.1

2.8 —

2.0 1.2

Source: China Disabled Persons’ Federation, Beijing University, China Bureau of Statistics (2009). Reports on The Conditions of People with Disabilities During the Year 2008. Retrieved January 14, 2009 from http://www.cdpf.org. cn/ggtz/content/2008-12/11/content_30199044.htm

and worked with students with special needs either at the end of or after the class. Seventy-three percent of the teachers reported that they called on students with special needs on average between 1 and 3 times per class. Sixteen percent of the teachers reported that they never called on students with disabilities in their class. The researchers also conducted random observations in regular classrooms with students with special needs. A common phenomenon was that regular classroom teachers rarely paid attention to students with special needs. These classrooms seemed no different from other classrooms if one was not informed of the presence of a student with special needs in those classrooms. The survey also revealed that a large majority of the regular classroom teachers did not have an individualized instructional plan for students with special needs. Regarding teacher training, 70% of the teachers and 69% of the special education administrators reported that they had never received any types of training on how to carry out LRC (He, 2004). Currently, access to public education remains a lofty dream for many Chinese children with severe disabilities and their families. Thirty-six percent of all students with disabilities are not enrolled in any type of schools (China Disabled Persons’ Federation, Beijing University, & National Bureau of Statistics of China, 2009). The current prevailing practice, that is, LRC, does not service students with severe disabilities. This phenomenon could be due to several reasons. First, qualified professionals to work with children with developmental disabilities continue to be severely lacking. Second, there are simply not enough classrooms and schools for students with disabilities as illustrated in Fig. 3.1. Third, there is the traditional belief of reserving the limited educational resources in China for those who can contribute to society most. Historically, individuals with disabilities have been considered a “burden” to the society. Even some parents of children with disabilities themselves do not believe in the benefits of

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Multiple disabilities 12%

Visual impairment 13%

Mental illness 6% Hearing impairment 14%

Physical disability 22%

Speech and language 10% Intellectual disability 23%

Figure 3.1 Percentages of unenrolled school-aged children with disabilities in year 2007 in China. Data source: China Disabled Persons’ Federation (2008b). Statistical Communique on Development of the Work for Persons with Disabilities in 2007. Retrieved January 14, 2009 from http://www.cdpf.org.cn/english/statistics/content/ 2008 -04/10/content____84890.htm

education (Deng et al., 2001). The impact of these challenges on parents will be discussed in the next section.

4. Chinese Families of Individuals with Disabilities 4.1. The concept of family in the Chinese context Pearson (1993) defined family as those kin living in the same household. The traditional Chinese family, deeply influenced by Confucianism, emphasizes family hierarchy and social and family harmony (Tsui, 1989). Inherent in Confucianism is the value of filial piety, which demands respect and obligation to aging parents, honoring the family name, and an emphasis on group harmony rather than individual identity. Chinese families are often described as highly cohesive, partially due to the cultural emphasis on harmony and mutual obligations, and the low tolerance for overt family conflicts (Ow & Katz, 1999). One example that illustrates this concept is that Chinese put the family name first before the given name, implying that the family identity supercedes the person’s individual identity (Xu, Xie, Liu, Xia, & Liu, 2007). The interdependence between parents and children is also important in the Chinese family tradition (Tsui, 1989). The parents, especially the mother, would sacrifice everything for the benefit of their children, especially sons. This strong bond between parents and their children is partially

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rooted in a traditional belief that parents’ old age security depended on their sons (and sometimes daughters when there is no son). In addition, rearing children would provide emotional fulfillment to the parents. Moreover, the success of the children is regarded as the social achievement of the parents (Tsui, 1989). Since the early 1980s, research demonstrates that family structure has undergone major changes in China, most noticeably in urban families as a result of socioeconomic developments, among them, the single child policy that has impacted family structure and family dynamics (Tsui, 1989; Xu et al., 2007). The establishment of China’s one-child policy in 1979 placed the burden of improving family status and wealth, looking after elderly parents and continuing the family lineage on the sole child. These expectations placed heightened pressures on the individual child to succeed. Hence the shame and stigma are ever enhanced when a child is unable to live up to these expectations (Yang, & Pearson, 2002). Despite the changes in Chinese family structure, research still suggests that the family remains the main pillar of social support network in China. Families provide mutual support in times of crisis and during difficult family situations (Xu et al., 2007).

4.2. Stigma Due to the general public’s lack of understanding of developmental disabilities and the Chinese etiological beliefs regarding disabilities, many Chinese still hold a “moral view” toward disability. According to this view, the existence of disability is a punishment for an ancestor’s misbehavior or current disregard of the family’s obligation to honor one’s ancestors (Chan & Lee, 2004; Deng et al., 2001; McCabe, 2007). Further, genetic transmission of all illnesses is another popular belief in Chinese society. Therefore, the disability of an individual is also experienced as a stigma for the family as a whole due to these intertwined factors (Yang & Pearson, 2002). This stigmatizing view translates into negative attitudes toward people with disabilities amongst the majority of the general public. Liu (2001) reported that when a group of Chinese students was asked about their attitude toward people with disabilities, they were more positive toward people with physical disabilities than toward people with developmental disabilities and mental disorder. They were also more accepting and sympathetic toward an acquired injury that causes physical limitations than toward a congenital physical or mental disorder. Not only the type of disabilities, but also the severity of the disabilities has an impact on people’s attitudes. Research has shown that parents of typically developing children enrolled in LRC reported more positive attitudes toward children with sensory impairment (e.g., deaf and hard of hearing) than those with intellectual disabilities (Niu, Liu, & Tian, 2005).


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Shame and guilt are generally associated with disability in Chinese culture (Liu, 2001). Within the family of persons with disabilities, shame and guilt can be a complicated mix. Individuals with disability may feel shame and guilt toward his or her family or the family toward the individual, as well as toward ancestors. These feelings often create conflicts and barriers to acceptance among family members (Liu, 2001). Coupled with the traditional preference for males, many Chinese families, especially those who have limited education, would often be confronted with the challenge of how to share the diagnosis of their son with all family members. It is not uncommon for the parents to withhold the diagnosis from the grandparents or for the mother to withhold that information from the father. Sometimes, even after the parents decided to seek rehabilitative services for the child, they may still be elusive about details of the child’s condition or services obtained. This is especially stressful for the mother, who is responsible for daily caregiving and for accompanying the child to all therapy sessions, even as she is not supposed to discuss her worries and concerns with her spouse or extended family members. In addition, because of poor understanding of the cause of developmental disabilities among Chinese families, parents often seek alternative treatments in search of a cure or total recovery (Wang & Michaels, 2009). Herbal medicine and acupuncture are two of the most frequently cited treatments for such disabilities as autism, cerebral palsy, intellectual disabilities, and multiple disabilities. The intense level of stigma in China toward disability also has a significant impact on the family’s interaction with the larger environment. Central to this shame is the concept of “face” in Chinese society, which has been defined as “one’s moral capital and one’s prestige in the social world” (Yang & Pearson, 2002). Disclosure of a child’s disability causes loss of face and negatively affects the prestige of the whole family. Families may adopt strategies to minimize the stigma experienced by the family unit (e.g., restricting the social activity of the individual with disabilities to decrease the likelihood that he or she will embarrass the whole family). The more the family is compelled to hide the existence of the disability, the greater the shame and perceived stigma. Family isolation disrupts the way in which information is received, interpreted, and transmitted between the family system and the larger environment (Yang & Pearson, 2002). Fear of stigma may cause families to lose contact with valuable social cultural resources, such as support from social networks or social contact from multifamily support groups. Families may become reluctant to access outside resources as well, such as schools for children with special needs and limited subsidies from the central and local government. The lack of information and resources received from the outside environment may increase a family’s vulnerability to potential stresses and imbalances by having a child with disabilities.

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4.3. Literature on Chinese families of individuals with disabilities Research on Chinese families of children with developmental disabilities is still in its infancy. After an extensive search in peer-reviewed journals in both English and Chinese language, only 16 studies were found. Three (Lei, 2001; Lv & Gao, 2005; Zhang & Zeng, 2000) were published in Chinese language journals. Topics were rather limited, ranging from characteristics and demographics of families of individuals with disabilities, parental roles in child’s education, to family needs, stress, social support, and coping. Each topic will be discussed in detail in this section. 4.3.1. Characteristics of Chinese families of children with disabilities Based on the existing literature, a large percentage of Chinese families of individuals with disabilities appear to be living at the poverty level (Wang & Michaels, in press; Zhang & Zeng, 2000). The unemployment rate for parents of children with severe disabilities was reported as high as 40% (Wang & Michaels). More than half of the parents in these studies have limited education (9th grade or lower). Researchers have begun examining the impact of the above-mentioned trends of demographics on family’s stress and coping strategies. Yang and Pearson (2002) pointed out that the scarcity of available services for families of children with disabilities forces a greater caretaking burden on families and often requires a shift within the family’s organization. This is especially stressful for families who are living at the poverty level. Parents may not have the knowledge, skills, or resources to advocate on behalf of their child for needed services. Similarly, Wang and Michaels (in press) found that families of children with autism often have to relocate to a new city at least temporarily in search of services due to severe shortage of services for their children in their hometowns. Additionally, relocating often means that one of the parents in a two-parent family has to quit his or her job to accompany their child to school and therapy sessions. This then strains the financial resources of these families and can further alienate families from social supports, as it may be difficult for a family to remain in the new city for an extended period of time with only one income. The 1981 China Marriage Law specifies that parents are responsible for children (regardless of age) who are unable to take care of themselves. Accordingly, the duty of the family to care for a family member with disabilities is necessitated not only by the scarcity of available services but also by law (Yang & Pearson, 2002). Another trend reported by Zhang and Zeng (2000) was that a higher percentage of single-parent families were found in this study in comparison


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to families of typical developing children. Half of the participants rated their marriage as strong and 40% as average. Two-thirds of the parents also reported that they had a good relationship with their extended family; however, parents of children with intellectual disabilities ranked the lowest in this area compared to parents of deaf or blind children. Studies have shown that Chinese parents primarily rely on their spouses and their extended families for social support (Chen & Tang, 1997; Wong et al., 2004; Wang & Michaels, in press; Wong & Wong, 2003). However, the current data on families of children with severe disabilities suggest that they are not getting the level of social support they need from other family members as indicated by Zhang and Zeng (2000). Furthermore, Chinese parents have traditionally been reluctant to request support outside of the family. With heightened stress and limited support, the mental health of these parents has reached a level of needing professional assistance (Huang, Rubin, & Zhang, 1998). Abidin (1995) suggested that parents whose scores on the Parenting Stress Index (PSI) were over 260 could benefit from professional assistance. Huang et al. found that 75% of the Chinese mothers of children with intellectual disabilities in their study scored 260 and above on the PSI. More studies are needed to further explore the connections between different stress factors and family characteristics. 4.3.2. Parental roles in child’s education There seems to be contradicting findings regarding the roles of parents in their own children’s education. Several studies reported that, due to extreme shortage of school placements, many parents of children with severe disabilities have become the only teachers for their children (McCabe, 2007; McCabe & Tian, 2002; Wang & Michaels, in press). For example, some parents opened their own schools for children with autism when no public schools would accept their sons or daughters (McCabe & Tian, 2002). Parent training has always been an invaluable component of services at these private schools for children with autism. Clearly, these parents take on an active role in their children’s education. However, studies published in the Chinese language seem to suggest otherwise. Lei (2001) observed that some parents believed that education was solely the schools’ responsibility, not theirs. Some parents assumed that they were only responsible for caregiving tasks, such as their children’s medical and daily living needs. Many did not know how to teach their child at home and reported that they could not wait for their children to be old enough to go to school. Similarly, Zhang and Zeng (2000) found that amongst three groups of parents of children with disabilities (deafness, blindness, and intellectual disabilities), two-thirds believed that their sole role was to assist the

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child’s teacher. Only one-third considered themselves the most important teacher in their child’s life. Furthermore, parents of children with intellectual disabilities scored the lowest in terms of the importance of education for their children, in comparison to parents of deaf or blind children. The authors speculated that parents’ own education might have hindered their involvement. Therefore, they seemed to rely more on schools for their children’s education. Besides, Chinese parents of children with intellectual disabilities have traditionally felt a certain level of guilt and shame for having such a child, which might have inadvertently kept them from fighting publicly (i.e., advocating) on behalf of their children for rights to education and rehabilitation services (Mao, 1993). Lei (2001) suggested that parent education is crucial. Some large cities (e.g., Beijing, Shanghai) have begun to develop publicly funded early intervention programs. As more families and children take advantage of these early identification and early intervention programs, parents should become more knowledgeable of their child’s conditions and competent in their own parenting skills. Subsequently, they should be more likely to take on an active role in their child’s education and development. 4.3.3. Parental needs and social support Two most frequently identified needs for Chinese families of children with disabilities are the need of school placements for children with severe disabilities, such as autism and intellectual disabilities, and the need of financial support for families to help pay for therapy-related expenses (Lv & Gao, 2005; McCabe, 2007; Wang & Michaels, in press). As discussed earlier in this chapter, the current educational practice in China (i.e., LRC) does not provide services or support to children with severe disabilities. Although each year there are new schools added for children with special needs, there are simply not enough special schools for children with severe disabilities as illustrated in Fig. 3.2. According to the latest report on education for individuals with disabilities during the year of 2007, there are no secondary and postsecondary programs established for students with intellectual disabilities (China Disabled Persons’ Federation, 2008b). McCabe (2007) and Wang and Michaels (in press) both observed that financial support is especially important for families of children with autism. Due to severe shortage of public schools for children with autism, families often have to relocate to larger cities (e.g., Beijing, Guangzhou) at least temporarily in search of private schools. In addition to the costs of having to keep two separate homes, parents have to pay tuition. The tuition costs for these private schools can be as high as a family’s entire monthly income. This further strains the financial resources of these families and alienates families from social support. Even the more affluent families still expressed

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Schools for the blind 36 Others 582 *

Schools for the deaf 688

Schools for children with intellectual disabilities 361

Figure 3.2 Establishment of new special schools for individuals with disabilities in year 2007. *Others included 83 special high schools, with a total enrollment of 4,978. Of which 68 were high schools for the deaf, enrolling 4,047 deaf students and 15 for the blind with an enrollment of 931 blind students.There were also 23 new centers for children with autism with an enrollment of 1,056. Data source: China Disabled Persons’ Federation (2008b). Statistical Communique on Development of the Work for Persons with Disabilities in 2007. Retrieved January 14, 2009 from http://www.cdpf. org.cn/english/statistics/content/2008 - 04/10/content_84890.htm

the need for financial support to pay for medical and therapy costs for their child (Lv & Gao, 2005). Another recurring theme across several studies was the need for information on developmental disabilities and normal child development (Lv & Gao, 2005; Wang & Michaels, in press; Wong et al., 2004; Wong & Wong, 2003). Parents also needed practical parenting skills (e.g., physical care and management of problem behaviors). A factor that is somewhat unique for parents in China is that the child with disabilities is often the family’s first and only child. Some parents may lack knowledge of normal child development and parenting skills. This lack of knowledge may be further compounded by the parents having grown up in single-child families, without the benefit of observing and caring for younger siblings. Others may have poor understanding of the causes of their children’s disabilities and be reluctant to have a second child for fear of having more children with disabilities, even though China’s single-child policy does allow for an additional birth if the first child is diagnosed with a disability (Wang & Michaels, in press). Several studies also reported that parents need professional support in coping with stigma (McCabe, 2007; Wong & Wong, 2003). For example, McCabe found that many families expressed the fear of others

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“discovering” they had a child with autism and they would be discriminated against. As a result of such fear, these families had not even informed their extended family members. Parents of adult children with disabilities expressed needs for employment, independent living, and other adult support systems (Wang & Michaels, in press; Wong & Wong, 2003). Many parents need help in developing future plans for their child. They are especially worried that no one will take care of their child after they die. With unemployment rate of 68% in urban areas and 48% in rural areas, people with disabilities continue to depend on other family members and limited government subsidies to cover their living costs (China Disabled Persons’ Federation, Beijing University, & National Bureau of Statistics of China, 2009). Even though the living conditions for individuals with disabilities have improved during the last decade, many still live near or below the poverty line (World Bank, 2008). In terms of family support, several studies found that Chinese parents primarily rely for social support on schools, their spouses, and their extended families (Chen & Tang, 1997; McCabe, 2007; Wong et al., 2004; Wang & Michaels, in press; Wong & Wong, 2003). The fact that Chinese parents are less likely to receive supports outside of the school and family may be explained by several factors. First, literature on typical Chinese families points out that the Chinese are willing to sacrifice for family members (Tsui, 1989). They tend to seek help from immediate and extended family first, before turning to neighbors, communities and professionals (Liu, 2001). Second, other resources such as medical professionals and mental health counselors tend to be lacking in most areas of China. Third, parents are reluctant to seek assistance outside of the family due to fear of discrimination and stigmatization. Disclosure to nonfamily members tends to be selective and keeping the disability condition a secret is more common because of the need to save face. There is a common expression in the Chinese language, “jia chou bu ke wai yang,” which literally means, “It is best to keep quiet of family’s embarrassment and shame.” 4.3.4. Parental stress and coping Similar to findings reported in Western countries, Chinese mothers of children with developmental disabilities tend to report generally high stress levels and negative characteristics associated with their children (Chen & Tang, 1997; Mak, Ho, & Law, 2007; McCabe, 2008; Tam & Cheng, 2005; Wang & Michaels, 2009). Also, Chinese parents reported that they were greatly concerned about planning for the future for their child with developmental disabilities and they were less confident in their own parenting skills and management of their child’s problem behaviors. Compared to parents of children with intellectual disabilities, parents of children with autism reported higher stress levels and lower levels of competency (Wang & Michaels, 2009).


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There also seem to be differences in the perception of stress for mothers and fathers. Wang and Michaels (2009) found that mothers appeared to perceive more parent- and family-related problems than fathers. For example, “I have given up things I really wanted to do in order to take care of my child.” McCabe (2008) reported that mothers felt their life was profoundly affected by having a child with autism; however, they also noted that their husband’s life “continued on as before, with little change, active avoidance of responsibilities and providing little help with childcare and household tasks” (p. 43). Studies also demonstrate that there was a more negative attitude toward persons with severe intellectual disability (SID) than toward persons with mild ID (Huang et al., 1998; McCabe, 2008; Zhang & Zeng, 2000). An increased negative attitude of neighbors may contribute to an increased level of maternal stress. Furthermore, it is generally recognized that, in Chinese society, parents value the academic achievement of their children highly. Since the more SID of a child constituted a greater obstacle to the fulfillment of the family expectation of child in academic achievement, the mother of a child with SID was susceptible to experiencing greater stress. For example, McCabe (2008) discovered that after learning the diagnosis of autism in their child, many Chinese parents experienced severe shock and devastation. Because parents’ hope and expectations for their one and only child were so high, accepting the autism diagnosis became extremely difficult. Therefore, the stress level was extremely high as well. Additionally, because of a lack of understanding, combined with denial and wishful thinking, many parents “wasted” so much time on getting a second or third opinion and search for cures. McCabe concluded that a national referral system for early intervention would be of great help to many Chinese parents. Tam and Cheng (2005) speculated about the reasons why parents of children with SID have lower concept of self than those parents of children who do not suffer from SID. First, parents might face many challenges in adapting to a new life of caring for a child with disability. Second, some parents might have false hope for their children and unrealistic goals for their rehabilitation program. Third, the slow progress of the children may cause parents to feel frustrated or discouraged. Research in the Western countries indicates that stress in parents of children with disabilities correlates with low levels of social support and coping. Specifically, those parents with more helpful or available supports tend to report less stress (Hastings et al., 2005; Weiss, 2002). Mak, Ho, and Law (2007) found that mothers of children with autism in Hong Kong, when they had a high level of sense of coherence, their stress was not affected by the child’s level of symptom severity. Here, the construct of sense of coherence included a component of available resources to meet various demands in one’s life. Mothers with high sense of coherence tended to show more confidence and greater acceptance, which in turn resulted in lower levels of parenting stress.

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Although some studies found that for some Chinese parents of children with disabilities, avoidance was the most dominant coping strategy (Cheng & Tang, 1995; Lam & Mackenzie, 2002; McCabe, 2008; Shek & Tsang, 1993), others found that acceptance and active coping were more frequently adopted (Wang & Michaels, 2009). Amongst four groups of parents (autism, intellectual disabilities, physical disabilities, and others), Wang and Michaels discovered that parents of children with autism were more likely to use active coping and planning than the other three groups. In terms of differences in coping strategies adopted by mothers and fathers, Chinese parents’ responses are similar to Western parents’. For example, Hastings et al. (2005) discovered that mothers used problemfocused coping more frequently than fathers. Wang and Michaels (2009) found that Chinese mothers were more likely to focus on and vent their emotions, and suppress competing activities than fathers, and fathers were more likely to engage in substance use than mothers as a coping strategy. Research also showed that mothers employed different coping strategies during different family life stages. Lam and Mackenzie (2002) explored the experiences of 18 Hong Kong mothers parenting children with Down syndrome. When the children were young (in this case, between birth and 6 months), mothers attempted to hide the disability from others to maintain face. Therefore, their coping resources tended to be limited. Usually, the husband was the only one who could provide support. At this early stage, parents’ coping strategies were emotion-focused and avoidance was particularly evident. When children got older, mothers used selfreliance, seeking social support, and avoidance as the most commonly used coping strategies. However, for mothers with the most difficulty in accepting their children, avoidance remained the dominant coping strategy. Hastings et al. (2005) identified four most frequently adopted coping strategies by Western parents: avoidance coping, problem-focused coping, positive coping, and religious coping strategies. Due to cultural differences, however, Chinese parents may adopt different coping strategies. Mak and Ho (2007) compared the effects of three different coping strategies (i.e., problem-focused, emotion-focused, and relationship-focused coping) on both positive and negative caregiving perceptions of children with ID in Hong Kong. According to Mak and Ho, relationship-focused coping aims at managing, regulating and preserving relationships during stressful periods. In addition to solving problems and managing emotions, successful coping also involves maintaining and protecting social relationships, particularly when couples and families are affected by chronic stress. Mak and Ho argued that with relationship harmony being an important aspect in the Chinese culture and the mother–child relationship being one of the most intimate relationships, it is logical that relationship-focused coping takes on a significant role in shaping mothers’ perceptions of their caregiving process. Caregivers who use relationship-focused coping strategies tended to


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have a greater appreciation for their caregiving role and tended to experience personal growth along the way. They also reported less sense of burden and negative feelings toward their children. Mak and Ho further suggest that under the strong influence of interpersonal relatedness in Chinese culture, relationship-focused coping for Chinese caregivers plays an important role in building and maintaining strong relationships between mothers and children in the caregiving process, as well as in enhancing caregivers’ perceptions of positive experience about their caregiving role. 4.3.5. Family education Two studies examined the effect of family-focused education on parental stress and parenting competency. Wong, Lai, Martinson, and Wong (2006) found that family-focused education was effective in enhancing parental skills, knowledge, and competency among 40 parents of children with developmental disabilities from Guangzhou, China. Specifically after the education program, parents in the experimental group scored higher in the knowledge domain and overall parental attitude, and reported a decrease in stress level. Parents also learned from each other and shared with each other during the training sessions. Wang (2008) evaluated the effects of a parent-training program on the interactive skills of parents of children with autism in a Northeastern city of China. Both Wong et al. and Wang (2008) found that, after training, parents in the experimental group demonstrated a more affectionate attitude toward the child, were more responsive, had more parent–child interactions, and used praise more often during parent–child interactions.

4.4. Siblings of individuals with disability To date, there are no empirical studies on siblings of individuals with disabilities in China, in English or Chinese. Although the Chinese Population and Family Planning Law does allow for a second child if the first child has a disability, families need to apply for permission from the local government (State Council of the People’s Republic of China, 2001). The local family planning office usually requires parents to get genetic counseling to determine if there is a hereditary cause of the disability. The policy implies that permission for a second child can be granted only after the genetic counseling rules out the hereditary factor. McCabe (2008) observed that many Chinese families of children with autism choose not to have a second child or wait a long time before having a second child due to society expectations that most families have only one child as well as the time and money need to raise children. Likewise, in the author’s interactions with Chinese families of children with developmental disabilities, parents were generally fearful of having another child with a

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disability and expressed the concern whether they would have enough time or resources to take care of both children. Across all of these areas, research on Chinese families of children with disabilities is severely limited. A majority of studies lacked control groups, and almost all the samples (with the exception of McCabe, 2007; Wang & Michaels, in press) were recruited from economically affluent and developed areas (e.g., Hong Kong, Shanghai, and Guongzhou). Families recruited for these studies were generally enrolled in some type of special training program. Yet the most recent national sample survey on disability revealed that 75% of the disabled live in rural areas, where services are scarce if not nonexistent. Therefore, generalizations from these studies may be limited, reflecting only those families who were already receiving services from private and public venues. The experiences of families of children with developmental disabilities who are not receiving services, like those living in rural and underdeveloped regions of the People’s Republic of China, remains unknown (Pearson & Chan, 1993; Sonnander & Claesson, 1997; Wang & Michaels, in press).

5. Conclusion Although China is the most populous country in the world with an estimated 83 million individuals with disabilities, little is known about the families of these individuals. Only 16 peer-reviewed studies were found for this review. More empirical studies are needed to assist policymakers in developing educational and social service infrastructure to effectively serve these individuals with disabilities. Much needs to be done for families of children with developmental disabilities in China. The paramount need of Chinese families of children with developmental disabilities is access to free and public education. The LRC initiative, imperfect though it may be, needs to expand its scope to include children with more severe disabilities, not just those with sensory impairment and mild ID. During the past two decades, China has devoted tremendous amounts of resources to achieve the goal of gender equality in 9 years of compulsory education for all children across China, especially girls in remote rural areas. In 2004, the enrollment of boys and girls was both at 98% (State Council of the People’s Republic of China, 2005). Similar levels of resources need to be dedicated to children with developmental disabilities in order to ensure educational rights of these children as stipulated by the Compulsory Education Law of the People’s Republic of China and the Law of the People’s Republic of China on the Protection of Persons with Disability.


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Parent empowerment through education is also much needed in China. In the existing literature, there are only two studies on family education. In addition to learning about their child’s condition and development, parents also need support in becoming better advocates for their children. Traditionally, Chinese parents have always viewed teachers and doctors as experts. This might explain why some parents do not see themselves as equal partners to the professionals in the education of their child with developmental disability. Finally, there are many attitudinal problems that must be addressed. In Chinese culture, there are serious doubts about the learning capabilities of children with disabilities. Learning has always been viewed as acquiring academic knowledge and skills. Learning for living and self-care skills are considered the responsibility of parents. Children with severe disabilities are regarded as a drain on the nation’s limited resources, as they are perceived to be unable to “pay back the debt” to society for their education. In some rural areas, parents of children with disabilities are reluctant to send their children to school because they do not perceive the incentive of a better life or enhanced employment prospects for their children as a result of schooling (McLoughlin et al., 2005). The attitude toward and stigma attached to being disabled or having a family member with disabilities, as well as views on normalization, are important aspects that need to be highlighted in the Chinese society. Such attitudes have implications for the establishment of effective support systems and treatment, such as the inclusion of children with severe disability in regular classes and families’ use of rehabilitation services. In 2007, China signed the United Nations Convention on the Rights of Persons with Disabilities. This is an important step toward promoting, protecting, and ensuring the full and equal enjoyment of all human rights and fundamental freedoms of persons with disabilities, and to promoting respect for their inherent dignity. As the ancient Chinese proverb tells us, a journey of a thousand miles begins with a single step. The journey has begun. Hopefully, future research will demonstrate the fruit of the perseverance of Chinese families of individuals with disabilities.

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C H A P T E R F O U R

Families of Children with Intellectual Disabilities in Vietnam: Emerging Themes Emily D’Antonio and Jin Y. Shin* Contents 1. Introduction 2. Context of Disability-Family Studies in Vietnam 2.1. Legal rights of Vietnamese individuals with disabilities 2.2. Vietnamese culture: Tradition and practices 2.3. Vietnamese concepts of disability 3. Major Themes Arising From Vietnamese Family Studies 3.1. Family needs assessment project 3.2. Issues related to diagnosing intellectual disabilities in Vietnamese children 3.3. Parental stress in Vietnamese families of children with intellectual disabilities 3.4. The importance of social support in decreasing parental stress 3.5. Comparison between mothers and fathers of children with intellectual disabilities 3.6. Summary of major themes 4. Development of Psychometrically Valid Scales in a Vietnamese Population 4.1. Vineland adaptive behavior scales 4.2. Social support scale for parents of children with developmental disabilities 4.3. The multidimensional scale of perceived social support 5. Conclusion References

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Abstract This chapter will review the current state of research and intervention programs for Vietnamese children with intellectual disabilities and their families. First, we *

Hofstra University, Hernpstead, New York, USA



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will highlight necessary cultural considerations when planning programs for these children and their families. We will then summarize the existing literature, with a focus on the relationship between social support and caregiver stress. Next, we will discuss the development of psychometrically sound assessment tools for use in a Vietnamese population. Finally, we will suggest considerations for future research and intervention programs. We hope that our work will inspire future programs to support these families.

1. Introduction Vietnam is a country of 86 million people, the third most populous country in Southeast Asia (Central Intelligence Agency, 2009). Vietnam is also a country that has had to recover from the ravages of wars. During the last few decades, Vietnamese leaders have committed to economic liberalization and international integration of the country, moving to implement structural reforms to modernize the economy and to produce more competitive industries. As of 2008, per capita GDP is $2,900 with exports to the United States increasing 900% from 2001 to 2007 (Central Intelligence Agency, 2009). Now Vietnam is considered one of the fastest emerging economies in Asia. However, it is still a relatively poor country and, apart from the Vietnam War, is less known to the West. The struggles associated with living in a relatively poor country compound the difficulties that Vietnamese families of children with intellectual disabilities must face. Special education programs for children with intellectual disabilities are lacking in Vietnam, as well as research examining the unique needs of these children and their families. The aim of this chapter is to summarize the existing research on Vietnamese families of children with intellectual disabilities and bring to light some of the problems these families face. We will begin with a review of Vietnamese laws, customs, and practices as they relate to individuals with disabilities to provide a context in which to discuss the research literature. Next, we will discuss research regarding Vietnamese families of children with intellectual disabilities, with a focus on the relationship between social support and parental stress. This will be followed by a description and discussion of the development of psychometrically sound scales for use in a Vietnamese population. Finally, we will suggest directions for future research and considerations relevant to intervention programs in Vietnam or other low-income countries. Due to the lack of any research published in English about such families in Vietnam, the research discussed in this chapter is limited to the results of projects conducted in Vietnam through collaborations between the second author and Vietnamese colleagues. The Family Needs Assessment Project was initiated in 2003 to assess the needs of Vietnamese families of children with intellectual disabilities in order to develop strategies for an early

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intervention program appropriate to the Vietnamese cultural context. This project then inspired additional studies. Since there were no diagnostic or screening measures to identify children with disabilities in Vietnam, we had to find ways of finding these children to recruit their families into the needs assessment project. We adopted a screening instrument and conducted an identification project before we could start the family needs assessment study. For the Family Needs Assessment Project, we interviewed both mothers and fathers of the children and compared their experiences. After finding high levels of stress among the mothers of children with intellectual disabilities, we attempted to place these results in context by interviewing mothers of typically developing children. While we conducted these three projects, we also experimented with approaches to adapting the scales developed in a Western cultural context and creating a measurement instrument that would be unique to the Vietnamese culture. With the general assumption that parents would be highly stressed due to their caregiving burdens and worries concerning the child with intellectual disabilities, we believe that certain scales that have face validity to the Vietnamese colleagues will be applicable to the culture. We believe that this will help us to validate the experiences of Vietnamese families that may be consistent with those of the families in other cultural context. However, we also attempted to describe the experiences that may be unique to these families by creating and developing scales on social support and stigma experiences. In this chapter, we report the results of these efforts. At the end of the chapter, we describe research strategies that worked for these families in Vietnam and also some remaining research issues, which we hope may be of help to other investigators conducting research in Vietnam or other low-income countries.

2. Context of Disability-Family Studies in Vietnam According to the most recent surveys conducted by the Vietnamese government and by various international programs, using a broad definition of disabilities to include individuals with physical, mental, or emotional impairment, there are approximately 5.1 million people with disabilities in Vietnam (Ministry of Labour, Invalids and Social Affairs (MOLISA), 1998; USAID, 2005). Approximately 9% of the 5.1 million with disabilities are considered to have an intellectual disability. Of these people with disabilities, 87.3% live in rural areas, 12.7% live in urban areas, and the majority live in poverty with their families.

2.1. Legal rights of Vietnamese individuals with disabilities As is stated clearly in the Law on Education (Center for International Rehabilitation, 2005), individuals with disabilities in Vietnam are legally

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entitled to equal educational rights. The 1999 Law on Education is the main law regulating education for children with disabilities. Article 58 of the law encourages organizations and individuals to establish schools and classes for people with disabilities, in order to promote their rehabilitation, education, vocational training, and social integration. In recent years, educational inclusion for children with intellectual disabilities has been a focus in Vietnam. The government projects on inclusive education were designed with an aim to decrease discrimination against children with disabilities through awareness-raising activities aimed at national and local education authorities, principals, and teachers and through encouraging community involvement to ensure effective inclusive education. Since the inclusive education model has been implemented, the number of children with intellectual disabilities attending schools appears to have been rising steadily (Center for International Rehabilitation, 2005). Yet despite of the efforts by the government and international organizations, the proportion of students with disabilities who receive appropriate services remains low, with about 5% receiving special education services (Center for Inernational Rehabilitation, 2005; Villa, van Tac, & Muc, 2003). The main reason appears to lie in the significant lack of programs to train teachers to work with these children. Training on teaching children with disabilities is included in the national teacher training curriculum. But, at this time, most teachers working with students with disabilities do not possess adequate knowledge, awareness, or skills. Although there has been some increase in the number and skill level of special education teachers, educational programs and the conditions of classes do not meet the demand for special education (Center for International Rehabilitation, 2005).

2.2. Vietnamese culture: Tradition and practices 2.2.1. Values As opposed to the Western idea of individualism, collectivism is an important cultural principle in Vietnam (Hunt, 2005). The core values of collectivism are harmony, duty, honor, respect, education, and allegiance to the family; and Vietnamese culture emphasizes these values. Harmony is achieved by living according to one’s role within the family, creating harmony within oneself and one’s family, and exercising moderation. Interpersonal relationships in Vietnamese society are guided by the core value of respect. Individuals are expected to show respect to others within the family, especially parents. Within the larger community, respect is given to elderly people, teachers, and other authority figures. Education is highly valued, often more than material wealth and successes. Parents are expected to sacrifice to educate their children. Honor, hard work, and duty to one’s family are emphasized, and these principles are taught at a young age (Hunt, 2005). Individuals are expected

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to act with the utmost dignity in all circumstances to avoid family shame. Inappropriate behavior brings shame not only to oneself but also to one’s family. Bringing shame or dishonor to one’s family is to be avoided at all costs. Fulfilling family roles, responsibilities, and duties is often valued higher than fulfilling one’s own desires. Within the family, roles and responsibilities are clearly defined, and individuals are expected to sacrifice for their family roles. Behavior is dictated by one’s role within the family. For example, the role of a parent is to properly raise their children. Vietnamese society is built around the family structure. Unlike American nuclear families, Vietnamese families include the extended family and follow a multigenerational pattern. This model extends to the entire community. In Vietnamese culture, society is conceived as one large extended family. This manner of thinking is demonstrated in the way that Vietnamese greet one another (Hunt, 2005). Kinship pronouns are sometimes used even among strangers greeting each other. In addition to showing respect, this practice reinforces the value of kinship and the concept of a larger extended family. A typical Vietnamese household may include parents, children, daughters-in-law, grandparents, grandchildren, and unmarried siblings. Family roles are hierarchical and clearly defined, with the father as the central figure (Hunt, 2005). Generally, the father is ultimately responsible for providing for his family and making family decisions. Respect for the wisdom of elderly family members is valued; however, grandparents and other elder relatives also play important decision-making roles. If parents are deceased, the responsibility falls upon the eldest male to provide for the family. After the father and elderly relatives, the eldest male sibling generally assumes the most authority. Family roles and responsibilities extend beyond living family members. Children’s duties often include caring for and maintaining ancestral tombs, and all family members are expected to pay homage to ancestral spirits. 2.2.2. Religion Both Confucianism and Buddhism have heavily influenced Vietnamese culture (Hunt, 2005). Confucianism, however, has mainly been influential in regard to the overall way of life in Vietnamese society, while Buddhism is the religion more commonly practiced. An understanding of Buddhist religious practices, therefore, is important in understanding Vietnamese culture. A fundamental concept in Buddhism is the idea that life is cyclical. This contrasts with the Western linear view of life. According to Buddhist beliefs, at the end of this life one’s soul is reincarnated. Each life cycle begins a new identity, human or animal. There is a hierarchy of life forms, with insects at the lowest rung and humans at the highest. Living a life of virtue may one day be rewarded by breaking this cycle and reaching Nirvana, a state of ultimate happiness. A person who commits evil deeds is punished and reincarnated as a lower life form. Responsibility to one’s family plays a role in the cycle of reincarnation. Not only

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is the individual punished for evil deeds, but their descendents may also suffer by being reincarnated as lower life forms. Vietnamese culture associates disabilities with imperfections of the soul. Therefore, disability is often viewed as being caused by sins or evil deeds committed by one’s ancestors.

2.3. Vietnamese concepts of disability There are two general perspectives on disability in Vietnamese society (Hunt, 2005). Until scientific evidence surfaced linking Agent Orange to many forms of congenital disability, Vietnamese ascribed disability to a more traditional belief that it is a punishment for sins committed by one’s ancestors. The modern perspective on disability attributes almost all forms of disabilities to Agent Orange and injuries from the war (Hunt, 2005). Both of these views have consequences for how society treats people with disabilities. The traditional view of disability in Vietnam is significantly influenced by religious beliefs and cultural values that attribute disability to bad deeds or sins committed by one’s ancestors. This belief stems from the concept of reincarnation, which holds that, from birth to death to the afterlife, a person assumes the same identity, in physical or spiritual form (Hunt, 2005). A person who commits evil deeds is punished and reincarnated as a lower life form, and being a person with disabilities is often viewed as punishment for sins committed in a previous life or by one’s ancestors. As a result, families of individuals with disabilities experience shame or guilt. Traditional beliefs about the causes of disabilities fall under four main explanations: moralistic, superstitious, metaphysical, and supernatural (Le, 1995). From a moralistic standpoint, disabilities are viewed as a punishment from a deity for moral sins committed by the person with disabilities, their relatives, or their ancestors (Chan, 1985; as cited in Le, 1995). To illustrate, a Vietnamese mother reported that after her teenage son was paralyzed in an automobile accident, she prayed at the Buddhist temple, promising to fast for 6 months to pay for her moral wrongdoings in a previous life, with the hope that this would cure her son’s disability (Chan, 1985). Superstitious explanations for disabilities suggest that a mother’s thoughts and behaviors during pregnancy caused her child’s disability. For example, the mother of an Asian child with a cleft palate believed her child’s birth defect was caused by the horror films she saw during pregnancy (Cheng, 1990). Metaphysical explanations of disability stem from traditional Chinese medicine regarding the Yin–Yang theory. Balance between basic opposite elements in the body, such as hot and cold or light and darkness, is a necessity for good health. Disequilibrium, according to this belief, could cause disability. Tung (1980; as cited in Le, 1995) described a Vietnamese father who believed that his daughter’s Down syndrome was the result of his wife’s nutritional intake, an imbalance of the Yin–Yang during pregnancy.

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Finally, supernatural causes, such as possession by a demon or evil spirit, are believed to have caused disability. Belief in supernatural causes is likely to result in individuals with disabilities experiencing stigma and shame (Le, 1995). Furthermore, families of individuals with disabilities may also experience shame. Due to the stigma associated with disabilities in Vietnamese cultures, support for the person with a disability is generally kept within the family. As a result, the family member with a disability maybe well-clothed and well-fed, but his or her emotional and psychological well-being may not be attended to (Le, 1995). In order to prevent ridicule and shame, the family member with disabilities may be kept at home and out of the public eye. They may have few social contacts outside of the immediate family. The beliefs about the causes of disabilities described above are fatalistic in nature. Buddhism, for example, suggests that an illness, disability, or physical deformity is fate (Cheng, 1990). The paucity of resources and early intervention programs for individuals with disabilities in Vietnam may stem from the belief that little can be done to help these individuals and their families. Since the ending of the Vietnam War, however, a new perspective on disability has emerged. This new perspective is more pragmatic and scientifically based. As more and more evidence emerges linking the Agent Orange that was deployed during the Vietnam War to a host of diseases, debilitating conditions, and disabilities, Vietnamese attribute many birth defects and congenital disabilities to the chemical agent. With the new perspective on Agent Orange, most people with disabilities are viewed as victims of the war (Hunt, 2005). Huer, Saenz and Doan (2001) found evidence that views about individuals with disabilities are changing in Vietnamese culture. The authors surveyed three groups of Vietnamese Americans, who either emigrated to the United States after adolescence, between the ages of 5 and 12, or before age 5. All three groups scored similarly on a questionnaire regarding attitudes toward children with disabilities and appeared to hold opinions contrary to traditional Vietnamese views on disabilities. For example, all groups on average disagreed with the statement, “Children with disabilities are punishments from God.” Therefore, there is evidence to suggest that views are changing.

3. Major Themes Arising From Vietnamese Family Studies Special education and medical services for children with intellectual disabilities are scarce in Vietnam, and there is a paucity of experts in the field who are conducting research on the families of children with these

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disabilities in Vietnam. So far the research publications identified through ERIC and PsycINFO are limited to the reports generated by a few research projects conducted by the second author of this chapter.

3.1. Family needs assessment project As noted above, we will discuss key issues identified in several reports generated by the Family Needs Assessment Projects conducted in Vietnam from 2003 to 2005 with families of children with intellectual disabilities/ delays. The project was conducted in Hue City, located in central Vietnam, which has a population of approximately 317,000 (Thua Thien Hue Statistical Office, 2004). Parents of children between the ages of 3 and 7 (with an average age of 5) who were identified as cognitively delayed by their teachers were recruited from 20 kindergarten programs, for a total of 120 children. One hundred and six (106) mothers and 93 fathers of the children identified with intellectual disabilities participated in the research described below. The discussion of the chapter will be focused on findings from these projects, with the hope that this will inspire additional thinking, discussion, and research on such families in Vietnam and other developing countries. This chapter will focus on social support and parenting stress, as examined through Family Needs Assessment Project. We will also attempt to discuss additional topics, such as poverty, health, and stigma, in the broader context of the Vietnamese culture. We hope that readers can imagine a situation in which there are no doctors or special education teachers to work with children with intellectual disabilities, let alone experts to conduct family research in the field. The stress that parents of children with intellectual disabilities experience is dramatic. With few professional or informational resources, Vietnamese parents of children with intellectual disabilities are likely to be less educated about their child’s disability and consequently to have little idea how to take care of their children or what to expect as their children grow up. If they receive few professional services, do they obtain some level of support from family members and neighbors, as one might expect in a collectivistic society? What kinds of social support do such children, mothers, and families really receive in Vietnam? The stigma associated with having a disability often alienates these children and families from their social support network. Therefore, they not only lack the help and support they need but also cut off from participation in normal social interactions. We attempted to connect the literature review of research in Western/ Asian countries to the situation of families in Vietnam and to discuss similarities and differences. The main methodological change involved adapting the surveys. We adapted the surveys for use in Vietnam by simplifying both the procedures and the questions. Throughout, we needed to be mindful

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that most Vietnamese teachers and parents had less education and training. Scales were also adapted to limit the number of responses for each question (e.g., from a 7-point to a 3-point Likert scale) and to make questions as concrete as possible. The psychometric properties of some of the scales were examined and reported, as is summarized at the end of the chapter.

3.2. Issues related to diagnosing intellectual disabilities in Vietnamese children Before describing major themes regarding the families in the Family Needs Assessment Projects, it is necessary to address the complexities associated with diagnosing intellectual disabilities. In any context, diagnosing young children with intellectual disabilities is complex. Often, consultation with an interdisciplinary team is necessary to determine a diagnosis and formulate an intervention plan. Parents are generally an important part of the diagnosis process. Parent observations inform professionals regarding the child’s behavior in the context of the home. The diagnostic process can help parents of children with disabilities better understand and accept their child’s intellectual disabilities. In formulating an intervention plan, parents’ understanding of their child’s disabilities can enhance agreement between parents and professionals with regard to the most appropriate intervention strategy (Glaun, Cole, & Reddihough, 1998; Ho, Miller, & Armstrong, 1994; Human & Teglasi, 1993). Lack of agreement between parents and professionals can have negative effects for the child. Lower agreement regarding diagnosis between parents and professionals is associated with poor compliance with intervention programs (Human & Teglasi, 1993; Rogers et al., 1992; Simeonsson, Edmondson, Smith, Carnahan, & Bucy, 1995). Predictably, parent–professional agreement on diagnosis is lower for less easily identifiably conditions, such as language disorders or mild developmental delays (Ho et al., 1994). Diagnostic issues for children with intellectual disabilities may be more complicated in developing nations, where diagnostic tools and trained professionals are often not readily available. As a result, children with special needs may not be identified early enough to benefit from early intervention programs. Diagnostic agreement between parents and professionals in particular cultures may be more difficult. In some Asian cultures, disabilities are associated with punishment for sins committed by one’s ancestors (Kim-Rupnow, 2005; Liu, 2005). Families of children with disability may experience shame and guilt and may be stigmatized by their communities (Hunt, 2005; Lam, 1992; Liu, 2005). Parents in these cultures could therefore be motivated to deny or downplay their child’s disability level (Kim-Rupnow, 2005). While the second author was working in Vietnam, a mother whose 8-year-old child had an intellectual disability and was enrolled in an intervention clinic at a children’s hospital in Hanoi denied

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the fact that her child had an intellectual disability. She believed that the child would become typically developing in the future. Intellectual disabilities can become challenging to diagnose when no formal diagnostic tools are available, and when no formal professionals are experienced with disability. In addition, the social stigma attached to having a disability is likely to increase the chance of parents hiding or denying the fact that their children have disabilities. The ABILITIES Index (Simeonsson & Bailey, 1988) is a potentially useful diagnostic tool in developing countries without a formal diagnostic procedure for children with disabilities and in regions with few professionals. Based on teacher- and parent reports of children with cognitive delays, the Index assesses functional capacity of children in areas of intellectual and physical ability, social skills, and communication. Non-professionals can easily be trained to administer this scale. Ratings based on the Index demonstrate agreement between parents, teachers, and clinicians. Bailey, Simeonsson, Buysse, and Smith (1993) found inter-rater agreement to average 67.2% for exact agreement and 86.2% for agreement within one point. Shin, Nhan, Crittenden, Valenti, and Hong (2008) assessed the utility of the ABILITIES Index in Vietnamese mothers and fathers of children with intellectual disabilities participating in the Family Needs Assessment Project. Considering the implications of diagnostic agreement for intervention compliance, parent and teacher ratings were compared. The ABILITIES Index provides 19 ratings subdivided across nine areas, including hearing (left and right side), behavior (inappropriate) and social skills, intellectual functioning, functioning of the limbs (right and left hand, arm, and leg), intentional communication (receptive and expressive), tonicity (hypertonicity and hypotonicity), integrity of physical status, eyes (left and right), and structural status (the shape and form of the body as compared to other children of the same age). Ratings were obtained from mother, father, and teacher for each child. Overall, parents rated the functional level of their children higher than the teacher ratings. This result was found in most domains of the ABILITIES Index, but particularly in intellectual functioning, social behaviors, and communication. For example, the mean teacher-rated score for social skills was 2.21 out of a possible 5; mothers’ average rating was 1.45 and fathers’ average rating was 1.49 (higher scores indicate higher severity of disability). For overall intellectual functioning, the teacher ratings averaged 2.74, while mothers’ averaged 1.81 and fathers’ averaged 1.80. These differences were statistically significant. There are several potential explanations for this discrepancy. Stigma and traditional cultural views about disabilities could have contributed to parents rating their children as higher functioning. Teachers have a more objective perspective on a child’s functional level compared to a child’s

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parents. Furthermore, teachers receive training and have more knowledge of age-appropriate behavior than parents, and therefore may be better able to judge functional level. Cultural issues may also have affected teacher ratings. Asian cultures set strict standards for academic performance, which could have resulted in lower teacher ratings. Finally, children may behave differently at school compared to at home, and teachers and parents may have simply observed different behaviors, resulting in different ratings. Gender differences also emerged. The difference in ratings between fathers and teachers was greater than that between mothers and teachers. Mothers are traditionally the primary caregiver in Vietnamese families, spend more time with their children and interact more with their children’s teachers compared to fathers. More frequent contacts between mothers and teachers could have influenced this finding. Severity of disability contributed to level of agreement in ratings between mothers and teachers. There was a larger difference between ratings for mothers with more severely disabled children and their teachers, with mothers rating their children’s ability level higher than teachers. Finally, the age of the child was related to the degree of agreement between mothers and fathers. Parents of older children were more likely to agree with each other regarding their child’s functional level, perhaps because they had more time to observe and come to a consensus regarding their child’s abilities. These patterns of agreement and disagreement between mothers, fathers, and teachers were similar to those found in other populations (Bailey et al., 1993; Buysse, Smith, Bailey, & Simeonsson, 1993). Since no comparison group of the same age has been studied in Vietnam, we cannot make comparisons, but it is notable that professionals more often encounter denials or resistance from parents when the child has no clearly identified genetic or physical disorder (Shin et al., 2008). The authors note that caution should be used in mild cases of intellectual disability. A child may be considered delayed due to lack of an enriched social and educational environment, which may commonly occur in developing countries where poverty is more widespread. In these cases the children do not have an intellectual disability and may be able to reach developmental milestones within normal limits with additional quality care and education.

3.3. Parental stress in Vietnamese families of children with intellectual disabilities One of the most widely documented findings regarding families of children with intellectual disabilities is that these families tend to experience elevated levels of parental stress. In Western populations, it has been documented that these families experience elevated stress levels compared to families of typically developing children (Emerson, Hatton, Llewellyn, Blacker, & Graham, 2006; Seltzer, Floyd, & Hindes, 2004). Parental stress has been

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found to correlate with degree of disability and maladaptive behavior of the child in these families (Abbeduto, Selzer, & Shattuck, 2004; Baker, McIntyre, & Blancher, 2003; Beck, Hastings, & Daley, 2004; Hodapp, Ricci, & Ly, 2003). Furthermore, in a developing nation such as Vietnam, variables like low educational attainment and SES are also likely to affect parental stress in both families with and without children with intellectual disabilities. Shin and Nhan (2009) compared parental stress among the 106 Vietnamese mothers of children with intellectual disabilities that participated in the Family Needs Assessment Project to 119 mothers of typically developing Vietnamese children. The typically developing children were chosen at random from the same class as the children with delays and matched for gender. The mean age of the two groups of children did not differ significantly, and in both groups there were approximately twice as many males as females. The divorce rate of both groups was very low, with 92% of parents married. Mothers of the typically developing children were of a significantly higher education level and had a higher SES. Mothers of children with intellectual disabilities reported poorer health status, with a higher incidence of chronic health conditions like backache, headache, rheumatitis, heart disease, high blood pressure, and asthma. Parental or caregiver stress was assessed with The Parenting Stress Index (PSI, Abidin, 1990). Parental stress, as measured by the PSI, encompasses sense of parental competence, restriction of role, and perceived level of social isolation from family and/or community. Shin and Nhan (2009) found that, overall, mothers of children with intellectual disabilities reported higher levels of parental stress compared to mothers of typically developing children. This is consistent with findings in Western and other non-Western populations, where, compared to mothers of typically developing children, mothers of children with intellectual disabilities also have higher levels of depression and stress (Baker et al., 2003; Emerson et al., 2006; Olsson & Hwang, 2001). Compared to mothers in the typically developing group, mothers of children with intellectual disabilities were also significantly older, poorer, less educated, reported less social support, and had more health issues. This is also consistent with literature regarding Western and non-Western groups, in that mothers of children with intellectual disabilities report poorer health (McConkey, Truesdale-Kennedy, Chang, Jarrah & Shukri, 2006; Murphy, Christian, Caplin, & Young, 2007) and report receiving less social support (Mobarak, Khan, Munir, Zaman, & McConachie, 2000; Pearson & Chan, 1993; Stoneman, 1997). Shin and Nhan (2009) found that, even after controlling for demographic and psychosocial predictors of stress, having a child with an intellectual disability was the most robust predictor of parental stress. It thus appears that the major source of parental stress stems from caring for their

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child with disabilities. Other factors including mother’s educational attainment, father’s health condition, economic status, and perceived social support also significantly contributed to parental stress. Lack of economic resources and low educational attainment create a burden on mothers caring for children with intellectual disabilities. Combined with a paternal health condition, these mothers are under a great deal of strain to care adequately for their children. In Western and non-Western cultures, social support buffers stress and is associated with increased caregiver well-being (Glidden & Schoolcraft, 2007; Horton & Wallander, 2001; Song & Singer, 2006; Stoneman, 1998). For these mothers, who are likely to have few strategies to cope with raising children with delays, increased social support could counteract some of the stress created by their lack of economic resources. For example, support in the form of child care from neighbors or relatives could give parents additional time for work or household tasks. Additionally, professional support such as teachers trained in special education could alleviate stress by working with children to improve functional level. Education by trained teachers regarding the causes of intellectual disabilities could decrease stress caused by traditional beliefs that suggest that having a child with disabilities is shameful. The relationship between child gender and stress was different for the two groups of mothers. Mothers of children with intellectual disabilities reported more stress if they had a female child, while mothers of typically developing children experienced more stress if they had a male child. It is possible that mothers of female children with intellectual disabilities experienced more stress because they anticipated difficulties with developmental issues like sexuality, pregnancy, and menstruation. Chen and Tang (1997) similarly found that concern about future developmental issues was a stressor among Chinese mothers of adult children with mental retardation. Mothers reported stress related to increasing curiosity toward sex and marriage among their adult children. Parents of children with intellectual disabilities may anticipate difficulty in obtaining professional help when their child with disabilities deals with these issues. The children in Shin and Nhan (2009) were between the ages of 3 and 7, with an average age of 5. Therefore it is unclear whether anticipation of these future difficulties was a stressor for mothers at this point in their daughter’s development. Additionally, boys are more valued than girls in Asian culture (Lam & Mackenzie, 2002), which may have played a role in the increased amount of stress experienced by mothers of daughters with intellectual disabilities. Another difference found between the groups of mothers was in regard to the relationship between stress and educational attainment. For mothers of typically developing children, there was a large difference in reported stress level between mothers with high- and low-educational attainment. Mothers with more education reported less stress. This gap

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was smaller, however, for mothers of children with intellectual disabilities. In the Vietnamese population, then, lower educational attainment is a parental stressor regardless of child disability status. Less education likely results in fewer resources, financial and otherwise. Higher educational attainment may buffer some of the stress associated with raising a child with intellectual disabilities. However, educated mothers of children with disabilities experience more stress than educated mothers of typically developing children. The study identified a host of problems surrounding children with intellectual disabilities and their families in Vietnam. Although the major source of parenting stress stems from having children with delays, the problems of mothers of children with intellectual disabilities are compounded by their lower educational levels, health issues, poverty, and reduced social support. When planning services for children with intellectual disabilities, programs should be designed comprehensively with consideration of their poverty and health-care needs.

3.4. The importance of social support in decreasing parental stress In families of children with intellectual disabilities, lower levels of social support have been associated with higher levels of stress (Plant & Sanders, 2007; Trivette & Dunst, 1992). There is an indication that social support protects families of children with intellectual disabilities from experiencing elevated levels of parental stress (Feldman, McDonald, Serbin, Stack, Secco, & Yu, 2007). It is therefore important for families of children with intellectual disabilities to have access to and successfully utilize available social support. Social support, however, may not be defined and experienced in the same ways across different cultural contexts. The majority of research that has investigated social support in these families has been conducted in Western populations. Culture has an impact on social support. Traditional Asian cultural values view disabilities as punishments for wrongs committed by descendents (Kim-Rupnow, 2005; Liu, 2005), leading Asian parents of children with disabilities to experience shame and guilt related to their child’s disability (Lam & Mackenzie, 2002; Liu, 2005). Asian families with children with intellectual disabilities may have difficulty seeking social support, because traditional Asian beliefs dictate that family members do not share these problems with non-family members (Pearson & Leung, 1995). This decreases opportunity to receive social support. Park, Glidden, and Shin (2009) examined the relationship between social support and parental stress, comparing the mothers participating in the Family Needs Assessment Projects and the sample of mothers of typically developing children described above. Parental stress was again

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measured using the PSI. The Vietnamese version of the Social Support Scale for Parents of Children with Disabilities (SSSPCDD; Shin & Crittenden, 2003) was utilized to assess the availability of several types of social support. For example, support may be formal (teachers or other professionals) or informal (family or neighbors). The scale was developed by asking Korean and Vietnamese caregivers of children with intellectual disabilities to generate lists of sources of social support in an open-ended format. An open-ended format allowed for the likelihood of certain types of support that exist in some cultures but not others. (For information on the types of informal and formal social support, refer to the report on the psychometric properties of the scale at the end of this chapter.) In line with previous research, Park et al. found that maternal stress was higher among mothers of children with intellectual disabilities when compared to mothers of typically developing children. Regarding social support, mothers of children with intellectual disabilities reported smaller informal social networks and more social isolation. Informal social networks include family and community sources of support. Mothers of children with disabilities, however, did report larger formal support networks, which included assistance from professionals. This suggests that these mothers are less likely to seek support from family, friends, or community members, possibly due to the stigma or shame associated with disabilities. These mothers are, however, likely to seek and receive help from professionals. Despite reporting more professional support than mothers of typically developing children, the proportion of utilization of professionals among mothers of children with intellectual disabilities was low, ranging from 3 to 35%, excluding help from teachers. Forty-nine percent reported support from teachers. Regarding the relationship between maternal stress and social support, stress was negatively associated with specific types of support. Child-directed support, family-directed support, and professional work support were relatively strong predictors of maternal stress. These types of support represent help with practical, everyday activities such as feeding or dressing children. Emotional support, however, was not associated with maternal stress. This suggests that Vietnamese mothers value practical forms of help rather than emotional support.

3.5. Comparison between mothers and fathers of children with intellectual disabilities Parental stress appears to affect mothers and fathers to different degrees and through different mediating variables. Some studies suggest that mothers report higher levels of parental stress compared to fathers (Shin & McDonaugh, 2008; Timko, Stovel, & Moos, 1992); other studies suggest similar levels of parental stress in mothers and fathers, but differences in sources of stress (Dyson, 1997; Flaherty & Glidden, 2000; Hastings, 2003;

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Krauss, 1993; Rousey, Best, & Blancher, 1992). Krauss (1993), for example, found that in the United States, mothers of children with intellectual disabilities report more stress related to their personal support networks, while fathers note stress related to their relationship with their child. In a population of Finnish families of children with disabilities, Saloviita, Italinna, and Leinonen (2003) found that stress in mothers of children with intellectual disabilities was related to maladaptive behaviors in their child, while fathers reported stress associated with perceived social acceptance of their child. Shin et al. (2006) explored social support and parental stress in Vietnamese mothers and fathers of children with intellectual disabilities. The Vietnamese version of the SSSPCDD (Shin & Crittenden, 2003) was again utilized to assess the availability of several types of social support. Furthermore, an individual’s perceived level of social support, as compared to an objective measure of available social support (such as number of supportive individuals), may be a more robust predictor of physical and mental health (Brissette, Scheier, & Carver, 2002; George, Blazer, Hughes, & Fowler, 1989). Shin et al. (2006) therefore assessed perceived social support with the Multidimensional Scale of Perceived Social Support (MSPSS; Zimet, Dahlem, Zimet, & Farley, 1988), a 12-item self-report questionnaire assessing three sources of perceived social support: family, friends, and significant others. Parental stress was again assessed with the PSI. Finally, the impact of stigma on families of children with intellectual disabilities was assessed with The Social Life Scale (SLS). The SLS specifically focuses on the degree to which stigma limits the social life of these families. Shin et al. (2006) found both similarities and differences between mothers and fathers of children with intellectual disabilities. When compared to parents of typically developing children, both mothers and fathers of children with disabilities reported smaller networks of social support. Regarding parental stress, mothers of children with lower intellectual functioning reported higher levels of stress compared to mothers of typically developing children. In contrast, there was no difference in reported stress level between fathers of children with intellectual disabilities and fathers of typically developing children. Overall, mothers of children with intellectual disabilities reported more parental stress than fathers of children with intellectual disabilities, consistent with traditional Vietnamese family roles in which mothers assume the majority of the responsibility for child care. Similar patterns have been found in Western populations. Hastings (2003), for example, found that among parents of children with autism, mothers’ stress levels were correlated with child problem behaviors, but fathers’ stress levels were not. Fathers may be less involved in caregiving activities in families of children with disabilities, and their level of stress may be affected more by other factors. Regarding perceived stigma, differences between mothers and fathers of children with intellectual disabilities emerged. There was no difference in perceived stigma between mothers of children with intellectual disabilities

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and mothers of typically developing children. The two groups of fathers differed, with fathers of children with disabilities reporting significantly more perceived stigma. These results suggest that traditional Vietnamese gender roles impact the types of stress experienced by mothers and fathers of these children. As the primary caregiver, mothers experience stress as a result of problems within the immediate family. Fathers, with the responsibility of integrating the family within the larger community, experience stress related to the acceptance of his family within the community. For both mothers and fathers, those with more stigma experienced more stress. This is consistent with literature regarding Chinese and Korean families of children with intellectual disabilities (Chen & Tang, 1997; Lam & Mackenzie, 2002). In a Western population, Krauss (1993) found that mothers of children with disabilities experienced stress related to their personal support networks, while fathers did not. Mothers of children with intellectual disabilities may be more concerned with maintaining their social network, while fathers are more concerned about maintaining the connection between their families and the larger community. Further analysis revealed other sources of the different experiences of mothers and fathers of children with disabilities. Child gender had a direct impact on mother’s reported stress level, with mothers of female children with intellectual disabilities reporting the most stress. In many Asian cultures, male children are more valued than female children (Lam & Mackenzie, 2002). Due to inadequate health care, a significant proportion of the fathers studied had a health condition. Mothers with a spouse with a health problem also reported significantly more stress. Again, maternal stress was associated with problems within the immediate family. Paternal stress was directly related to socioeconomic status (SES) and the number of individuals available for support, with more stress associated with lower SES and fewer sources of support. Vietnamese fathers appeared to experience more stress when their ability to provide for their family, and their family’s connection with the larger community was compromised. Interestingly, when other factors that could cause stress were taken into account, such as child’s level of intellectual functioning or SES, perceived stigma was not related to parental stress. This was true for both mothers and fathers. Both perceived stigma and parental stress are explained by these demographic variables, rather than a causal relationship between stigma and stress. Overall, when compared to previous findings regarding Korean families, Vietnamese families reported less professional support (Shin, 2002). This is not surprising, considering that Vietnam has fewer established training programs for special education teachers and a less-developed special education system. Both Vietnamese mothers and fathers reported less availability of support from informal and professional sources when their child’s level of intellectual functioning was lower. Mothers who reported a larger network of informal and professional support perceived

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higher levels of social support and somewhat less parental stress. For fathers, larger support networks were associated with higher levels of perceived support and significantly less parental stress. No differences were found between mothers and fathers in amount or type of informal and professional support or perceived social support, but some gender differences did emerge. Mothers reported more parental stress when immediate family members, such as a spouse or other children, were less available for support. Fathers, however, noted more parental stress with less availability of friends and neighbors for support. This suggests that mothers, as the primary caregivers, receive a great deal of direct support from immediate family members and feel stress without that support. As the head of the family in traditional Vietnamese culture, fathers value their family’s acceptance into the community of friends and neighbors and experience stress without that acceptance.

3.6. Summary of major themes Vietnamese parents of children with intellectual disabilities experience more parental stress compared to parents of typically developing children. Stress levels also related to educational attainment, father’s health, and SES, but having a child with intellectual disabilities is the most robust predictor of stress. Stigma appears to have an impact on the lives of these families in several ways. Mothers of children with intellectual disabilities report less informal support from friends or family, possibly because of the shame associated with disabilities. Fathers of children with (vs. without) intellectual disabilities report more perceived stigma. This relationship was not found among mothers, perhaps because fathers are more concerned with the place of their family in the community. Vietnamese families typically adhere to traditional gender roles, in which mothers are more involved in child rearing and fathers are more involved in their family’s role in the larger community. Finally, parents tend to rate their children’s abilities as higher compared to teacher ratings. This also may be due to stigma, as parents wish to downplay their child’s level of disability.

4. Development of Psychometrically Valid Scales in a Vietnamese Population Cultural context plays an important role in the relationship between parenting stress and social support in families of children with disabilities. Instruments that measure these constructs in Western and other nonWestern populations may not demonstrate the same psychometric properties when used in Vietnamese populations. Studies investigating issues

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related to children with intellectual abilities often examine ability to reach developmental milestones and age-appropriate behavior. Age-appropriate behavior, however, may be defined differently in different cultures. Zhang, Wheeler, and Richley (2006), for example, note that a commonly used measure of communication ability assesses whether a child is able to indicate a preference when offered a choice. In Chinese families, however, parents generally make choices for their children, and children are expected to follow out of respect for authority. Therefore, it is important to validate the use of these measures in culturally different populations.

4.1. Vineland adaptive behavior scales (Sparrow, Balla, & Cicchetti, 1984) The Vineland Adaptive Behavior Scales (VABS) is one of the most popular measures of adaptive behavior in children in the United States and other Western countries (Sparrow et al., 1984; Sparrow, Balla, & Cicchetti, 2005). The VABS has been successfully applied to non-Western populations, including rural and suburban infants in India (Anjum, Khadi, & Phadnis, 1990) and Indonesian children with disabilities (Tombokan-Runtukahu & Nitko, 1991). Goldberg, Dill, Shin, and Nhan (2009) assessed the psychometric properties of the Vietnamese version of the VABS (VVABS). Three bilingual Vietnamese clinicians translated the VVABS and evaluated it for cultural relevance (content) and semantic equivalence. Seventeen of the 297 VABS items were reworded to meet content or semantic equivalence. For example, an item in the daily living skills domain was reworded from a reference to using a knife and a fork to using chopsticks. The VVABS was then administered to 120 mothers of typically developing preschool age children and 31 mothers of preschool age children with intellectual disabilities who participated in an early intervention program. The VVABS was found to have an acceptable level of internal consistency, similar to the original Vineland. The VVABS was also able to successfully discriminate between children with an intellectual disability and typically developing children. Regarding construct validity, results were similar to those reported in the original VABS. Therefore, it appears that adaptive behavior is a construct that can be appropriately measured in a Vietnamese population, and the VVABS is an adequate instrument for use in this population.

4.2. Social support scale for parents of children with developmental disabilities (Shin & Crittenden, 1997, 2003) Social support refers to the assistance an individual receives from others (Findler, 2000). Social support has been documented as an important

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factor in helping parents to mitigate their stress and adapt to their children with disabilities (Armstrong, Birnie-Lefcovitch, & Ungar, 2005; Hassall, Rose, & McDonald, 2005). It is also considered a complex multidimensional construct consisting of two broad categories: (1) formal/informal and (2) structural/functional (Boyd, 2002; Findler, 2000; Glidden & Schoolcraft, 2007). Whether the support is formal or informal depends on the people and the relationships involved (Findler, 2000). Support from immediate or extended family, relatives, friends, neighbors, other parents of children with disabilities, religious organizations, or social clubs is considered informal (Boyd, 2002; Dunst, Trivette & Deal, 1988). Formal support comes from professionals such as teachers, social workers, physicians, or social agencies (Dunst et al., 1988; Findler, 2000; Glidden & Schoolcraft, 2007). The second broad category relates to structural versus functional aspects of social support. Structural aspects of social support include size and range of support. Size of social support refers to actual number of individuals one relies on for support. Range refers to different types of individuals and types of support available. Functional aspect of social support can be divided into emotional, instrumental, and informational (Cohen & Syme, 1985). Emotional support involves having someone to provide encouragement and to talk to about problems or personal issues (Chen & Tang, 1997). Instrumental support includes assistance with household chores, such as gifts of money and material goods. Informational support refers to advice or guidance concerning possible solutions to a problem (Findler, 2000). Using the conceptual framework of functional and structural support, a scale was created for a comparative study of Korea and the United States (Shin, 2002) and then adapted for a Vietnamese population. In the study comparing Korean and United States parents, caregivers from the two cultures listed types and availability of social support in an open-ended format and rated the helpfulness of each type of support using the SSSPCDD (Shin & Crittenden, 2003; Shin & Crittenden, 1997, unpublished questionnaire). The open-ended format of this questionnaire is ideal for use in a cross-cultural context, allowing for the likelihood of certain types of support that exist in some cultures but not in others. The questionnaire was then presented to Vietnamese clinicians and adapted to create a Vietnamese version of the scale. Types of social support that are potentially available to Vietnamese families were added, and types that were highly unlikely to apply to a Vietnamese population were dropped. The adapted SSSPCD was then presented to the mothers of children with intellectual disabilities and the mothers of typically developing children from the Family Needs Assessment Project. Twenty-two different types of formal and informal social support were generated when the SSSPCD was presented to Vietnamese

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clinicians (14 informal and 8 formal). Informal support included spouse, wife’s father, wife’s mother, husband’s father, husband’s mother, wife’s siblings, husband’s siblings, sons, daughters, other relatives, friends, neighbors, parents of other children with disabilities, and social helpers. Professional support included teachers, therapists, doctors, private therapists, home helpers, social agencies, babysitters, and doctors in community health clinics. These sources of social support can be divided into five main categories: immediate family members, other relatives, friends, neighbors, and professionals. These categories are similar to those found in Western populations. The Vietnamese clinicians suggested a larger number of informal support sources compared to other populations and were gender-specific when describing these roles (e.g., the role of a father-in-law is different from the role of a mother-in-law). Compared to Western populations, there was a significant lack of availability of professional support, and utilization of available professional resources was low. Five aspects of functional support emerged in this population: material support, child-directed support, family-directed support, emotional support, and professional work support (Park, Glidden & Shin, in press). These categories were similar to the three found in Western populations (emotional, instrumental, and informational; Cohen & Syme, 1985). Regarding child-directed support, the authors found that mothers reported receiving a lot of support with activities such as helping children to bathe, getting dressed, and picking up children at school. In the category of familydirected support, mothers reported that siblings of children with disabilities provided a higher percentage of support than their fathers, with daughters providing more assistance than sons. This may reflect Vietnamese traditional gender roles. Within the category of emotional support, mothers rarely reported receiving support from their husbands. This is best understood in the context of traditional Vietnamese gender roles, in which men are expected to provide material rather than emotional support. Regarding professional support, teachers provided the highest level of support compared to other types of professionals such as therapists. The majority of the families surveyed were of low SES and therefore more likely to have access to teachers rather than private professionals like therapists or doctors. The Vietnamese version of the SSSPCDD showed an acceptable level of reliability and successfully discriminated between the two groups of mothers of children with different cognitive status. The scale appears promising by providing specific information on who is available to provide support and what types of activities are involved in providing the support when Vietnamese mothers raise young children with cognitive delays. These results indicate that there are aspects of social support that are unique to Vietnamese families of children with intellectual disabilities.

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4.3. The multidimensional scale of perceived social support (Zimet et al., 1988) As described above, social support plays a large role in alleviating the level of stress families of children with disabilities experience. Subjective appraisal of the quality of one’s social support may be as important as the actual amount of support one receives. Perceived social support has been implicated as a more robust predictor of physical and mental health than objective measures of social support (Brissette et al., 2002; George et al., 1989). Subjective appraisal of social support has an impact on the stress level of families of children with intellectual disabilities. Lower levels of perceived support have been correlated with higher levels of stress in these families (Hassall et al., 2005; Horton & Wallander, 2001; Plant & Sanders, 2007), and social support may protect these families from experiencing elevated levels of parental stress (Ben-Zur, Duvdevany, & Lury, 2005; Feldman et al., 2007). The MSPSS (Zimet et al., 1988) is a widely used measure of subjective social support, with several useful features. The MSPSS is easily administered and scored, but assesses social support from three sources (family, friends, and significant others). The MSPSS is easy to understand and is minimally affected by social desirability (Dahlem, Zimet, & Walker, 1991; Kazarian & MCabe, 1991). The inclusion of a third broadly defined category of social support (significant other) allows for the assessment of social support from individuals other than family or friends, such as teachers or religious figures. This may be especially important in different cultural contexts (Cheng & Chan, 2004). The psychometric properties of the MSPSS have been well established in non-Western populations. Strong reliability, validity, and factor structure have been demonstrated in Turkish samples (Duru, 2007) and in samples of Chinese adolescents and young adults (Cheng & Chan, 2004; Chou, 1999, 2000). In most populations, the three-factor structure of the measure, encompassing support from family, support from friends, and support from a significant other, has been confirmed. However, in a group of adolescents in Hong Kong, Chou (2000) found two factors, with the “significant other” factor loading onto the “friends” factor. Cultural differences may account for this difference. D’Antonio, Shin, and Dill (unpublished manuscript) examined the psychometric properties of the MSPSS in a population of 106 Vietnamese mothers of children with intellectual disabilities and 119 Vietnamese mothers of typically developing children. Scale items were translated into Vietnamese by three bilingual clinicians and evaluated for content (cultural relevance) and semantic equivalence (same meaning as English version). No items were deleted or reworded in the Vietnamese version.

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D’Antonio, Shin, and Dill confirmed the three-factor structure of the MSPSS in the Vietnamese population. The MSPSS was also able to adequately discriminate between the two groups. Further analysis suggested that the discriminent function coefficient mainly consisted of the “friends” and “family” subscale, not the “significant other” subscale. Consistent with other literature (Pearson & Chan, 1993; Shin, 2002; Shin & Crittenden, 2003), mothers of children with intellectual disabilities reported less social support from friends and family. The MSPSS appears to be a valid measure of perceived social support in a Vietnamese population.

5. Conclusion In this chapter, we have reviewed the research on Vietnamese families of children with intellectual disabilities, focusing on social support and parenting stress. Due to lack of research conducted in Vietnam, a problem that applies to many other developing countries, the scope of the information has been limited to the projects conducted by the second author in collaboration with Vietnamese colleagues. The studies attempted to cover topics that may be consistent with the literature in the Western cultures, with some unique information relevant to the Vietnamese families of children with intellectual disabilities. However, we hope that more information will be generated by other researchers in the field to create a variety of perspectives and information. The lack of research on families of children with intellectual disabilities itself testifies to the problems of obtaining information about such families in developing countries. Conducting family research requires rigorous training at the graduate level, which is not widely available in developing countries. Although Western researchers may be interested in conducting research in such places, language and other cultural barriers often make the collaboration difficult, as do the constraints of finding time and resources to work far from their homes and regular jobs. It is important to explore ways of training and supporting experts in this field to generate more information about these children, especially considering that the majority of children with disabilities (about 85%, Helander, 1993) live in the developing world: many more children than we know from the documented literature are placed in dire conditions without the benefit of access to health or educational services. Based on the studies conducted in Vietnam, one challenge we encountered while attempting to carry out our family surveys in Vietnam was the identification of children with intellectual disabilities. Many of them were not yet diagnosed. When we attempted to locate these children through the reports of their teachers, we discovered that parents and teachers often did not agree about their level of intellectual functioning, with parents

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regarding the disability as milder. Depending on the types of disability, we also found that parents and teachers were more likely to disagree when the etiology of the child’s disability was unknown. This confirms the fact that the views about disability are changing in Vietnam, and that parents may be more comfortable in acknowledging the disability and its degree of severity when they have children with known etiology, such as Down syndrome or cerebral palsy. The scientific information available on the etiology of the disability appears to enhance the parents’ ability to cope with their children psychologically. We also need to be aware that an unknown etiology may spark the traditional views among parents and affect how they experience and think of childhood disability and how they face the community where the stigma is strongly prevalent toward individuals with disabilities. Traditional gender roles also play an important role in parenting experiences within Vietnamese families. Mothers were more stressed than fathers (Shin et al., 2006). While the main source of stress for the mothers was caring for the child with delays, fathers showed a higher level of stress when their financial status was lower and their social network was smaller. When these mothers were compared to the mothers of typically developing children, it was clear that they experience considerably more stress (Shin & Nhan, 2009). The main source of stress among the mothers was having a child with an intellectual disability, but the mothers of these children also had a host of other problems. They were less educated and poorer, obtained less social support, and had more health issues than the mothers of children without cognitive delays. Many factors, such as the availability of resources or the severity of the disability, affect the outcomes of the families of children intellectual disabilities. We found there were a host of problems affecting these families, especially the mothers, in a variety of ways. When social support is not provided adequately, mothers of these children struggle alone to grapple with problems or to find ways of coping with their children. There should be a social support network among the mothers of children with similar conditions at earlier stages. Probably when their children begin to attend special education schools (many with severe intellectual disabilities still do not), these mothers meet and socialize, but it is critical that mothers obtain information at an earlier stage of their children’s delay. As they experience stigma and suffer alone, it may be more critical that they are connected among themselves to exchange information and provide emotional support to each other. In Korea (Shin, 2002), mothers of children with intellectual disabilities in the elementary school age range reported drawing the majority of emotional support from those mothers they met in school, and some even collaborated to plan the futures of their children together. Along with the social support network among the mothers, mothers need the leadership or organizational skills to advocate for the rights of their children and the

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empowerment that will enable them to do so. It is important to educate them to be empowered to speak for their children. They need to know what kind of services are available and what they have a right to ask for. Some level of formal education should be provided to these mothers, possibly in the form of formal schooling that encompasses services or rights of individuals with disabilities as well as the nature of disability. If this type of formal education can be introduced to the mothers, there may be many ways to alleviate the level of stress by dispelling uncertainties due to lack of information. For example, the effects of gender on parenting stress was found in the way that mothers with girls with intellectual disabilities experience more stress than those with boys (Shin & Nhan, 2009). Mothers identify issues and problems of female development and anticipated some of the future problems of their daughters related to sexuality, pregnancy, and menstrual issues. When children have an intellectual disability, it is difficult to train them regarding such issues, and parents also would now know where to turn to obtain professional help. This type of problem could be alleviated by sex education related to puberty, which should be incorporated into the educational curriculum for the mothers. Dealing with sexual problems could be more difficult in Asian culture, which is not easily open to discussion of this topic. Formal education could help to dispel myths and address the parents’ uncertainties/ worries about sexuality problems for their children in the future. The same study (Shin & Nhan, 2009) identified a host of problems surrounding children with intellectual disabilities and their families in Vietnam. Although the major source of parenting stress stems from having children with delays, the problems of mothers of children with intellectual disabilities are compounded by their lower educational levels, health issues, poverty, and reduced social support. When planning services for children with intellectual disabilities, the programs should also be designed comprehensively with consideration of their poverty and health-care needs. For example, our Vietnamese colleagues currently operate a loan program in which the parents of children with disabilities can borrow money to buy buffalos to farm lands. With buffalos, they can farm the land they live on and do not need to take a job away from home. While they work on their farms to support themselves, they can also take care of their child with a disability at home in close proximity. This type of vocational and financial support for the families could be enhanced creatively along with developing educational services for the children and families. Measurement issues need to be explored further to more adequately describe the experiences of Vietnamese families. In addition, as new measures are introduced, their psychometric properties should be thoroughly examined to ensure that the scales measure what they are expected to measure in the Vietnamese culture. Ideally, some scales could be developed to address the unique issues of the culture, but more widely used scales can

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be applied as appropriate to the culture to permit cross-cultural and crossnational comparisons. We attempted these two strategies by applying the measure of social support as it was developed in the Western culture and by creating a new or additional social support scale with feedback from the Vietnamese clinicians. Regarding the measure of child functioning that plays an important role in family outcomes, the Vineland scale was proven effective in Vietnam. In a context where diagnostic or identification tools were lacking, relatively simple adaptive measures, such as the Vineland scale, have proven to serve as valid identification or assessment tools. When the second author attended a regional conference on disability for Asian professionals, she realized that the Vineland scale has been relatively widely adopted in many Asian nations. However, without reporting the results in English, along with the scale’s psychometric properties, it is hard to judge whether the scale is working or how well it is adapted. There should be a channel for information on the psychometric properties and relevancy of commonly applied scales in different cultures. Based on MSPSS (D’Antonio et al., unpublished manuscript), we found that the measure on the social support developed in the Western culture is applicable to the Vietnamese culture with some adaptation. When the response choices were limited, it was found that the psychometric properties were intact, which is encouraging for the adaptation to a population with lesser education. Based on SSSPCD (Shin and Crittenden, 2003), we learned that the social support network (structural aspects of support) was important in playing a significant role in experienced stress in the mothers, with the status of the children significantly affecting the size of the support network. When the qualitative information was analyzed, we found the functional aspects of the support in the areas of emotional, material, child-directed, family-directed, and professional work. However, when they were considered in combination of other demographic factors, the fact that the mothers had the children with disabilities affected them most profoundly, suggesting much of the stigma-related thinking, emotions, and experiences along with worries about the future, may affect them more than any other factors. Further qualitative information should be gathered by using more in-depth interview strategies using ethnographic studies. Follow-up and longitudinal studies of the families will also delineate some causal relationships that will predict the outcomes as these families struggle to cope as their children grow older. This will guide us to design more effective intervention strategies that may increase social support and reduce stress among the family members. In addition, with the strong negative attitudes toward disability that exist in Asian culture in general, the predictors of parenting stress should be examined in a broader context that includes social stigma and attitude toward individuals with disabilities.

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It is challenging to conduct research in developing countries due to lack of expertise, resources, and training in those countries, but such research can be rewarding once the collaboration is forged and research is conducted to generate information, which is hard to obtain to address the issues on childhood disability in the developing world.

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C H A P T E R F I V E

Special Education and Social Services in Korea: Past, Present, and Future Sun Young Ryu Contents 1. Introduction 2. An Overview of Korea 3. The History of Special Education and Family Services 3.1. The quickening era (approximately 1445–1876) 3.2. The establishing era (approximately 1894–1945) 3.3. The transitional era (approximately 1946–1976) 3.4. The developing era (approximately 1977 to present) 3.5. Inclusion 4. Current Status of Special Education and Family Services 4.1. Statistics on individuals with disabilities 4.2. Delivery of special education services 4.3. Stigma 4.4. Family relationships 4.5. Social services 5. Discussion and Future Directions 5.1. Public awareness 5.2. Inclusion 5.3. Social support and services 5.4. Family research on disabilities 6. Conclusion References

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1. Introduction In the 1990s, the inclusion movement became a topic in Korean special education (Jung, 2008). In the beginning, much of the attention and debate concerned philosophical or theoretical frameworks of inclusion, Executive Director, Community Advocacy Network, Inc., Elmhurst, NY 11373, USA.



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“Why should it be inclusion?” After stakeholders, mostly researchers and educators, agreed on inclusion as a means for equal educational opportunity and social participation for people with and without disabilities, the attention and debate moved to approaching methods for inclusion, “How should inclusion be?” (Kwon, 2005). Meanwhile, various legal acts were enacted to promote inclusion of individuals with disabilities in the school system. After 20 years, the fervor for inclusion somewhat cooled, in spite of the fact that more individuals with disabilities and their families want inclusion (Jung, 2008). Students, teachers, and parents all seem to be discouraged because they have faced considerable barriers in attaining an inclusive education. Although the laws secure inclusive education for most individuals with disabilities, educational environments are not ready to accept them in terms of teacher preparation, human resources, teaching materials and financial support. Only 11.6% of students with disabilities were receiving special education services in inclusive classrooms (The Ministry of Education and Human Resources Development, 2007). Nevertheless, individuals with disabilities and their families still dream of inclusion, where students and their families can be equally treated with respect and care. The aim of this chapter is to introduce special education and social services in Korea. To many readers, Korea may be an unfamiliar country. In order to help readers understand Korean special education and social services better, the chapter begins with an overview of Korea. The overview provides background information and national statistics. Then, the history of Korean special education and social services is discussed briefly, followed by the current status of special education and family services. Statistics on individuals with disabilities, delivery of special education service, and social services are discussed with a review of the related legal establishments and existing research. Finally, implications and future directions are discussed for Korean special education and social services.

2. An Overview of Korea Korea is a relatively small country with a long history. It is located to the east of China and to the west of Japan. Until Japanese rule in 1910, Korea was a monarchy. After liberation from Japan in 1945, the country has been divided into democratic South Korea and communist North Korea since 1948. South Korea (Korea here) consists of the southern half of the Korean Peninsula and its size is slightly larger than the state of Indiana. Its population is about 48.5 million and per capita gross domestic product as of 2008 was $26,000 (Central Intelligence Agency, CIA, 2009). For a long time the majority of the labor force was engaged in agriculture. However, industry and services have now replaced agriculture, employing 25.1% and 67.7% of the labor force, respectively. Korea has also gone through rapid

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urbanization, with about 81% of the total population living in urban areas (CIA, 2009). The Koreans are one ethnic family speaking one language. However, the Korea Research Institute for Human Settlements (2009), a government office, predicts that, from 2006 to 2050, the foreign population in Korea will increase from 1.9 to 10% of the total population. Korea is traditionally a collectivist-oriented country (Kim & Choi, 1994). Collectivism highlights the self as collective and interdependent, group goals, communal relationships, and social norms. Like most Asian nations, Korea was influenced by Confucianism and Buddhism. Although Koreans have adopted both, Confucianism plays a larger role in everyday life (Kim & Choi, 1994; Oak & Martin, 2000). Koreans emphasize Confucian values such as a hierarchy of human relationships, loyalty to one’s true nature, filial piety, education for self-improvement, a respect for authority, and a sense of duty and honor to the family (Schneider, Hieshima, Lee, & Plank, 1994). As of 2005, Koreans’ religious composition is 29.3% Christian (Protestant and Catholic), 22.8% Buddhist, 1.4% others, and 46.5% none (The Korea National Statistical Office, 2008). The respondents who report their religious affiliation as “none” are assumed to be people who believe in Confucianism not as religion but as a tradition. The most important aspect of Korean life is the family (Kim & Choi, 1994; Oak & Martin, 2000; Sorensen, 1986). Even though the nuclear family has become more common, the extended family unit remains the model of the Korean family system. Extended family members keep close relationships and gather together on such special occasions as birthdays, graduations, weddings, funerals, anniversaries of deaths, and holidays, especially Chusok (Korean Thanksgiving) and New Year’s Day. In Korean families, the family’s welfare is much more important than the individual. It is not unusual for individuals to sacrifice their own happiness and ambition in order to bring honor to parents and family. Generally, the needs of the family as a whole come first. Choices or decision-making of individual family members reflect the needs of the family (Oak & Martin, 2000). However, with rapid globalization and the introduction of advanced communication and transportation technologies, different cultures are being introduced and their practices at least partially adopted. The Korean value system is also changing. As more women participate in economic activities, women’s power both at home and in society has increased greatly. Various family decisions that used to be made by fathers are now made by mothers. Children have their voices in family decisions, too. On the other hand, family size has reduced to an average of two children. Moreover, one-child or no-child families are increasingly common. Communication among family members, family meal time, and family activities have also diminished in the family system. In Korea, education is a critical means for personal development and success. Higher education at top schools often elevates one’s social position and brings honor to the whole family. Therefore, individual preference or

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choice of college or major is often ignored in favor of family needs (Kim & Choi, 1994; Oak & Martin, 2000; Seo, Oakland, Han, & Hu, 1991; Sorensen, 1986). Currently, to keep up with globalization, English is widely taught at each educational level. Many students go to Englishspeaking countries to learn English. According to the U.S. Immigration and Customs Enforcement’s report, as of 2008, Korean students comprised the largest percentage (15.2%) of international students, followed by India, China, Japan, and Canada (Ohmy, 2009). Koreans’ devotion to education has resulted in a 98% literacy rate (CIA, 2009) and one of the world’s highest enrollment rates for postsecondary education (Seo et al., 1991).

3. The History of Special Education and Family Services Compared to general education, special education in Korea developed much later and more slowly. The history of Korean special education can be broadly divided into four eras (Cho, 2006; Kim, 2008): the quickening era, the establishing era, the transitional era, and the developing era.

3.1. The quickening era (approximately 1445–1876) Although some charitable activities or relief offices for the needy have been reported, few documents indicate any educational or social services for individuals with disabilities during this era. Selected individuals with blindness/visual impairments were taught the art of divination as vocational training at Seounkwan, a government office, in 1445 (Cho, 2006; Kim, 2008). During the quickening era, the few services for individuals with disabilities were limited to persons with blindness/visual impairments.

3.2. The establishing era (approximately 1894–1945) At the end of the nineteenth century, Korea opened its door to foreign countries. During the beginning of this era, special education was introduced by foreign missionaries (Cho, 2006; Kim, 2008; Lim, 2003; Park, 2005; Seo et al., 1991). Rosetta Sherwood Hall, an American missionary and physician, introduced Braille adapted from the New York Point system in 1894 and initiated special education in 1903 (Lim, 2003). This is considered the first special education in the modern school system in Korea. Alice Moffet and Jean Perry also started special education for individuals with blindness/visual impairments. They taught general subjects as well as vocational training through Braille.


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Koreans began to engage in self-examination and there was an awakening of awareness about special education. In particular, Duseong Park created and distributed the Korean Braille point system in 1926 (Cho, 2006; Kim, 2008; Lim, 2003). This was a very important historical event for Koreans because the Japanese tried to erase the Korean language, mandating integration of the two countries. A special class for students with health disorders was set up for the first time in Dongdaemoon Public Elementary School in 1937 (Cho, 2006).

3.3. The transitional era (approximately 1946–1976) When the Education Law was enacted in 1949, special education in Korea began a phase of systematic development. The law provided a basis for equal education for all students and mandated the establishment of special schools in each province and special classes in regular schools. The law also clarified the regulation of private special schools. However, education for individuals with disabilities had been carried out mostly in private rather than in public schools (Cho, 2006; Seo et al., 1991). In 1967, the Korean government adopted “The Five Year Special Education Plan” to improve special education. However, when the government gave priority to promoting the national economy, the execution of the plan was curtailed (Seo et al., 1991).

3.4. The developing era (approximately 1977 to present) The Act for the Promotion of Special Education for the Handicapped was enacted in 1977. The act dictated compulsory elementary and middle school education and free kindergarten and high school education for individuals with disabilities. It also secured related services such as medical examination, vocational training, and necessary therapies. The act was amended in 1994, with provisions for inclusion and the establishment of individualized education plans (IEPs) for students with disabilities. Whereas before Korean special education had mainly been led by the private sector, the 1994 act promoted more involvement of the public sector in special education. In 1981, the Disabled Persons Welfare Act was enacted; this law provided legal grounds for employment of individuals with disabilities. The act stipulated employment and vocational rehabilitation opportunities for persons with disabilities. In the same year the Office of Job Development and Placement Services was established to guide employment services for individuals with disabilities. The Ministry of Health and Welfare supervised the implementation of the law. The outcome, however, was minimal. The Paralympics in 1988 signified a turning point for the public awareness of people with disabilities. There had never before been an event in

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Korea in which individuals with disabilities were themselves the subject of nationwide attention. The public saw that individuals with disabilities could not only participate in sports but could excel in them. It was not just a human story in sports but in everything. Moreover, the Paralympics drew a huge number of volunteers and the concept of “togetherness” among people with and without disabilities became a reality. The event helped the public understand more about individuals with disabilities, the importance of sports for persons with disabilities, and togetherness. In 1990, the Disabled Persons Employment Promotion and Vocational Rehabilitation Act was endowed under the supervision of the Ministry of Labor. The act imposed the requirement that any enterprise or government office that had over 300 workers should employ persons with disabilities for at least 2% of its total work force. An enterprise that did not meet the requirement would pay a fine, while an enterprise that hired individuals with disabilities for more than 2% of its total work force would receive an employment subsidy from the government. However, most enterprises, even government sponsored enterprises chose to pay the fine rather than to follow the guideline (Bokjitimes, 2007; Daejeonilbo, 2009; Nocut News, 2008). The imposed employment rate was amended in 2004 from 2% of over 300 workers to 2% of over 50 workers. The amendment also directed that the imposed employment rate would be adjusted every 5 years based on the population, employment, and unemployment rates of persons with disabilities. Although those two ministries have been in charge of employment of individuals with disabilities, the Ministry of Health and Welfare has focused primarily on vocational rehabilitation such as career assessment and prejob training, the Ministry of Labor mostly on job placement (Seo et al., 1991).

3.5. Inclusion Inclusion in Korea was initiated early in 1903 by Rosetta Sherwood Hall (Lim, 2003). She set up a resource room in an elementary school and taught girls with blindness/visual impairments. Ms. Hall hired a teacher who could teach Braille and assist with other special needs of the students. This is considered the first special education as well as the first inclusion class in the modern school system in Korea. Although not a full inclusion class, it was a forerunner to today’s inclusion. Mary Hill, the first principal at Chungju Elementary School for Children with Blindness/Visual Impairments, also began to send the graduates to neighboring middle schools in 1955 and continually supported those students as they pursued their high school education in general schools. In the mid-1960s, however, those efforts for inclusive education were stopped when those schools were transferred to Korean management. Although special classes in general schools continued to be set up, they were separate classes (Lim, 2003).


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Motivated by the inclusion movement in developed countries, the Korean government amended the Act for the Promotion of Special Education for the Handicapped in 1994. The amendment of the law adopted the basis for inclusion and IEP for students with disabilities. Although inclusion is documented by law, as of 2007 only 11.6% of students with disabilities were receiving special education services in inclusive classrooms (The Ministry of Education and Human Resources Development, 2007). Moreover, existing inclusion is managed with little support and collaboration among stakeholders, partly due to the lack of knowledge and understanding of disabilities and ineffective school logistics.

4. Current Status of Special Education and Family Services 4.1. Statistics on individuals with disabilities Approximately 2 million people, or 4.5% of the Korean population, have various types of disabilities (The Korea Institute for Health and Social Affairs, 2006). The percentage per regional population was 4.13% in Seoul alone, 4% in metropolitan cities, 3.84% in small cities, and 6.88% in rural areas. Most elders with disabilities who cannot afford city life reside in rural areas. However, the percentage of individuals with disabilities under 17 years old was 0.88% in Seoul alone, 0.75% in metropolitan cities, 0.69% in small cities, and 0.68% in rural areas. Most disability-related service facilities, especially therapy and rehabilitation facilities, are more available in big cities. As revised in 2007, the Act for the Promotion of Special Education for the Handicapped classifies disability into 11 areas: blindness/visual impairments, deafness/hearing impairments, intellectual disabilities, physical disabilities, emotional/behavioral disorder, autistic spectrum disorders, communication disorder, learning disabilities, health disorders, developmental disabilities, and other disability conditions. Physical disability comprised the largest proportion of disabilities followed by blindness/ visual impairments, brain disorder, deafness/hearing impairments, multiple disabilities, and intellectual disabilities (The Korea Institute for Health and Social Affairs, 2006). Investigating the statistics on students with disabilities in Korea, Park et al. (2006) reported that the entire population of school-aged children from age 3 to 17 was 9,751,259, and about 86% of these children were attending school. Students who were receiving special education services through special schools, special classes, and inclusion classes numbered 62,903, constituting 0.65% of all school-aged children and 0.76% of children attending school. Children who were school-aged but were not

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required to attend school for personal reasons totalled 55,967, or 0.57% of all school-aged children. About 9% of children were not attending school due to their disability conditions. The Ministry of Education and Human Resources Development (2007) released the statistics on students with disabilities. The total number of school-aged students with disabilities who were receiving special education services in the school system was 65,940, a number increased by about 3,000 compared to the previous year. Students with disabilities who were receiving visiting class services were not counted in the total. Of the total number of students receiving special education services, 4.5% of the students were in kindergarten, 49.6% in elementary school, 23.3% in middle school, and 22.4% in high school. However, as of 2005, only 62.5% of school-aged individuals with disabilities were receiving special education in school. About 38% of school-aged individuals with disabilities were out of the school system. Among them, 18.8% of children with disabilities could not enter elementary school (Choi, 2005). Special schools were serving 34.8% of the students, while special classes were serving 53.6% of the students. The majority of students with disabilities were served in special classes. Inclusion classes were serving 11.6% of the total. About 88% of students with disabilities were still receiving special education services in segregated settings. Students with intellectual disabilities (54.7%) comprised the largest proportion followed by students with physical disabilities (11.7%), with emotional/behavioral disorders (11.6%), and with learning disabilities (10.6%). Students with autistic spectrum disorders were included in the category of emotional/behavioral disorders. Children who did not receive special education but required special support services numbered 16,468, or 0.17% of all school-aged children. These include children with emotional/behavioral problems (27.83%), with learning problems (26.11%), with intellectual problems (15.53%), with speech problems (8.24%), with physical problems (7.58%), children with sight problems (3.53%), with hearing problems (5.44%), and with health problems (5.74%). Children who were school-aged but were not required to attend school for personal reasons totalled 55,967. About 9% of them were not attending school due to their disability conditions (Park et al., 2006).

4.2. Delivery of special education services Centralized around the Ministry of Education and Human Resources Development, the Korean educational system has a top-down hierarchical system. Control of educational operation begins with the ministry and passes to the provisional offices and then to the schools. The Act for the Promotion of Special Education for the Handicapped was amended in 2007, reflecting demands from the education field and the current trend in special education. The amendment states that kindergarten, elementary,


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middle school, and high school education for students with disabilities are compulsory and early childhood education for children under 3 years old with disabilities is free. The act also provides the basis for lifelong education for individuals with disabilities. Special education services are provided through special schools, special classes in general schools, inclusion classes, and visiting classes. Although special education services are guaranteed by the law, many school-aged individuals with disabilities are being neglected due to limited resources or lack of information. Choi (2006) reported that, at the preschool and kindergarten levels, only 29.8% of children with disabilities were receiving special education services in the school system; figures were 73.7%, 57.7%, and 60.9% of children with disabilities at the elementary, middle school, and high school levels, respectively. Until the 2007 amendment, most early childhood education services (e.g., screening, assessment, education) for young children with disabilities had been offered by the private sector and parents paid for them. Since the amendment took effect in June, 2008, there has not yet been any report regarding its effect. According to the Congressional report prepared by the Ministry of Education and Human Resources Development (2007), the delivery of special education services is as follows. 4.2.1. Special schools There were a total of 144 special schools: 5 national, 50 public, and 89 private. About 62% of special schools were private, serving 53% of the special school students. The total number of 22,963 students with disabilities was being served by 6,141 teachers. The average ratio of teacher to student was about 1:3.7, Currently, there are 89 schools for students with intellectual disabilities, 18 schools for students with physical disabilities, 18 schools for students with deafness/hearing impairments, 12 schools for students with blindness/visual impairments, and 7 schools for students with emotional/behavioral disorders. Schools for children with intellectual disabilities comprise 61.8% of special schools. 4.2.2. Special classes in general schools There are 5,753 special classes in 4,530 general schools, where 35,340 students with disabilities are being served by 6,108 teachers. The average ratio of teacher to students is about 1–5.8. The percentage of students being served in special classes was 1.7% in kindergarten, 63.7% in elementary school, 21% in middle school, and 13% in high school. 4.2.3. Inclusion classes There are 6,263 inclusion classes in 3,621 schools, serving 7,637 students with disabilities. The number of students in each inclusion class averages

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1.2. The percentage of students being served in inclusion classes was 20% in kindergarten, 34% in elementary school, 19% in middle school, and 27% in high school. Inclusion classes are continually being increased as the demand for inclusion of students with disabilities increases. 4.2.4. Visiting classes A total of 4,649 students with disabilities were being served through visiting classes. Dispatching agents for visiting classes are special schools, special classes, and special education supporting centers. Special classes send most visiting teachers (50%), followed by special education supporting centers (29%) and special schools (21%). The percentage of students who receive visiting class services are 34% in the institute, 32% in the inclusion class, 19% in home, 12% in the special class, and 3% in the hospital.

4.3. Stigma In Korea, the word “disability” is perceived more as implying physical disabilities than intellectual disabilities, and individuals with intellectual disabilities tend to be indentified with greater stigma. The stigma includes the public’s view of individuals with disabilities as being incapable, unsociable, irresponsible, fearful, or pitiful (Lee, 2003b; Seo et al., 1991). The stigma experienced by an individual with disabilities often extends to the whole family. There is a cultural explanation that partly accounts for the Koreans’ negative view of individuals with disabilities (Kim, 2002). Historically, Koreans have kept a culture of homogeneity. Homogeneity was an important quality in the old agricultural society. Therefore, Koreans tend to pursue homogeneity and refuse heterogeneity. If individuals with disabilities exhibit “differences” that the culture may not easily accept, the individual may be stigmatized. Other studies (Choi, 2000; Lee, 1998; Seo, 1999) suggest that individuals without disabilities may form negative images or attitudes against people with disabilities due to their “unfamiliarity” with individuals with disabilities. Unfamiliarity may cause a wrong impression and lack of understanding about individuals with disabilities, thereby leading to stigma. Due to the social stigma associated with disabilities, individuals with disabilities often face discrimination in their social lives. Lee (2003a) reported that over 85% of persons with disabilities experienced some form of discrimination due to their disabilities, including: • 71% complained about the public transportation system and services and 62.5% experienced refusal to a ride on public buses or taxis; • 81.6% felt discrimination in employment and 78% in promotion;


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• 33.5% experienced refusal in admission to a daycare center or kindergarten; • 46.1% were forced to go to a special school; • 45% had been denied a request to rent a house or a room by the landlord; and • 43.3% felt that they were left out by neighbors. Unfortunately, discrimination also takes place in the home. Individuals with disabilities are often overprotected or ignored. They may not be invited to family affairs and they are especially likely to be hidden during siblings’ wedding-related events. The families are afraid that the family member with disabilities would be a negative influence on the family. In 2008, the Korean government established the Prohibition Law of Discrimination against People with Disabilities. The prohibition law covers five broad categories of discrimination prohibition: human rights matters such as autonomous decision-making, pregnancy, and parenting; employment matters such as recruitment, wages, promotions, and retirement; education matters such as admissions, transfers, and field trips; goods and services matters such as public transportation, communication, and facility use; and legal rights matters such as suffrage and administrative services.

4.4. Family relationships Family is the most basic and important social institution for human beings (Duffy & Leeds, 2001; Dunphy, 2001). The family system is very important for the development of its members because home is the first and perhaps the most important place where lifelong education takes place. A family that includes an individual with disabilities may not have typically assumed family relationships. Family members can be stressed, as they are required to make life adjustments due to their caregiving responsibilities and the support they must provide for the member with disabilities. Ko (2008) investigated the relationships among the burden of parenting, depression, and self-esteem of mothers of children with developmental disabilities. In a study of 173 mothers whose children were receiving services in rehabilitation centers, rehabilitation hospitals, or clinics, mothers reported greater parenting burden than that of mothers of children without disabilities. Worrying about their children’s future was the strongest stress factor. About 62% of the mothers in the study reported that they were depressed, and 40% had a diagnosis of depression. The mothers reported more parenting burden, more stress, and more depression when the child’s disability was severe. However, mothers manifested positive self-esteem regardless of the severity of the child’s disabilities. Mothers’ depression had a significant effect on parenting burden and psychological burden.

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Jeon (2008) examined how typically developing siblings of an individual with disabilities perceived their lives. From in-depth interviews with 13 siblings of children with intellectual disabilities or autistic spectrum disorders in urban areas, respondents demonstrated three themes: emotional problems; problems of daily living; and appreciation and hope. Emotional problems include confusion, guilt, and sadness about their siblings’ disabilities, over identification, depression, and burden for future. Many typically developing siblings felt responsibility for their siblings with disabilities after the parents’ death. Problems of daily living involve coping with characteristics of their siblings’ disabilities, social stigma, and caregiving responsibilities. Appreciation and hope comprise joy and gratitude, positive and responsible perspectives on life, and assertiveness. Although typically developing siblings confront difficulties coping with their brother/sister with disabilities, they also experience positive family relationships and feel gratitude for their lives. Another study reported similar results. Lee (2003b) surveyed 77 mothers who had children with autistic spectrum disorders and typically developing school-aged children. The purpose of the study was to examine service needs of typically developing school-aged siblings of children with autistic spectrum disorders from mothers’ perspective. Parts of the questionnaire were about psychological well-being of typically developing siblings. The mothers responded that their typically developing children seemed to experience both positive and negative effects. Positive effects include increased understanding of other people and strong responsibility. Negative effects were emotional problems such as fear of being known to have a sibling with disabilities, a sense of being neglected from family and friends, and overly high expectations from parents. Ryu (2006) studied perspectives on family relationship, sibling relationship, self-concept, and sibling commitment involving an individual with disabilities. The participants were 66 typically developing Korean young adults between 18 and 30 years of age who had at least one sibling with developmental disabilities and their parent. Both siblings and their parents perceived their family relationships as adequate. Among five factors of health/competence, conflict, cohesion, leadership, and emotional expressiveness, the siblings perceived their family relationships as more satisfactory and happier in terms of family closeness and togetherness than did their parents. This may be that most siblings still lived with their parents, sharing more time and space together. Both groups demonstrated strong family leadership. Concerning sibling relationships, both siblings and their parents showed a moderate level of sibling warmth and a low level of sibling rivalry. The level of sibling conflict was moderate for the siblings and low for their parents. Siblings might be busy with their schooling and decisions about college, job, and marriage. Even though their affection and commitment toward their siblings with disabilities might increase over time,


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siblings might become increasingly engaged in outside activities and relationships, spending less time with their sibling with disabilities (Brody, 1996). Concerning sibling self-concept, both groups fell in the low percentiles compared to the normal distribution percentile. Among three internal categories (self-perceived identity, self-satisfaction, and behavior) and five external categories (physical self, moral-ethical self, personal self, family self, and social self) categories and an additional subscale of selfcriticism, the sibling group was more positive than their parents about moral/ethical self and family self. Confucian teachings emphasize individual morality and the importance of family/nation. Korean siblings seem to carry those old values in their self-concepts. As for sibling commitment, siblings were more positive than their parents about future commitment. Especially concerning future living arrangements, most siblings expected their siblings with disabilities to live with them. Sibling relationships did, however, seem to be limited in terms of intimacy and connection. Although the sibling group demonstrated a high level of commitment toward their siblings with disabilities, their actual involvement with their siblings did not appear as vigorous. Siblings were committed to their siblings with disabilities more affectively than instrumentally, and their commitment was more future-oriented than presentfocused. A number of the siblings commented that they could not have a typically assumed sibling relationship with their siblings with disabilities because their siblings were nonverbal in many cases. While coping with and adjusting to disability conditions of the family member, families involving an individual with disabilities experience positive as well as negative aspects (Jeon, 2008; Ko, 2008; Lee, 2003b; Ryu, 2006). Common positive features are increased affection for family and understanding of other people, increased responsibility and compassion, and greater appreciation of their own good health. Common negative aspects are stress from caregiving responsibilities, burden for future well-being of the member with disabilities, and social stigma. Especially, many typically developing siblings demonstrated their willingness to live with their siblings with disabilities after the parents’ death. This can be interpreted in two possible ways. First, the Korean siblings are truly committed to their siblings with special needs, so they are willing to take care of their siblings under their roofs. Second, other sources—such as family expectation and social demand along with the lack of public support system—might lead them to take responsibility for their siblings with special needs.

4.5. Social services Families of an individual with disabilities require more information and resources. Social services assist individuals with disabilities and their families to stay intact and enhance the quality of their family life by providing

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necessary information and resources. Generally, those services are available to those who registered with a government office and are provided in two forms, financial support and related facility access. As stipulated by current Korean law, social services for individuals with disabilities and their families are available in four ways. First, individuals with disabilities or families of a child with disabilities under 18 years old are entitled to financial support that covers basic living expenses, medical expenses, and education-related costs. Second, rehabilitation services such as counseling, education, therapy, and vocational training are available through the welfare and rehabilitation centers. The government provides vouchers to cover rehabilitation therapy to individuals with disabilities who are under 18 years old and whose family income is less than 50% of the national average. Third, individuals with disabilities are entitled to employment related services through self-supporting support centers. Career counseling, job assessment, job training, and job placement services are offered. Besides the imposed employment policy, the government assists manufacturing businesses run by group homes or disability institutions by searching potential markets together. The government created 3,500 jobs in peer counseling, health assistance, library assistance, etc. for individuals with severe disabilities. Fourth, early childhood education and therapy are provided for young children with disabilities. Day care programs are also offered for working parents. The visiting nurse program is available for homebound individuals with disabilities and pregnant or nursing mothers with disabilities. In addition, the government runs hotline services in local offices to help persons with disabilities in emergency situations. According to a survey of persons with disabilities in 2005 in Daejeon, a large city, among 47,986 individuals with disabilities who registered and 653 persons with disabilities who lived in institutions, 64.5% replied that they did not get much help from those supports and services. Concerning the welfare and rehabilitation centers, 69.5% of the respondents said that they were aware of them but only 23.4% used them, while 47.9% were willing to use them. Concerning the self-supporting support centers, 71.3% did not know about them or their services and only 2.2% used the service, while 36.2% expressed a desire to utilize the services. As for the vocational rehabilitation programs, 69.6% were not aware of them, only 6.9% had experience with them, and 31.9% wanted to know more about them. The supports and services most wanted by individuals with disabilities were financial assistance, rehabilitation and medical services, and employment and vocational training, in that order (Lee, 2005). The Korea Institute for Health and Social Affairs (2008) investigated persons with disabilities and their families’ awareness of social services. The study reported that majority of individuals with disabilities were aware about the monthly disability allowance (67.9%), parking lot benefit


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programs (82.8%), and discount or exemption programs in communication-related bills (92.7%), in transportation fares (90.2%), and in public places such as museum and theater. However, not many individuals with disabilities were aware about financial supports in education of children with disabilities (26%), monthly allowance for children with disabilities (25.6%), medical expenses (39%), and visiting nurse programs (21.8%). Concerning facility-related services, 74.3% of individuals with disabilities were aware about welfare and rehabilitation centers, 69.6% were aware about special schools and classes, and 50% were aware about the access support centers.

5. Discussion and Future Directions Korean services for individuals with disabilities and their families are at a turning point. Although the government has focused on establishing the legal bases and frameworks for these services, these legal frameworks generally do not include subordinate details that guide practical implementation or carry legal binding force (Choi, 2005). For example, the law dictates that kindergarten, elementary school, middle school, and high school education for students with disabilities is compulsory. As of 2005, however, only 62.5% of the whole school-aged populations with disabilities were receiving special education services (Choi, 2005). No person or agency takes responsibility for the 37.5% of neglected school-aged individuals with disabilities. Likewise, many individuals with disabilities are not aware of social services or unable to access to those services (Lee, 2005). Services that consumers are not satisfied with or cannot reach are meaningless. To achieve maximum satisfaction and efficacy, there is a need for public discussion and consensus about these services. In addition, outreach efforts should be made so that as many individuals with disabilities and their families as possible can benefit from those services. In particular, the areas of public awareness, inclusion, social support and services, and family research require more attention.

5.1. Public awareness The biggest barrier to the well-being of individuals with disabilities is a negative impression and a lack of understanding about people with disabilities (The Korea Institute for Health and Social Affairs, 1996). Yet stigma can be reduced through public awareness and education about people with disabilities. When both persons with and without disabilities truly understand the differences between those with and without disabilities, there may not need the words “discrimination” or “stigma.”

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Various efforts have been made to reduce stigma against individuals with disabilities and positive outcomes have been reported. For example, there has been a disability advocacy movement proposing that individuals with disabilities be called “Jangaewoo” meaning “friends with disabilities.” Several movies and television documentaries have successfully conveyed positive messages of individuals with disabilities. Korea is one of the strongest countries in information technology and more than 80% of households have Internet access (The Korea National Statistical Office, 2009). Families of individuals with disabilities, various disability advocacy groups and individuals, and professionals in the field exchange information and promote education and public awareness about disabilities through websites or blogs in cyberspace. The netizens, the general public who uses the internet actively, also share and distribute information about education and public awareness about disabilities. Unfortunately, those efforts are scattered and unsystematic, minimizing their outcomes. To maximize their effects, there must be government-led as well as public-led, systematic, lasting, and efficient education about persons with disabilities. Also, individuals with disabilities and their families need to gather their voices in order to be heard.

5.2. Inclusion Inclusion refers to the placement of students with disabilities in a general education setting with their nondisabled peers with provision of appropriate and quality educational services. Inclusion is not simply placing students with disabilities in general education settings. It implies inclusive curriculum and social inclusion as well as physical inclusion. Cho and Um (2002) surveyed 202 early childhood general education and early childhood special education teachers in partial or full inclusion settings regarding degrees of satisfaction with inclusion and variables affecting satisfaction. The results indicated that the overall degree of satisfaction with inclusion practice was relatively low regardless of the teachers’ previous experience with children with disabilities, teaching certificates, or types of inclusion. Early childhood general education teachers manifested slightly lower satisfaction with inclusion practice than did early childhood special education teachers. However, both groups demonstrated relatively high levels of awareness on inclusion, implying that they acknowledged its importance. Awareness of inclusion, qualification for early childhood special education practice, support for inclusion, and collaboration were all variables that significantly affected the degree of satisfaction with inclusion. In particular, collaboration was the most important contributing variable. The study suggests that, in spite of increasing demands, inclusion practice was not being implemented effectively. In other words, meaningful and high-quality educational services were not provided in inclusion settings.


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Another study (Kwon, 2007) investigated general education teachers’ perceptions of in-service training programs for special education. Of 294 general education teachers from kindergarten through high school, most did not have an opportunity to participate in special education related inservice training programs but were willing to join those programs for their professional development. In order to prepare for inclusion, the respondents demonstrated that they preferred courses such as psychology, counseling, teaching strategies, and assessments. The study implies that special education related in-service training programs are critical and should be offered more often. It also proposes that practical courses and empirical teaching methods should be presented to prepare for inclusion. More attention should be paid to these aspects of inclusion in order for inclusion to be successful in Korea. First, the scope of inclusion should be extended. In spite of the legal provision for inclusion in 1994 and other acts, only 11.6% of students with disabilities in the school system were receiving special education services in inclusion classes as of 2007. The percentage partly indicates that the benefit of inclusion has not been recognized widely. Students with disabilities can gain a sense of belonging, experience a diverse community, develop friendships, and increase opportunities for learning through inclusion (Kim & Horn, 2008). Students without disabilities can also develop an appreciation of human diversity, sensitivity toward other’s limitations, and feelings of empowerment and empathetic skills. Studies indicate that the most important factors contributing to inclusion among teachers were collectivism-oriented culture and legal rights (Chung, 2006; Koun & Chung, 2008). In other words, inclusion was not implemented as a result of recognition of its benefits but as a result of social pressure. To extend the scope of inclusion, then, Korea needs to promote awareness of its importance and benefits. Second, Korean general education is characterized by high academic standards and competition. Many personnel in the field still hold the idea that inclusion will not benefit both general students and students with disabilities, assuming that general students may not achieve their academic goals and students with disabilities may suffer from the competitive environment. Therefore, teachers need to have a knowledge base for their teaching practices in inclusive settings (Yoon, Woo, & Kim, 2008). The knowledge base includes understanding of the student body, subject contents, teaching methods, behavior management, and communication skills. When teachers know about the characteristics of their students, which subject content is the most difficult for students to learn, which subject content is the most difficult for teachers to teach, which teaching method is the most effective for students, how to manage behavior problems, and how to communicate with students effectively, they can provide better educational services. It is therefore necessary to develop an inclusive curriculum that satisfies general students’ academic demands and motivates

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special students’ desire to learning. Under the same curriculum, students with and without disabilities set different educational goals according to their learning abilities and processes. Teachers need to develop various lesson plans and instructional methods in order to meet each student’s unique educational needs. Third, to achieve social inclusion, the same attention needs to be given to students without disabilities. Teachers need to help them express how they feel about inclusion, recognize students with disabilities as classmates, and form friendships with students with disabilities. A study (Jeon & Shin, 2007) reported that students with disabilities in elementary inclusion classes described their friendships with students without disabilities as bringing them pleasure, satisfaction, and help. When students with and without disabilities exchange reciprocal interaction, meaningful social inclusion can be achieved. Inclusion is like an orchestra. In order to produce the best music, every orchestra member should not only be knowledgeable about the music he/ she is going to play, but must also make harmony with other members. Likewise, to bring about successful inclusion, every stakeholder should know about inclusion and disabilities and should actively participate and collaborate. Kim (2002) suggested that we should focus our attention more on commonality than on differences with individuals with disabilities, and that the differences should be taken into account only in asking how, not why or what, one with a difference may be served best. Most of all, students with disabilities should be accepted not as a burden but as members of the school community by encouraging a welcoming environment. No matter how knowledgeable and skillful musicians might be, they will not make wonderful music if their instruments are not good. Similarly, attempts at inclusion will not be successful without a support system. Inclusion requires various resources such as manpower and special equipment. Financial support is critical, too. Limited resources are one barrier that hampers inclusion.

5.3. Social support and services Social support and services are crucial for families of individuals with disabilities in order for them to cope with parenting stress and achieve psychological well-being. Although social supports and services for individuals with disabilities and their families are provided by law, they are carried out poorly due to lack of understanding and limited resources. The budget for those services is often neglected in favor of other national agendas, whereas individuals with disabilities and their families’ needs for support services are increasing (Choi, 2005). Moreover, social and artistic activities are very important for individuals with disabilities (Son, 2007). Through those activities, persons with disabilities may express their inner


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selves, develop their full potential, and gain energy for change and growth. The government needs to support the social and artistic activities of individuals with disabilities. According to a study by The Korea Institute for Health and Social Affairs (2008), individuals with disabilities wanted medical services most (30%) followed by financial supports (22%), and housing services (15.4%). Medical services and financial supports have been the top two most wanted services since 1980. Individuals with disabilities still experience considerable financial hurdles, even as they sometimes require extra medical attention. The need for housing service increased more than 10% compared to that in 2005, suggesting that more individuals with disabilities want a stable and independent housing. In addition, outreach efforts are needed for those who do not know about existing supports and services due to the lack of information. Through those supports and services, every individual with disabilities and his/her family should benefit from and maximize community inclusion and participation as well as personal and family autonomy and empowerment. Disability advocacy groups and parents advocacy groups also need to demonstrate organized efforts for the rights of individuals with disabilities. They need to act as partners in creating and implementing those supports and services.

5.4. Family research on disabilities Although researchers of individuals with disabilities are making progress in fashioning a research base to guide practice, little research has yet been done on services for families. Family research is critical in guiding the direction of social supports and services for individuals with disabilities and their families. The government as well as professionals should encourage and participate in family research on disabilities.

6. Conclusion Once called “the hermit nation” and “the land of morning calm,” Korea is a small country with a long history. During the last four decades, the country has attained a tremendous growth and integrated into the hightech global economy (Central Intelligence Agency, 2009). Special education and social services in Korea has also made remarkable progress since the Special Education Promotion Act in 1977. Until then, Korean special education had mainly focused on education for individuals with visual impairments/blindness and with hearing impairments/deafness. The country has provided various legal grounds and educational systems in order to better serve more individuals with disabilities.

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In 2008, the Korean government presented “The five year (2008–2012) policy development plan for individuals with disabilities,” an ambitious and comprehensive plan for persons with disabilities. The development plan covers four broad categories related to the well-being of individuals with disabilities and their families: welfare, education and culture, economic activities, and social participation. The purpose of the plan is to enhance well-being of individuals with disabilities and their families to the similar level in advanced countries by improving the quality of life, expanding the scope of social participation, and pursuing an inclusive society. Regarding special education, the plan emphasizes promoting inclusive educational environments. In spite of their significant growth, however, Korean special education and social services need to improve in both their quantity and quality. Services should parallel the legal provisions. In order for this to occur, the government should actively supervise the execution of those services and aggressively finance them.

REFERENCES Bokjitimes (2007). Employment rate for individuals with disabilities is still low. Retrieved from http://www.bokjitimes.com/ Central Intelligence Agency (2008). The world factbook. Retrieved from https://www.cia. gov/library/publications/the-world-factbook/geos/ks.html Cho, H. J. (2006). The history of Korean special education. Retrieved from http:// chohongjoong.com Cho, K. S., & Um, S. J. (2002). Degrees of satisfaction with inclusion and variables affecting satisfaction among early childhood and early childhood special education teachers.The Journal of Special Education: Theory and Practice, 3(1), 21–39. Choi, S. Y. (2005). Current status of Korean special education and solution for educational services for persons with disabilities. National Assembly Review, 2005(9), 30–36. Choi, S. Y. (2006). Education is like life for individuals with disabilities. National Assembly Review, 2006(5), 24–27. Chung, Y. S. (2006). A Korean cultural analysis on the inclusion: With focus on the selecting process and selected result of inclusive class. The Journal of Special Education: Theory and Practice, 3(1), 455–487. Daejeonilbo (2009). Imposed employment for individuals with disabilities neglected. Retrieved from http://www.daejonilbo.com/news/ Duffy, M., & Leeds, M. (2001). Which story shall we tell? In P. L. Munhall & V. M. Fitzsimons (Eds.), The emergency of family into the 21st century (pp. 27–36). Boston: Jones and Bartlett Publishers. Dunphy, L. H. (2001). Families on the brink, on the edge. In P. L. Munhall & V. M. Fitzsimons (Eds.), The emergence of family into the 21st century (pp. 3–15). Boston: Jones and Bartlett Publishers. Jeon, H. I. (2008). A qualitative study on the experiences of the siblings of children with disabilities. The Journal of Special Education: Theory and Practice, 9(2), 159–179. Jeon, J., & Shin, H. K. (2007). The study on friendship quality for students with special needs in inclusive elementary schools. The Journal of Special Education: Theory and Practice, 8(3), 403–418.


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Jung, J. K. (2008). Inclusion, the current status. Retrieved from http://cafe.daum.net/ happych7 Kim, B. H. (2008). A historical study on the pioneer of special education in Korea. The Journal of Special Education: Theory and Practice, 9(3), 167–198. Kim, B. H. (2002). Identity of special education: What & how. The Journal of Special Education: Theory and Practice, 3(2), 151–159. Kim, T. Y., & Horn, E. M. (2008). Korean parents’ perceptions of their child with disability’s opportunities for community participation. The Journal of Special Education: Theory and Practice, 9(2), 424–444. Kim, U., & Choi, S.-H. (1994). Individualism, collectivism, and child development: A Korean perspective. In P. M. Greenfield & R. R. Cocking (Eds.), Cross-cultural roots of minority child development (pp. 227–257). Hillsdale, NJ: Lawrence Erlbaum Associates, Publishers. Ko, J. S. (2008). The relationships between the parenting-burden, depression and selfesteem of mothers of the child with disabilities. The Journal of Special Education: Theory and Practice, 9(1), 1–17. The Korea Institute for Health and Social Affairs (1996). 1995 Survey on conditions of individuals with disabilities. The Korean Ministry of Public Health and Welfare. The Korea Institute for Health and Social Affairs (2006). 2005 Survey on conditions of individuals with disabilities. The Korean Ministry of Public Health and Welfare. The Korea Institute for Health and Social Affairs (2008). Public support for people with disabilities. Retrieved from http://www.kihasa.re.kr/ The Korea National Statistical Office (2008). Disabled persons by type & numbers of disabled persons per 100 persons. Retrieved from http://www.kosis.kr/ The Korea National Statistical Office (2009). The rate of the internet access and computer holdings per household. Retrieved from http://www.nso.go.kr/ The Korea Research Institute for Human Settlements (2009). Grand vision 2050. Retrieved from http://www.krihs.re.kr/ Koun, S. M., & Chung, Y. S. (2008). The recognition of teachers on the factors toward inclusive education, inclusive settings, and efficiency of special classroom. The Journal of Special Education: Theory and Practice, 9(3), 221–252. Kwon, H. S. (2007). General education teachers’ perceptions about special education in-service training programs. The Journal of Special Education: Theory and Practice, 8(4), 477–497. Kwon, H. S. (2005). Inclusion in South Korea: The current situation and future directions. International Journal of Disability, Development and Education, 52(1), 59–68. Lee, E. Y. (2003a). A study on the program development for school-age children with autistic siblings. Department of Social Work, The Graduate School of Seoul Women’s University. Lee, G. S. (2005). The study of living condition of women with disabilities and support system. Daejeon Metropolitan City. Lee, H. G. (2003b). Disability and discrimination. The Journal of Special Education: Theory and Practice, 4(1), 171–178. Lim, A. S. (2003). The legal comparison of integrated education between the United States and Korea. The Journal of Special Education: Theory and Practice, 4(4), 21–39. The Ministry of Education and Human Resources Development (2007). Special education yearly reports. Sun Myung Press. Nocut News (2008). Choose rather fine than employment for individuals with disabilities? Retrieved from http://www.cbs.co.kr/nocut/ Oak, S., & Martin, V. (2000). American/Korean contrasts: Patterns and expectations in the U.S. and Korea. Elizabeth, NJ: Seoul, Hollym. Ohmy (2009). In the U. S. A., Korean international students reach over 110 thousands. Retrieved from http://cafe.daum.net/ohmycanada/Erhq/256

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Park, S. W., Kwon, T. W., Woo, E. G., Kim, E. J., Kim, H. I., & Han, H. M. (2006). Statistics on individuals with disabilities who require special education services. Korean Institute for Special Education. Park, Y. K. (2005). Development and current status of special education in Korea. Journal of the International Association of Special Education, 6(1), 15–24. Ryu, S. Y. (2006). Perspectives on sibling relationships involving an individual with special needs: A comparison of parents and normally developing siblings of different cultural backgrounds. Unpublished doctoral dissertation, Teachers College, Columbia University, New York. Schneider, B., Hieshima, J. A., Lee, S., & Plank, S. (1994). East-Asian academic success in the United States: Family, school, and community explanations. In P. M. Greenfield & R. R. Cocking (Eds.), Cross-cultural roots of minority child development (pp. 323–350). Hillsdale, NJ: Lawrence Erlbaum Associates, Publishers. Seo, G. H., Oakland, T., Han, H. S., & Hu, S. (1991). Special education in South Korea. Exceptional Children, 58(3), 213–218. Son, B. S. (2007). Achievement of culture and welfare policy for individuals with disabilities. National Assembly Review, 2007(6), 26–29. Sorensen, C. W. (1986). A teacher’s guide. The Asia Society’s Focus on Asian Studies, (1), 31–35. Yoon, K. B., Woo, J. H., & Kim, Y. S. (2008). A survey on a knowledge-base for teaching activity of teachers in inclusive education. The Journal of Special Education: Theory and Practice, 9(1), 483–508.

C H A P T E R S I X

Perception, Cognition, and Action: New Perspectives on Down Syndrome N. Virji-Babul* and D. Weeks† Contents 1. 2. 3. 4. 5.

Introduction Action and Object Recognition Interpersonal Interactions Imitation and Gestures The Neurobiology of Down Syndrome: Implications for the Action, Perception, and Cognition Framework 6. Neural Mechanisms Underlying Perception, Cognition, and Action 7. Perception–Action Coupling in DS 8. The Influence of Action Experience on Perception and Cognition 9. Impact on Development and Intervention 10. Conclusion Acknowledgments References

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Abstract The motor development of individuals with Down syndrome has traditionally focused on identifying strengths and weaknesses in relation to typical developmental milestones. In this chapter, we consider motor behavior from the context of a multifaceted interaction between action, and the physical and social environment. We review recent findings from neuroscience and developmental psychology that are revealing the mechanisms underlying learning and the relationship between perception and action that support learning. These findings will stimulate new directions for research and offer new insights for intervention for Down syndrome.

* †

Down Syndrome Research Foundation, Burnaby, B.C. Canada University of Lethbridge, Lethbridge, Alberta, Canada



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1. Introduction Our ability to interact with and understand the world around us is dependent on a number of factors. These include perceptual information about our own actions, the impact of these actions on the external environment, and social-cognitive information related to perceiving and interpreting the actions, intentions, and emotions of others. In the human information processing tradition, cognition, and perception have been considered as distinct, mutually exclusive processes. In addition, the neural substrates underlying each process have also been considered to be separate and distinct. This theoretical view has not only influenced our understanding of brain–behavior relations, but also how we approach intervention in individuals with developmental disabilities. The separation of cognition, perception, and action into distinct, encapsulated constructs has led to a functional compartmentalization of the brain. This in turn, has led to the development of intervention strategies targeted at specific regions within the brain. The brain and body are often neatly divided into sections with specialized professionals dealing with specific skills. For example, language is under the domain of speech and language pathologists, and motor skill development is managed by physical and occupational therapists. Motor development is often further subdivided into upper and lower body so that the skills associated with locomotion, postural control, and balance are treated differently from skills requiring reading, writing, and activities of daily living. Such a division is quite remarkable when one considers that over half a century ago, Piaget (1952, 1954) proposed that cognition emerges from the infant’s active engagement with the world, and that the experience of acting in the world is intertwined with perception and cognition of the world. In terms of motor development, action has generally been considered primarily in relation to developmental milestones. As such, when considering the motor development of children with Down syndrome (DS) or other developmental disabilities, the primary focus has been on identifying strengths and weaknesses in relation to achieving developmental milestones. Viewed from this traditional developmental framework, DS has long been associated with delays in cognitive and sensory-motor development. These delays are not characterized by a global pattern of delay but appear to be more complex with an unequal development of skills in different domains of function. While a number of studies have offered important insights into the underlying structural differences in the brain, there has been little direct study of how these structural changes impact the function or the neurophysiological activity associated with specific structures. There is now a vast literature on DS, with reports of various (often conflicting) anatomical, physiological, and behavioral differences. Yet it is striking that there is no unified theory that explains the seemingly disparate findings.

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In the last few decades, considerable effort and resources have been directed at early intervention services. However, there is little evidence that “enriched environments,” in the form of educational and therapeutic programs, have improved long-term developmental outcomes in DS (Gibson & Harris, 1988; Spiker & Hopmann, 1997; Wishart, 1996). In fact, after reviewing recent motor intervention studies, Mahoney, Robinson, and Perales (2004) suggest “there may be fundamental flaws with the treatment methods and strategies used in contemporary practice.” These methods and strategies have often been influenced by the “delay versus different” debate which is now outdated and has not helped to inform practice. Recently, new lines of research and evidence from neuroscience and innovative paradigms from developmental psychology are helping to redefine the perception-action divide. This new research “has the potential to lead to a shift in thinking about development: not as something that occurs simultaneously and in parallel across a number of areas but as a number of causally interacting areas whose complexities can be understood only by conjointly considering various mechanisms of change.” (Rakison & Woodward, 2008, p. 1210). In this chapter we will briefly review recent findings in relation to how individuals with DS process information about the world they interact in. A key feature in this chapter is the shift from considering perceptual-motor skills to considering behavior in the context of a multifaceted interaction between action, and the physical and social environment. For the purpose of this review, we will emphasize findings from the past decade. These findings are helping to develop a new framework for understanding perception, cognition, and action, and stimulate new directions for research on DS and other developmental disabilities. We will start by discussing the perceptual processes involved with simple recognition of objects and human motion. We will then turn to more complex behaviors involving emotional content and imitative behavior. We will then review the current findings on the neurobiology of DS to determine how structural and functional changes in the brain may influence the development of, and relationships between, perception, cognition, and action. Finally, we present some general concepts for readers to consider in developing intervention, and present directions for future studies.

2. Action and Object Recognition The recognition of human action is a fundamental prerequisite for the development of imitation and motor learning (Rizzolatti & Arbib, 1998). Numerous researchers have investigated the neural mechanisms that subserve the visual perception of human movement (Blake & Shiffrar, 2007). One important paradigm has relied on the use of point-light stimuli (Johansson, 1975) in which complex dynamic events are reduced to the movements of a few markers, strategically positioned on the joints or surfaces of moving people or

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objects. Point-light stimuli are typically unrecognizable when presented statically, but are rapidly recognized once set in motion. Thus, point-light displays clearly depict motion cues, but only depict very sparse and largely unrecognizable cues as to the static form. Adults can accurately identify specific actions (Johansson, 1975), gender (Pollick, Lestou, Ryu, & Cho, 2002), identity (Jacobs et al., 2005), and emotional states (Dittrich, Troscianko, Lea, & Morgan, 1996) from the bodily movements defined in these dynamic point-light displays. Indeed, children as young as 3 years of age are able to recognize and differentiate human from nonhuman motions depicted in point-light displays (Pavlova, Krageloh-Mann, Sokolov, & Birbaumer, 2001). Furthermore, by 3 months of age, children begin to discriminate different types of point-light actions (Booth, Pinto, & Bertenthal, 2002; Fox & McDaniel, 1982). Given our interest in DS, we examined children with and without DS on visual tasks involving the perception of human and object motion portrayed in animated point-light sequences (Virji-Babul, Kerns, Zhou, Kapur, & Shiffrar, 2006). All children were first asked to discriminate between human and object motion. Object motion consisted of nonbiological motion depicting the movement of objects such as a wheel, skipping rope, or a Frisbee. Human motion consisted of common human action such as walking, running, and jumping. Our results showed that children with DS were able to differentiate between the motion of people and the motion of nonbiological objects; however, they did not perform this discrimination as accurately as typically developing (TD) children. In a second experiment, the visual perception of emotion from human action was examined. An experienced female dancer was recruited to perform expressive body movements representing the four target emotions of happiness, sadness, anger, and fear. The dancer had reflective markers attached to the major joints of her body. Short sequences of dance movements representing the target emotions were created. Overall, the children with DS did not perform as well as TD children, and appeared to have more difficulty in accurately perceiving the emotional signals embedded in the human movement. In particular, children with DS had a significantly poorer performance in identifying the emotions of anger and fear, in comparison with the emotions of happy and sadness. Zoia, Pelamatti, and Rumiati (2004) have also shown that the processing of static visual representations may also be impaired in adults with DS. In their experiment, line drawings depicting real or nonreal objects and animals were presented, and participants were asked whether or not the drawing represented a real object/animal. Adults with DS had difficulty determining the spatial relationships between the parts of the illustration and were unable to accurately perceive the visual representations. Taken together, these findings suggest that individuals with DS have difficulties in processing visual information related to static as well as motion-related representations. Furthermore, the perception of complex


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human actions, appear to be particularly problematic. The visual perception of human action is a fundamental perceptual ability that is the basis of interpreting social interactions. Impairments in this ability may therefore have a significant impact on the ability of children with DS to perceive, and interpret social communication, as depicted by human motion.

3. Interpersonal Interactions Engaging in effective social interactions not only requires the ability to recognize the actions and gestures of others, but also the ability to interpret emotional cues from the face. Much of the literature describing the development of emotional processing in children has focused on recognition of facial expressions. A number of studies have examined emotion perception in DS using photographs of facial expressions or puppets with different expressions. From these studies, it appears that many children with DS have difficulties in recognizing and identifying emotional information from faces (Kasari, Freeman, & Hughes, 2001). Wishart and Pitcairn (2000) found the perception of emotions related to surprise and fear to be impaired, whereas Kasari et al. (2001) reported that the perception of fear and anger were most impaired. Recently Williams, Wishart, Pitcairn, and Willis (2005) conducted a larger study and found that children with DS were less able than the TD group in matching all the expressions, but that only the expression of fear was statistically significant. Cebula and Wishart (2007) have recently provided an excellent review of the components of social cognition that may underlie impairments in the recognition of emotion. The reader is referred to their chapter for more information. In this section, we will present data from several new studies relating to the neural processing of facial expressions and studies relating to interpersonal relationships. Cheung and Virji-Babul (2008) conducted a study using magnetoencephalography in TD children and children with DS, between the ages of 10 and 12 years. The results from this preliminary study suggest that although viewing emotional facial expressions activates similar neural regions in children with DS, the timing and pattern of these responses are delayed and more variable. Although further work is clearly needed, there may be reasons to reconsider behavioral studies of emotion recognition given that our neural evidence suggests that individuals with DS may require more time to interpret the cues embedded within a social context and may even miss social cues that are expressed early during a social interaction. In everyday life, faces are rarely seen in isolation but rather are embedded within a rich and dynamic social and physical context. While there is now recognition of the importance of contextual cues in emotional recognition (e.g., Da Fonseca, 2009), very little work has been done in the area of DS. Watt, Johnson, and Virji-Babul (2009) recently conducted a

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study on adults with DS to investigate emotional processing by assessing both facial emotional recognition and the interpretation of social interactions on the basis of contextual cues. In the first experiment, participants were shown photographs of faces depicting the following emotions: happy, mad, sad, and scared. In the second experiment, participants were shown a pair of photographs depicting two or more individuals under a variety of social situations (e.g., going to the movies, talking on the phone) and asked to select the photograph which depicted “friends.” Participants were then shown similar social interactions one at a time and asked, “Is this something that friends do?” Adults with DS not only showed difficulties with pure facial emotion as reported by previous investigators, but also had difficulties in extracting the appropriate cues within a social context to determine the construct of friendship. These findings are supported by Wishart (2007) who showed that under collaborative learning situations between a child with DS and a typical child, there were fewer communicative exchanges. In their study, the children with DS often assumed a passive role during the social exchange, with the TD child dominating the interaction.

4. Imitation and Gestures Given that action recognition may be impaired in DS, how does this impact on the ability to imitate the actions of others? Our ability to imitate the actions of other people is central to understanding that others are “like me.” It is a critical indicator of sociocognitive understanding as well as emerging representational skills. Although newborn infants show a capacity for imitating simple actions (Meltzoff & Moore, 1989), it is not until 14–18 months of age that they can imitate more complex actions (Meltzoff, 1990). Meltzoff and Decety (2003) propose that imitation provides a link between perception and execution of human behavior. They provide evidence that infants can map the actions of others onto actions of their body, and through experience, the relationship between their actions, their own experiences and mental states, do begin to link the mental states and behavior of others. What do we know about imitation in infants and children with DS? In spite of the important role of imitation in language development, symbolic function and identity formation, very few studies have specifically examined imitation skills in DS, particularly in terms of how imitation changes with the nature of the behavior being imitated. Imitation has primarily been examined in the context of visual-motor assessment batteries. In this context, individuals with DS generally perform simple imitation skills as well as controls. In fact, individuals with DS have long been known to have strengths in imitation of limb gestures (Down, 1866). However,


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executing simple imitative behaviors such as tongue protrusion (Meltzoff and Moore, 1989) is quite different from the ability to delay imitation (i.e., delay in time from when the action is presented to when the child is asked to imitate the action) or imitating complex behaviors involving novel actions. Both require symbolic representation of objects and events. Rast and Meltzoff (1995) evaluated the ability of children with DS to perform a deferred imitation task by observing a novel action performed with a novel object. Their results indicated that children were able to successfully perform the deferred imitation task. This finding corroborates other reports in the literature suggesting that imitation is a relative strength in children with DS (Hodapp et al., 1992, others). However, Dunst’s largescale longitudinal study of infants (1990) tested between birth and 36 months on a variety of standardized tests of sensory-motor development, including gestural and vocal imitation tasks. In addition, differences, rather than simply delays were identified in the development of vocal imitative skills. Dunst also reported a slowing down of developmental rate with increasing age, with vocal imitation abilities showing the steepest decline. Recently, several new studies have provided some interesting insights into the nature of imitation in DS. Zoia et al. (2004) provided a unique perspective on imitation by evaluating how individuals with DS imitate gestures under three different conditions: In the visual condition, participants were shown common objects, for example, a comb, and asked to demonstrate how the object would be used without touching the object. In the verbal condition, participants were told the name of the object and asked to show its use. Under the real use condition (tactile and visual), participants were given the object and asked to demonstrate its use. Compared to children without DS, the adults with DS performed better under the verbal and “real use” conditions. Bunn, Roy, and Elliott (2007) conducted a similar investigation of how children with DS perform on subtests of an apraxia battery that required imitating gestures that were meaningful (e.g., a |“thumbs up” gesture) or nonmeaningful limb actions that the participants had never seen before. The subtests included both transitive gestures (i.e., action directed toward an object such as grasping a cup) and nontransitive gestures (actions not directed toward an object such as up and down motions of the hand). They found that participants with DS performed as well as control participants on all subtests that involved imitation of gestures. However, the children with DS performed worse when they were required to imitate nonmeaningful gestures that were not directed towards an object. In contrast to Zoia et al. (2004), Bunn et al. (2007) found that children with DS had more difficulty than control participants in verbal pantomiming but not imitating limb movements. This difference may be related to the developmental age of the children in their experiment—the mental age of the children was approximately 5 years. Von Hofsten (1990) has shown that that visual information is the

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most dominant sensory input between the ages of 5 and 6 years; integration between visual, tactile, and proprioceptive information occurs only some time after the age of 8. The children with DS in this experiment may still be relying exclusively on visual information. In addition, Zoia et al. (2004) have shown that imitation of intransitive gestures is more dependent on visual and proprioceptive information than visual information alone. Recently, Wright, Lewis, and Collis (2006) conducted an elegant series of experiments to examine the degree to which imitation skills in children with DS might influence the development of other cognitive skills and to evaluate the impact of imitative strength under specific task situations. Very young children (chronological age (CA) = 2 years; mental age (MA) = 15 months) participated in three different experiments. In the first experiment, performance on an object permanence task was compared to an imitation task. At the start of each trial, a tray with wells was placed on the table out of reach of the child. In the search condition, objects were hidden in one of the wells. In the imitation condition, no object was hidden but the experimenter made hand movements to mimic those involved in retrieving a hidden object. Typically developing children were reluctant to search when no object was hidden. In contrast, children with DS searched to the same extent whether an object was present or not, suggesting that their search strategies may be driven by imitation. In the second experiment, an object permanence task was set up such that imitation would not lead to a successful outcome. The children with DS performed poorly in this task, likely due to an imitation strategy that did not lead to a successful outcome. Finally, in the last experiment, the frequency of the children’s spontaneous play acts were compared to the frequency with which the children imitated a “counterfunctional” act made by the experimenter. Two toys were placed in front of the children—a doll and a toy truck. A set of functional toy items (i.e., toy cup, toy bottle) were put in the middle of the two toys. The children’s spontaneous use of these items was recorded. In the next condition, the experimenter took one of the functional toy items and paired it with the toy truck (e.g., bringing the toy bottle to the truck and giving it a drink). The same toy was then given to the child. The children with DS were much more likely to imitate the counterfunctional acts displayed by the experimenter than TD children, even though children with DS were able to use the functional toy items appropriately during the spontaneous condition. The results from the three experiments suggest that children with DS are much more likely to employ imitative solutions during object search and in play. In novel situations, the strategy was to imitate rather than generate solutions based on representations of the properties of the object. This atypical reliance on imitation may have significant impact on tasks that require a higher level of engagement. Thus far, the discussion has focused at the level of information processing. We reviewed behavioral evidence that suggests individuals


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with DS have difficulty in decoding affective information, in extracting and interpreting social cues as well as perceptual cues that impact on action. We will now consider the underlying neural level—in terms of brain mechanisms underlying information processing difficulties as they relate to the action–perception–cognition framework.

5. The Neurobiology of Down Syndrome: Implications for the Action, Perception, and Cognition Framework Historically, our understanding of brain–behavior relations has been built on a lesion or “deficit” approach. Specific lesions in the brain were correlated with specific clinical deficits—for example, language productions deficits are associated with damage in Broca’s area. While this approach has provided important insights into the knowledge of brain structures and localized brain functions, modern neuroscience is showing that the idea of such localized brain functions is probably too simplistic and that the brain should be viewed in terms of functional interactions between multiple neural networks (e.g., Bressler, 2002; Stam & Dijk, 2002). The traditional view of correlating structural deficits with specific functions has also permeated the DS literature and has led to a theoretical platform in which development is often considered from the perspective of strengths and weaknesses in specific skills. In this section we will review recent literature pertaining to the neurobiology of DS and consider the implications of these new findings within the perception, cognition, and action framework. Over the past decade, there have been unparalleled advances in the application of molecular genetics and neuroimaging techniques to developmental disabilities in general and to DS in particular. These advances are providing new information regarding our understanding of brain function in DS. Contrary to the reported studies in the 1980s and early 1990s that suggested that at birth the brains of infants with DS are structurally within the normal range of typical infants, there is increasing evidence of abnormalities in the fetal DS brain. Freidl, Gulesserian, Lubec, Fountoulakis, and Lubec (2001) reported deficits of transcription, splicing, and elongation factors in the fetal DS. Kircher, Kim, Fountoulakis, and Lubec (2002) found a significant reduction in fetal brain proteins that are necessary for growth and differentiation of the brain. At birth there are several neuroanatomical differences observed. These include reductions in brain weight, smaller cerebellum and frontal lobe, and narrowing of the superior temporal gyrii (Becker, Mito, Takashima, & Onodera, 1991; Kaufmann & Moser, 2000). Compared to age-matched controls, brain volume is

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smaller (Raz et al., 1995). Changes in the neuronal structures have also been observed and include smaller numbers of neurons in the cerebral cortex, decreased basilar dendrites, and decreased dendrites in the visual cortex (Takashima, Becker, Armstrong, & Chan, 1981; Wisniewski, 1990). Within the first year of life additional structural differences include reduced volume in the frontal cortex, superior temporal gyrus, brain stem, cerebellum, and hippocampus. These and other previous structural imaging and autopsy reports highlight that there are in fact, significant differences in the structure of the DS brain at birth. The cause of these developmental differences is still unclear, although there is increasing evidence of abnormalities in fetal protein and neurotransmitters early during gestation, which may be related to the documented abnormalities during neurogenesis (Teipel & Hampel, 2006). From early childhood through young adulthood (5–23 years of age), the brains of children and adults with DS show smaller overall volumes, smaller cerebellar volume, and larger adjusted volumes of subcortical and parietal gray matter and temporal white matter (Pinter, Eliez, Schmitt, Capone, & Reiss, 2001). With age, there are additional structural changes that are superimposed on the DS brain. Significant decreases in gray matter in bilateral parietal, frontal, occipital, temporal cortex, and the parahippocampal gyrus have been reported (Teipel et al., 2004; White, Alkire, & Haier, 2003). Recently, Head, Lott, Patterson, Doran, and Haier (2007) have shown that there appears to be a compensatory response that occurs in the brain in individuals with DS, prior to the development of dementia. Middle-aged adults without dementia were studied with positron emission tomography (PET) to evaluate the cerebral glucose metabolic rate, and correlated with gray matter volume with magnetic resonance imaging (MRI). Interestingly, in comparison to controls, the DS group showed increased cerebral glucose metabolic rate in the temporal/entorhinal cortex and in the parahippocampal gyrus. Head et al. suggest that the increased metabolism may be evidence for compensatory responses related to neuronal sprouting. Whether these changes are functional remains to be determined; however, this evidence of plasticity in the aging DS brain is an important finding and suggests that there may be a greater potential for reorganization in the DS brain that previously thought. Taken together, the anatomical differences summarized above provide a very general picture of the structural development of the brain in DS. They do not, however, tell us about the functional relationship between these multiple, atypical brain systems, their interactive functional integrity, and corresponding cognitive processes. For example, many behavioral studies have highlighted that visuospatial processing is a strength in DS and consequently visual rather than verbal instructions are more beneficial for imitation and motor performance (Elliott, Weeks, & Gray, 1990). This


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behavioral pattern has been corroborated by studies showing that the areas in the brain responsible for visual spatial processing are spared—that is, occipital and parietal regions (Pinter et al., 2001). However, using functional imaging, Karrer, Karrer, Bloom, Chaney, and Davis (1998) found significant differences in event-related potentials during a visual recognition memory paradigm. They found significant differences in the infants with DS in the amplitude and area of the responses. In addition, they found that there was less decrease in the event-related potentials as a function of experience with the visual stimuli, suggesting deficits in visual adaption. Hill Karrer et al. suggest that these differences are indicative of decreased efficiency of memory processes early in development. Interestingly, no differences in attention behavior were observed in spite of significant differences in neural responses. The view that infants with DS are within the “normal range” in their development during the first few months of life (cf. Fidler & Nadel, 2007) may be the due to the lack of sensitive measures for assessing cognitive function early in infancy (Karrer, Karrer, Bloom, Chaney, & Davis, 1998). Even in later infancy and childhood when structural differences in the brain are observed, there are significant gaps in our understanding of the functional significance of these differences and in particular how these differences are related to the dynamics of neural activity and development.

6. Neural Mechanisms Underlying Perception, Cognition, and Action Although Piaget (1952) and William James noted the importance of the relationship between perception/cognition and action, it is only recently that a possible neural mechanism for this relationship was discovered. This neural substrate named the “mirror neuron system” was first discovered in the inferior prefrontal cortex (area F5) and inferior parietal lobule in the monkey. Cell units in these areas were found to discharge both when the monkey produced an object-related action and when it observed another monkey or an experimenter performing the same or similar action (Gallese, Fadiga, Fogassi, & Rizzolatti, 1996; Rizzolatti, Fadiga, Gallese, & Fogassi, 1996). This discovery has led to the hypothesis that action understanding may be achieved by mapping the visual representation of the observed action onto the observer’s motor representation for the same actions (Rizzolatti et al., 2001). Functional neuroimaging evidence now suggests that the inferior parietal lobe and inferior frontal gyrus may also be involved in both action observation and production in humans. Observation and

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execution of grasping movements or simple finger movements has been shown to activate Brodmann area (BA) 44 or 45 in a number of PET, fMRI (functional MRI), and magnetoencephalography studies (Buccino et al., 2001; Grafton, Arbib, Fadiga, & Rizzolatti, 1996; Iacoboni et al., 1999). These findings are consistent with the possibility that human prefrontal cortex contains a mirror neuron system similar to that described in the monkey (Gallese et al., 1996). The mirror neuron system appears to be somatotopically organized (i.e., there is a spatial arrangement of neurons in the brain that corresponds to the sensory-motor regions of the body). Buccino et al. (2001) found that observation of video sequences presenting actions performed by different parts of the body (i.e., the mouth, hand, and foot) resulted in activation of different regions in the premotor cortex and Broca’s area. As in the actual executions of actions, during action observation specific somatotopically organized frontoparietal circuits were recruited. This mirror neuron system is thought to play a crucial role in how we understand the goal-directed actions of others (Rizzolatti et al., 1996). Inferences about the mental states of others are thought to occur via simulation of the actions and emotions within the observer’s own neural systems and it is through this perceptual experience that the intentions and thoughts of others are perceived and understood.

7. Perception–Action Coupling in DS We recently investigated the role of the mirror neuron system in DS. Adult participants with DS and 10 participants without DS were asked to make self-paced movements consisting of reaching with the dominant hand to grasp and lift a cup. Rather than using static picture or video clips depicting an isolated motion, we had an experimenter sit beside the subject and perform a reach to grasp motion with a coffee cup. Participants therefore viewed both their own actions and the actions of the experimenter from the same perspective and within a real-world context. Several differences were found between the DS and control groups in both conditions. In the control group, execution of the action was characterized by a network of activity primarily in the contralateral premotor, frontal, and parietal regions. Additional significant peaks were also observed in the ipsilateral premotor region as well as the superior temporal gyrus (Fig. 6.1 top). In the DS group, we did not observe a clear hemisphere dominance during movement execution. Figure 6.1 (bottom) shows peak activity in bilateral motor, frontal, and parietal regions. In the observation condition, the average overall peak activity in the control group showed a distinct shift from contralateral to ipsilateral activations in a network of areas including the right mid-temporal gyrus, premotor, and parietal regions (Fig. 6.2 top). Of particular interest was that, in

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Movement execution

Control group

DS group

(L)

(R)

Figure 6.1 Average overall activations projected onto the surface of the cortex of the movement Execution condition. Top figures show activations for the control group and bottom figures show activations for the DS group.

the DS group, while there were strong activations observed in the right parietal, frontal, and bilateral temporal regions, no significant peak activity was observed in the motor areas (see Fig. 6.2, bottom). The most significant finding of this study was the diminished correspondence between the networks involved in action execution and action observation in the group with DS. The results from our study on action execution/observation show some similarities with those reported in autistic spectrum disorders (ASD). Children with ASD are known to have poor social skills, problems with imitation (Williams, Whiten, Suddendorf, & Perrett, 2001), and with what has been termed “theory of mind” (Baron-Cohen, Leslie, & Frith, 1985). Although perceptual-motor impairments have generally not been extensively studied in this population, the hypothesis that a dysfunction in the mirror neuron system may be the cause of impaired social cognition, has been gaining support (Welsh, Ray, Weeks, Dewey, & Elliott, 2009; Williams et al., 2001). If the mirror neuron system is indeed the neurophysiological mechanism by which we perceive and understand the external world, then the consequences of a dysfunction in this system would include difficulties not only in interpreting physical cues, but emotional and social cues as well, leading to difficulties in social competence. This study has important implications for our understanding

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Movement observation

Control group

DS group

(L)

(R)

Figure 6.2 Average overall activations projected onto the surface of the cortex for the movement Observation condition for the control group (top) and the DS group (bottom).

of the perceptual-motor coupling in DS, particularly in light of new developments in neuroscience in relation to the representation of action and the influence of action on perception and cognition.

8. The Influence of Action Experience on Perception and Cognition The previous section has highlighted the importance of actions—we act and interact within a social environment. Our ability to control our own actions and to understand the actions of others is of critical importance. Recent work from the infancy literature is leading to a resurgence of ideas on the influence of action on perception and cognition and the way in which action structures perceptual and cognitive development. These studies are briefly reviewed here. Although none of these studies have directly addressed the question of how delayed motor development impacts on perception and cognition. We will briefly review them here because the findings have direct relevance to DS and other developmental disabilities. In the first year of life, TD infants make huge strides in motor development. They progress from a limited repertoire of spontaneous and reflex movements, to more purposeful, goal-directed movements with their


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upper limbs. Moreover, they achieve greater balance in more upright positions, from sitting and crawling to standing and walking. This rate of development is influenced by a number of factors: maturation of the nervous system, individual/genetic make up, and ability to process sensory stimulations such as touch, sound, vestibular, muscle, and joint sensations, and experience with the environment. Movement experience has always been recognized as important for motor learning. However, recent evidence underscores the critical role of action experience on cognitive development. Indeed, infants rapidly learn from active experience and are able to transfer this knowledge to viewing the actions of others (Sommerville, Woodward, & Needham, 2005). The onset of locomotion is one of the major transitions in early development which results in changes not only in motor skill but also in perception, spatial cognition, and social and emotional development (Campos et al., 2000). Campos et al. (2000) conducted an exhaustive review of findings that link locomotor experience to the development of social cognition. For example, as the child becomes more autonomous there is a change in the way and manner by which the caregivers communicate. Vocal and facial signals are conveyed at a distance and require different type of processing by the infant to understand the emotional cues. Apparently, the onset of locomotion also changes the attitudes and behavior of the mother as the infant becomes more independent. As infants become more mobile and start to explore their environment, they make discoveries and learn about their own bodies as well as about objects, places, and events that may have consequences for mobile exploration. Walking has tremendous implications on all areas of development. The opportunities for exploration, play, and interaction with peers increases significantly. In addition, walking has an impact on the development of the perception of space, objects, and is a prerequisite for more advanced locomotor skills. More recently, Uchiyama et al. (2008) showed that infants who received specific experience with a mobility device responded differently in terms of postural compensation and emotional experience than infants who did not. Meltzoff and Brooks (2008) also showed that infants who had experience with an opaque blindfold were more likely to follow the gaze of a blindfolded adults compared to infants who did not receive this direct experience. Meltoff and Brooks (2008) have proposed that infants take information from their own experience and from observing the actions of others. This information is then mapped onto a shared abstract framework in which infants’ experience is informed by understanding like experiences in others. Such an ability to form abstract representations of goal-directed actions would provide infants with a powerful learning mechanism in development. Infants would, then, be able to rapidly transfer action information across modalities and from one agent to another. As a result, infants could

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now acquire a host of cognitive abilities that rely on recognizing goal structure in action.

9. Impact on Development and Intervention If, as Meltzoff and others have suggested, infants learn about the physical and social world through self-movement experience and observation of the actions of others, there are a number of issues that must be considered in relation to DS. First, there are well-documented delays in motor development and postural control in infants and children with DS (see Virji-Babul & Latash, 2008 for review; Health et al, 2000). These motor delays will likely limit motor experiences and motor exploration. The question of whether and how these delays impact on perception and cognition has not been studied directly. However, given the findings in the TD literature, the logical assumption would be that motor delays would negatively impact on the overall movement experiences available to the infant with DS and this in turn will lead to some degree of change in perception, spatial cognition, and social and emotional development. In terms of intervention, it would be too simplistic to propose that intervention strategies should focus on facilitating movement and enriching self-movement experiences. Campos et al. (2000) have shown that while locomotor experience is a key feature of developmental change, it is not necessary or sufficient to bring about such change. In many instances, the infants they investigated already showed evidence of perceptual/cognitive skill development prior to the onset of locomotion. These skills were heightened following the onset of locomotion. In other words, developmental processes related to cognitive functions must be in place in order for locomotion to exploit these functions. The question is whether infants with DS who receive movement-based interventions that have been shown to accelerate the onset of motor skills, show a concomitant change in cognition. Ulrich, Ulrich, Angulo-Kinzler, and Yun (2001) reported that a partial-body-weight-support treadmill training paradigm significantly advanced the onset of independent walking by about 3.5 months in infants with DS. It would be interesting to track changes in cognitive task performance as a function of these movementbased interventions. Another important point to consider is the relationship between behavior and the underlying neural responses. We recently investigated the neural responses to simple voluntary, self-paced right index finger movement in adults with DS (Virji-Babul, et al, 2009). In the control group, the most significant activations were found in the left primary motor and sensory-motor regions as has been reported in numerous


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studies (Cheyne, Bakhtazad, & Gaetz, 2006; Cheyne, Weinberg, Gaetz, & Jantzen, 1995). Smaller but also significant activity was also observed in the ipsilateral motor region. As expected, we observed a clear left hemisphere dominance for the control of these simple movements made by right-handed participants. In contrast, the DS group showed a right hemisphere dominance despite the fact that all the participants were right handed. Considerable behavioral research in DS suggests a left hemisphere dominance for the control of movements, similar to the typical population. What is the significance of the atypical right hemisphere dominance for the control of movements in DS? One possible explanation is that this shift may be the result of sensory-motor reorganization resulting from changes during the course of development. There is now an extensive literature showing the remarkable ability of the nervous system to reorganize following injury based on continuous sensory experience (Merzenich et al., 1983; Mogilner et al., 1993). It is possible that as development progresses and structural abnormalities begin to emerge as a consequence of the expression of the genetic abnormality, there are accompanying attempts at neural reorganization to compensate for the structural and functional changes. We are currently expanding this study to include children to determine whether such a shift occurs early in development. Other factors critical to effective interventions are motivation, learning styles, and strategies. The previous section reviewed the literature on imitation and clearly indicated that children with DS show an atypical reliance on imitation which may be detrimental for tasks that require more concentrated levels of engagement. Previous behavioral studies have also shown that infants with DS manipulate toys less often and show more passive behavior in response to their mothers’ efforts to direct attention (Landry & Chapieski, 1990). During free play, children with DS spend more time in passive behavior, shift back and forth from play to passivity more often than typical children (Linn et al., 2000), display shorter sequences of goal-directed tasks, and higher rates of toy rejection (Ruskin, Kasari, Mundy, & Sigman, 1994). In a comparative observational study on the spontaneous play of infants, Mundy, Sigman, Kasari, and Yirmiya (1988) reported that the children with DS made fewer requests for either objects or assistance, and seemed to be less motivated to explore or to request information. In videotaped sequences of mother–child play with infants and toddlers from 20 to 76 months, Beeghly et al. (1990) also observed similar characteristics. In group play, Sigman and Ruskin (1999) showed that children with DS displayed no preference for social over nonsocial play and were less receptive of social initiations in comparison with typical peers. Children with DS have also been shown to be more apt to reject another child’s initiation into play by pushing the child or walking away (Schlottman & Anderson, 1975).

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These children also have difficulty understanding and interpreting social situations, even if a peer continues to make repeated social initiations for play often leading to social isolation (Sinson & Wetherick, 1981). Requesting toys and negotiating with other children about desired toys or objects are also skills that are challenging for children with developmental disabilities (Guralnick, 1995). Parents often describe their children with DS as less persistent than their TD peers, and this may deter the ability to negotiate with peers and request objects or toys from playmates (Spiker, Boyce, & Boyce, 2002). In addition, a number of researchers have reported that children with DS overuse their social skills in order to compensate for their weaknesses in others areas of function (Fidler, 2006). Kasari and Freeman (2001) showed, for example, that children with DS tended to show more off-task social behaviors during a puzzle task than their mental age-matched peers. The use of such social behaviors to avoid cognitively challenging tasks may have negative repercussions for later learning (Fidler, Most, Booth-LaForce, & Kelly, 2008). Given the challenges outlined above, how can intervention be structured to enhance/maximize the complex processes associated with perception-action and cognition? Clearly, these processes are inexorably linked and attempts to separate each process and intervene in an isolated, domainspecific manner will be challenging if not counterproductive. Iarocci, VirjiBabul, and Reebye (2006) recently reported on an innovative intervention program with a focus on functional skills within ever-widening circles of emotional attachment. The Learn At Play Program prioritizes the goals of nurturing and shaping the development of interpersonal skills and social competence among children with DS. Within this framework, the initial developmental tasks involved maximizing the quality of early dyadic interactions between infants with DS and their parents within an active play context. As the children develop, tasks are modified to reflect developmentally appropriate goals that emphasize the social and emotional skills that are essential for children during the preschool years and the transition into formal schooling. Interventions are designed to target the specific domains (e.g., motor, language, short-term memory) that are typically affected by DS within the broader context of social competence goals. While the initial results seem promising, the impact of this type of program on development across a broader timescale remains an ongoing research goal. The findings from the research reviewed here highlight some important principles for intervention. Developmental change and progress must be considered as a dynamic process involving rich and complex interactions between different areas of function. As such, assessment must be continuous and involve multiple levels of analysis: information processing ability, the impact of the social environment, and the neural substrates underlying behavior. In terms of intervention, the importance of environments rich


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in motion and emotion cannot be overemphasized. This will naturally entail observing children in more naturalistic environments such as playgrounds where action is natural and where perception and cognition can intersect. From a research perspective, what is needed are new approaches based on models that consider the interaction between systems. For example, Tamis-LeMonda et al. (2008) recently conducted an interesting study to examine how toddlers use perceptual and social information to guide their decisions to actions to walk down slopes of different risk levels. The infants’ mothers either encouraged or discouraged the infants. Their findings indicate that the toddlers ignored their mothers’ advice to walk down slopes that were risky, and ignored advice about avoiding slopes that were safe. Interestingly, they took the advice at slopes that might be considered borderline, suggesting that social information took precedence when perceptual information was ambiguous. How would toddlers with DS behave under these conditions given their altered motor abilities, their unusual reliance on the face of their caregiver, coupled with their inability to extract the appropriate cues from the physical and social environment? The development of such multifaceted paradigms that combine perception, cognition, and action in combination with behavioral and neural measures, will require a dramatic shift of perspective. It will also require a collective effort to take the strengths from disparate traditions to ask new and interesting questions about how individuals with DS perceive the world, and how we can best facilitate their engagement within their physical and social space.

10. Conclusion The study of motor control has undergone significant changes over the past few decades as new evidence has emerged showing that action perception and action production are not only tightly linked to each other, but also influence each other. The discovery of “mirror neurons” that respond both when the individual executes an action and while he/she sees or hears another individual performing a similar action, has provided the neurophysiological mechanism for linking perception with action. This mirror neuron system is thought to play a crucial role in how we understand the goal-directed actions of others, develop imitation skills, and in motor learning (Rizzolatti et al., 1996). More importantly, the development of the mirror neuron system appears to be experience dependent, possibly through a process of repeated observation of self-produced movements, that links visual and sensory-motor representation of actions (Del Giudice et al., 2009). In other words, sensory-motor experience may in fact, modulate the mirror neuron system.

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Given the well-documented pattern of movement difficulties in individuals with DS, it is reasonable to hypothesize that perceptual and motor processes may, to some extent, be decoupled in these individuals. We have reviewed evidence in this chapter that suggests this may indeed be the case. However, intervention strategies for individuals with developmental challenges are still primarily based on our understanding of typical developmental trajectories. The challenge for interventionists working in the field of developmental disabilities is to move away from the traditional developmental model and to view development within a broader context of both physical and social experiences. If it is true that visual-motor cortical connections are established through the correlated experience of action observation and execution, our task is to reconceptualize intervention for individuals with DS with a major focus on actively engaging both their motor and social networks.

Acknowledgments We would like to acknowledge all the individuals and families that have supported our research at the Down Syndrome Research Foundation. Thanks also to H. Mahmood, S. Fawcett, G. Chen, J. Hayden, T. Lalani, S. Poonja and A. Jobling for their comments and help with the manuscript.

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C H A P T E R S E V E N

Agenesis of the Corpus Callosum: A Literature Review Shihfen Tu, Donna Doherty, Kathryn J. Schilmoeller, and Gary L. Schilmoeller* Contents 1. Introduction 1.1. Prenatal development 1.2. Disorders of the corpus callosum 1.3. Causes and diagnosis 1.4. Prevalence 1.5. Associated conditions 2. Physical/Health-Related Issues 2.1. Concurrent medical conditions 2.2. Sensory abnormalities 2.3. Daily living 2.4. Developmental outcomes 3. Social/Emotional/Behavioral Traits 3.1. General descriptions 3.2. Other behavioral or emotional disorders 4. Language/Communication/Cognitive Characteristics 4.1. Language development 4.2. Pragmatics/social communication 4.3. Cognition 5. Therapeutic Interventions for Individuals with ACC 5.1. Common themes 6. Conclusion and Future Directions 6.1. Summary 6.2. Future directions References

172 173 173 174 175 175 176 176 177 178 178 180 180 181 181 181 182 183 185 185 187 187 189 190

In this chapter, we will review recent literature on the condition of agenesis of the corpus callosum (ACC), in an effort to promote general understanding of this condition as well as to provide relevant findings most applicable to *

College of Education and Human Development, The University of Maine, Orono, Maine



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educational and service providers. We begin with a brief overview of the condition, its biological basis, types, and potential health-related consequences before turning to a discussion of its broad developmental and intellectual impact. We will summarize the current understanding of the effect of these callosal conditions on communication, cognition, and behavior. In addition, we will discuss the challenge of determining the most appropriate services and supports for early intervention, special education, and lifelong assistance. Finally, we will call attention to questions related to our current understanding and treatment for persons with ACC and identify important areas for future study.

1. Introduction

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The corpus callosum is the major structure of the human brain that connects the two cerebral hemispheres. It is a broadband of more than 200 million nerve fibers that provide the main route for transfer and integration of information between the two hemispheres (Kamnasaran, 2005; Shonkoff & Marshall, 2000; Windhorst, 1996). Please see Fig. 7.1 for an illustration of the

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Figure 7.1 Medial view of a halved human brain. From http://en.wikipedia.org/wiki/ File:Gehirn,__medial__-__beschriftet__lat.svg (Permission to copy granted under the terms of GNU Free Documentation License. This file is licensed under the Creative Commons Attribution ShareAlike 3.0 License.)

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structure. Studies of brain lateralization suggest that each hemisphere of the brain performs specialized functions. The corpus callosum provides the means for integrating the information from each hemisphere in order to perceive, comprehend, and act fully upon sensory input. Although there are other smaller commissures (cross-hemisphere connections) in the brain, those are comprised of significantly fewer nerve fibers than the corpus callosum. Thus, absence of the corpus callosum is considered a major brain anomaly (Mercer, 1998; Shonkoff & Marshal, 2000; Windhorst, 1996).

1.1. Prenatal development Typically the corpus callosum begins to develop very early in prenatal life. The lamina reuniens, which serves as a precursor to the corpus callosum, forms as early as the sixth to eighth week of pregnancy (Kamnasaran, 2005; Shonkoff & Marshall, 2000; Windhorst, 1996; Wisniewski & Jeret, 1994). Sometime around the 10th or 11th week of gestation, callosal nerve fibers begin to cross the midline to connect the cerebral hemispheres. These connections appear in the anterior portion of the brain first, forming the genu, with the body and splenium of the corpus callosum developing later, from the front to back. At about 18–20 weeks gestation, the most anterior portion of the corpus callosum, the rostrum, develops, finalizing the formation of the structure (Kamnasaran, 2005; Lassonde, Sauerwein, & Lepore, 2003). Once in place, these fibers continue to mature throughout pregnancy. Full maturation and myelination of callosal fibers continue throughout childhood and adolescence (Kamnasaran, 2005; Ramaekers, 1991; Shonkoff & Marshall, 2000; Windhorst, 1996).

1.2. Disorders of the corpus callosum Any disruption to the typical sequence of growth of these nerve fibers or their precursors can lead to a failure of the corpus callosum to develop normally (Kamnasaran, 2005; Njiokiktjien, 1991). Depending on the timing and cause of the disruption, the structural abnormality may be partial (p-ACC) or complete (c-ACC). In some cases, the corpus callosum is present, front to back, but abnormally thin. This is sometimes referred to as hypoplasia. Researchers are working to better understand the similarities and differences in outcomes based on the type of callosal condition. Individuals with congenital absence of the corpus callosum appear to be affected differently than those whose corpus callosum is severed in an attempt to control intractable seizures (i.e., commissurotomy, see Lassonde et al., 2003; Paul et al., 2007). While patients who undergo commissurotomies often exhibit a “disconnection syndrome” characterized by the inhibition of sensory information being transferred across hemispheres and the impediment of some bimanual motor tasks, individuals with ACC perform better on tasks related to interhemispheric integration. It is possible that individuals with a

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congenital absence of the corpus callosum may benefit from early neural plasticity, which allows alternate neural pathways to develop, although the extent of this compensation is limited (Paul et al., 2007).

1.3. Causes and diagnosis It is often difficult to pinpoint with certainty the exact cause for a callosal abnormality, but early prenatal insults or injuries, such as maternal infections, toxins, or asphyxia are sometimes suspected (e.g., Jeret, Serur, Wisniewski, & Lubin, 1987; Shevell, 2002; Wisniewski & Jeret, 1994). In certain cases, prenatal or postnatal genetic testing can identify a chromosome abnormality (Fratelli et al., 2007; Paul et al., 2007; Serur, Jeret, & Wisniewski, 1988). Agenesis of the corpus callosum sometimes occurs in association with fetal alcohol syndrome or underlying metabolic disease in the developing fetus (Jeret et al., 1987; Marszal et al., 2000; Prasad et al., 2007; Wisniewski & Jeret, 1994). In one recent study, researchers found that mothers over 40 years of age were found to be at increased risk for having children with a callosal condition in association with a chromosomal abnormality such as Patau syndrome, Edward syndrome, and Down syndrome (Glass, Shaw, Chen, & Sherr, 2008). See Table 7.1 for a summary of risk factors. Callosal disorders are diagnosed by ultrasonography, computerized tomography (CT), or magnetic resonance imaging (MRI). Currently, radiologists and neurologists consider MRI the most accurate and reliable diagnostic technology in detecting callosal disorders because it provides the most information about the corpus callosum (Fratelli et al., 2007; Glenn et al., 2005; Hetts, Sherr, Chao, Gobuty, & Barkovich, 2006). Today, prenatal specialists can diagnose a fetus with a callosal abnormality as early as 20–22 weeks gestation (Fratelli et al., 2007; Glenn et al., 2005; Moutard et al., 2003). Callosal abnormalities are not always identified prenatally, however, and some newborns and young infants are diagnosed after birth during assessments for other visible or suspected birth defects (e.g., Table 7.1 Risk factors for agenesis of the corpus callosum

Risk factor

Example

Environmental factors

Maternal infections, toxic exposures (e.g., fetal alcohol exposure), asphyxia Trisomy 13, 15, 18; Andermann syndrome, Aicardi syndrome

Chromosome abnormalities/ genetic syndromes Inborn errors of metabolism Increased maternal age

Nonketotic hyperglycinemia, mucolipidosis Over 40 years


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hydrocephaly), medical complications (e.g., seizure activity), or developmental delays. Children and adults have also been identified later in life, especially during diagnostic procedures for head injuries, headaches, seizure activity, or other medical, neurological, or behavioral concerns (Schilmoeller & Schilmoeller, 2000; Shevell, 2002; Sorensen, 1997; Taylor & David, 1998).

1.4. Prevalence The reported prevalence of callosal conditions varies depending on the population studied and technology available at the time. Historically, the prevalence has been reported as high as 3–7 per 1,000 in the general population and 2–3 per 100 among individuals with developmental disabilities (Jeret et al., 1987; Wisniewski & Jeret, 1994). However, the estimated prevalence rates from more recent studies are lower. For example, based on a review of the recent neonatal and prenatal imaging studies, ACC appeared to occur in 1 in 4,000 live births (Paul et al., 2007). However, results from a study utilizing the California birth defect registry from 1983 to 2003 indicated a lower overall birth prevalence of callosal malformations as 1.8 per 10,000 live births (Glass et al., 2008). Yet, these more recent studies do not reflect those cases diagnosed after the first year of life, pregnancies that were interrupted due to stillbirth or termination, or those individuals that are not diagnosed. It is not clear how many children and adults with callosal abnormalities go undiagnosed or misdiagnosed, but one study suggests that these underdiagnoses may be common. In a retrospective study of 90 children with idiopathic developmental delay at the Hospital for Sick Children in Toronto, 40 (44%) were identified with undiagnosed callosal abnormalities revealed during diagnostic brain imaging (Widjajay, Nilsson, Blaser, & Raybaud, 2008). Notably, in 2003 a subcommittee of the American Academy of Neurology recommended that neuroimaging, such as MRI, be included as part of the evaluation of children with global developmental delays when certain criteria are met (Shevell et al., 2003). It is likely that the more common use of diagnostic technologies will help answer the question of prevalence and may lead to early and more frequent identification of children with callosal conditions. See Table 7.2 for a summary of prevalence estimates.

1.5. Associated conditions Although ACC can occur as an isolated condition (sometimes referred to as “primary ACC” or “isolated ACC”), it is frequently associated with other brain or central nervous system abnormalities and genetic or medical conditions that also affect health and development (e.g., Doherty, Tu, Schilmoeller, & Schilmoeller, 2006; Glass et al., 2008; Jeret et al., 1987; Shevell, 2002; Wisniewski & Jeret, 1994). Based on a review of the literature, some researchers suggested that 30–45% of those with ACC have identifiable chromosome abnormalities or genetic syndromes, which

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Table 7.2 Prevalence reported by various studies with the rates per 10,000 included in parentheses

Study

MR/DD population

General population

2.2–2.4% (220–240)

1–3 per 1,000 (10–30) 0.0005–0.7% (0.05–70)

Jeret et al. (1987) Wisniewski & Jeret (1994)

Live births

Paul et al. (2007)

1 per 4,000 (2.5)

Glass et al. (2008)

1.8 per 10,000 (1.8)

Widjajay et al. (2008)

44 per 100 (4400)

may be an inherited trait or a new mutation (Paul et al., 2007). Several syndromes, such as Aicardi, Andermann, and Shapiro, are often associated with ACC; however, ACC also occurs infrequently with many more syndromes such as Chiari’s syndrome, Dandy–Walker’s syndrome, Apert syndrome, neurofibromatosis, and fetal alcohol syndrome (see Lassonde & Jeeves, 1994 or Paul et al., 2007 for discussions). The effect of these syndromes on individuals ranges from mild to severe depending on the constellation of features associated with the particular syndrome. Additional brain abnormalities are also common in individuals with a callosal disorder. Hydrocephaly and microcephaly are reported frequently (Glass et al., 2008; Goodyear, Bannister, Russell, & Rimmer, 2001; Jeret et al., 1987; Shevell, 2002; Wisniewski & Jeret, 1994). Other abnormalities (e.g., cortical malformations, posterior fossa abnormalities) that may affect health and intellect are also described (Glass et al., 2008; Hetts et al., 2006). Mental retardation often is reported, but the prevalence varies widely depending on the population studied, which can include individuals with “isolated ACC” as well as those with ACC and additional brain abnormalities or syndromes (e.g., Doherty et al., 2006; Goodyear et al., 2001; Serur et al., 1988).

2. Physical/Health-Related Issues 2.1. Concurrent medical conditions Compared to the general population, persons with ACC are more likely to have physical and structural abnormalities outside the central nervous system.


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Head and oral–facial defects, such as cleft lip or palate and facial asymmetries, are sometimes present, although the reported prevalence varies broadly (Doherty et al., 2006; Goodyear et al., 2001; Jeret et al., 1987; Marszal et al., 2000; Serur et al., 1988; Shevell, 2002; Wisniewski & Jeret, 1994). Some individuals experience motor impairments such as hypotonia, spasticity and coordination problems, and cerebral palsy (Doherty et al., 2006; Schilmoeller & Schilmoeller, 2001; Serur et al., 1988; Shevell, 2002). Other less commonly associated conditions include cardiac and genitourinary anomalies (Bedeschi et al., 2006; Glass et al., 2008; Goodyear et al., 2001) and costo-vertebral defects such as spina bifida or scoliosis (Bedeschi et al., 2006; Doherty et al., 2006; Jeret et al., 1987; Wisniewski & Jeret, 1994). Epilepsy and seizures are more common in children and adults with callosal conditions, with the reported prevalence varying from 27.3 to 85.7% depending on the population studied (Doherty et al., 2006; Goodyear et al., 2001; Jeret et al., 1987; Serur et al., 1988; Shevell, 2002; Taylor & David, 1998; Wisniewski & Jeret, 1994). For example, in a survey of parents of children with ACC who contacted the ACC Network (an international support organization for families), 27.3% reported a seizure condition diagnosed by a medical professional (Doherty et al., 2006). A much higher prevalence (85.7%) was reported in a population of children with ACC from a pediatric neurology clinic (Marszal et al., 2000). This difference may be attributed to the fact that children with seizure disorders will more likely be under the care of a neurologist.

2.2. Sensory abnormalities Individuals with ACC often have sensory deficits or abnormalities, with vision issues being the most commonly reported (Doherty et al., 2006; Goodyear et al., 2001; Marszal et al., 2000; Schilmoeller & Schilmoeller, 2000, Serur et al., 1988). Specific vision problems include near- and farsightedness, strabismus, nystagmus, and problems with depth perception. Hearing deficits, both mild and profound, have also been reported, sometimes with atypical patterns over time (Doherty et al., 2006; O’Brien, 1994; Schilmoeller & Schilmoeller, 2000; Serur et al., 1988). In a longitudinal case study documenting the assessment and treatment of a young girl with ACC, she was initially diagnosed with severe hearing loss and vision impairments (Skinner & Hickson, 2002). Over a 4-year period, this child’s vision and hearing abilities matured to near-normal abilities. This phenomenon has been reported anecdotally in other infants and young children as well. Based on parental reports, some children with ACC have abnormal reactions to touch and pain (Doherty et al., 2006; Schilmoeller & Schilmoeller, 2001). In a survey of caregivers of 189 individuals with ACC and their siblings (Doherty et al., 2006), 46.7% of individuals with ACC were described to have tactile defensiveness and were “more” or “much more” sensitive to touch than would be expected. Interestingly, while those

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with ACC may be more sensitive to touch, they may also have a higher tolerance for pain. About 56.6% of the respondents in the same study indicated that their child with ACC had little or less pain perception than would be expected (Doherty et al., 2006). Parents of children with ACC also report this phenomenon anecdotally, including serious injuries (e.g., broken bones, burns) and illnesses (e.g., burst eardrums, appendicitis) that go undetected until much later than would be expected. Awareness of the potential for undetected conditions to occur is important and, as yet, largely unstudied.

2.3. Daily living Other developmental and health-related issues also affect the daily living of children with ACC. Researchers report early sucking, chewing, and swallowing difficulties, and esophageal reflux (Doherty et al., 2006; Ng, McCarthy, Tarby, & Bodensteiner, 2004; Sorensen, 1997; Stickles, Schilmoeller, & Schilmoeller, 2002). Children with ACC also often accomplish toilet training at a much later age than their typically developing siblings. In the previously noted survey of 189 caregivers of children with ACC (Doherty et al., 2006), 40.5% of those over the age of 5 years were not yet toilet trained, which can be problematic when they reach school age. In the same study, 45.5% of the children over the age of 5 years experienced bedwetting (enuresis) either occasionally or frequently. Individuals with ACC often experience sleep problems as well, including difficulty getting to sleep and frequent nighttime waking (Doherty et al., 2006; O’Brien, 1994; Schilmoeller & Schilmoeller, 2000). Lack of restorative sleep or interrupted sleep may be of particular interest since recent literature suggests an association between disordered sleep and behavioral problems or learning difficulties (Anstead, 2000).

2.4. Developmental outcomes When assessing newborns and children for developmental progress, healthcare providers should be aware of the potential for delays in this population so that needed evaluations and services can be provided. Developmental delays are quite common among children with ACC, with the reported prevalence ranging from 60.8 to 80.7% of those studied (e.g., Doherty et al., 2006; Goodyear et al., 2001; Schilmoeller & Schilmoeller, 2000; Shevell, 2002). Some researchers suggest children with isolated ACC have better developmental outcomes than those with ACC and additional conditions (Francesco, Maria-Edgarda, Giovanni, Dandolo, & Giulio, 2006; Moutard et al., 2003; Ramelli, Zanda, Wyttenbach, Bronz, & Schnider, 2006). Yet a retrospective study of prenatally diagnosed children with ACC (n = 117) found that 36% of those diagnosed with isolated ACC exhibited significant developmental delays (Fratelli et al., 2007).


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Researchers rarely address specific domains of developmental delay, but some provide clues. For example, motor skills frequently are delayed or impaired in children with ACC (Goodyear et al., 2001; O’Brien, 1994). Some children may be delayed in achieving such developmental milestones as walking and in manual dexterity, like using a pencil (O’Brien, 1994). We will address language and cognitive development and other social and behavioral outcomes in later sections. Finally, many researchers agree that it is difficult to predict developmental outcomes in infants and children with ACC; close monitoring and screening for emerging developmental and educational needs is recommended (Chadie et al., 2008; Fratelli et al., 2007; Moutard et al., 2003; Ramelli et al., 2006). For health-related issues and associated conditions, see Table 7.3. Table 7.3 Health-related issues and other conditions associated with agenesis of the corpus callosum

Concurrent medial conditions

Sensory abnormalities

Motor impairments Eating/feeding impairments Elimination Sleep issues Developmental delays

Chromosomal abnormalities and syndromes – Aicardi syndrome, Andermann syndrome Central nervous system abnormalities – hydrocephaly, microcephaly, cortical malformations Mental retardation Epilepsy/seizures Costo-vertebral defects – spina bifida, scoliosis Head and oral-facial defects – cleft lip and palate Cardiac anomalies Genitourinary anomalies Visual disturbances – near/far sightedness, strabismus, nystagmus, depth perception issues Hearing deficits—mild to profound Increased sensitivity to touch Unusual tolerance to painful stimuli Hypotonia, spasticity, coordination problems, cerebral palsy Early feeding problems, esophageal reflux, swallowing difficulties Late toilet training, enuresis, constipation Difficulty falling asleep, frequent nighttime waking Delays in meeting motor milestones, speech delays, cognitive delays

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3. Social/Emotional/Behavioral Traits 3.1. General descriptions Some researchers have described individuals with ACC as happy, friendly, and sometimes immature for their age (e.g., Brown & Paul, 2000; Stickles et al., 2002). Parents and researchers have also observed children, adolescents, and adults with ACC as having difficulties in social and emotional development (Badaruddin et al., 2007; Schilmoeller & Schilmoeller, 2000). Although they enjoy being with others, persons with ACC may have particular trouble in developing and sustaining close friendships with peers of the same age and may prefer interacting with younger children (Badaruddin et al., 2007; Sorensen, 1997; Stickles et al., 2002). Such poor peer relations may reflect an inability to process and interpret social and emotional signals, a pattern observed in psychological testing and consistent with what some parents have reported as trouble discerning their children’s feelings (Brown & Paul, 2000; O’Brien, 1994; Paul, Van LanckerSidtis, Schieffer, Dietrich & Brown, 2003). For example, in one study where two adolescent males with ACC were administered a battery of cognitive and psychosocial tests, compared to the control group, both scored the lowest on the Thematic Apperception Test (Brown & Paul, 2000). They were shown six cards based on which they were asked to tell a story with a beginning, a middle, and an end. The two young men were also told to explain the thoughts, feelings, and actions of the characters. The investigators reported that the two adolescents gave inadequate logic for their stories and showed little complexity of social understanding. The adolescents also failed to mention the content that is usually brought out by the cards and tended to overlook the emotional content and social implications of the testing materials. The researchers also observed that these two young men were somewhat unsophisticated in their self-awareness and were socially na¨ıve. The authors suggested that individuals with ACC may be more successful in routine social exchanges and interactions, but have more difficulties in new situations, where complex or abstract thought processes are important. Some researchers have observed subtle behavioral characteristics, such as the tendency to perseverate, exhibited by individuals with ACC (Brown & Paul, 2000; Schilmoeller & Schilmoeller, 2001). In a survey of parents of individuals with ACC and their siblings (n = 118 pairs), caregivers rated persons with ACC (compared to their siblings) as more likely to perseverate (Tu, Schilmoeller, Schilmoeller, Doherty, & Moes, 2008). In addition, they reported that it was more difficult to read the emotional state of the individuals with ACC, who were also rated as more likely to show unusual facial expressions, to make unusual gestures, and to have more difficulty using appropriate personal space.


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3.2. Other behavioral or emotional disorders Extreme emotional or behavioral disorders are rarely reported among individuals with ACC; however, conditions such as depression or anxiety disorders have been observed (Párraga, Párraga, & Jensen, 2003; Taylor & David, 1998). In one case study, an 11-year-old boy with ACC reportedly showed aggression and suicidal ideation, with prior diagnoses of depression, attention deficit/hyperactive disorder, and oppositional defiant disorder (Panos, Porter, Panos, Gaines, & Erdberg, 2001). In contrast to severe problems, individuals with ACC are more likely to show emotional and behavioral problems that are of lesser severity (Doherty et al., 2006; O’Brien, 1994; Schilmoeller & Schilmoeller, 2000). For example, some have temper tantrums (found in 23.4% of 47 individuals in O’Brien, 1994) and exhibit inflexibility handling change in routines (13.2% of 596 individuals in Schilmoeller & Schilmoeller, 2000). In addition, compared to their siblings, persons with ACC were rated as more likely to show rapid mood changes and stubbornness (Doherty et al., 2006). Even though a few studies indicated that some children with ACC exhibited autistic-like behaviors, it is rare that these children received a formal diagnosis of autism (Badaruddin et al., 2007; Doherty et al., 2006; O’Brien, 1994; Schilmoeller & Schilmoeller, 2000). In a study in which 61 children with ACC (ages 2–11) were compared to a group of 52 children with autism (ages 6–11), both groups had difficulties with social interactions and communication, but children with ACC did not frequently display the repetitive or restrictive behaviors (e.g., spinning, lack of interest in others) more commonly seen in children with autism (Badaruddin et al., 2007). Similarly, children with ACC usually do not receive a diagnosis of ADD, ADHD, or Asperger syndrome. However, compared to their siblings, they are more likely to be diagnosed as having a learning disability, developmental delay, or mental retardation (Doherty et al., 2006).

4. Language/Communication/Cognitive Characteristics 4.1. Language development Individuals with ACC typically have language delays and communication deficits (e.g., O’Brien, 1994; Sauerwein & Lassonde, 1994; Schilmoeller & Schilmoeller, 2000; Sorensen, 1997; Stickles et al., 2002). For example, in one study in which 20 prenatally diagnosed children with isolated ACC were followed for 3–16 years, investigators reported mild to severe speech delays, cognitive disorders, and abnormal neurodevelopmental testing results (Chadie et al., 2008).

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In one case study, a young girl’s development over a 10-year period was examined through observation and testing (Sorensen, 1997). Even with an ongoing intervention that began at the age of 2, the girl continued to exhibit language delay and communication deficits, primarily in her pragmatic skills. Interestingly, despite the delay in language development, the child managed to perform at the grade level in academic coursework, except for math. In another case study, Stickles et al. (2002) researchers reviewed the language development of a 23-year-old man with ACC. Despite normal intelligence and extensive intervention, this young man also exhibited language delays and deficits in social communication. Specifically, he showed an inability to take the perspective of his listener. The young man sometimes changed topics without providing the listener the reason for switching or spontaneously started a new conversation without any relevant context or reference to make his comments meaningful. However, the authors noted that his written language skills did improve over time with consistent practice. Parents and caregivers have also observed delays in language development and communication deficits in their children with ACC. In one survey mentioned earlier (Tu et al., 2008), parents indicated that almost all siblings understood messages at their corresponding age level, while less than half of the children with ACC were able to do so. These parents gave similar responses when asked about their children’s ability to engage in conversation.

4.2. Pragmatics/social communication Among some individuals with ACC, the communication deficits are primarily in the area of pragmatic skills, such as understanding jokes, nonverbal communication, and emotional communicativeness (e.g., Brown & Paul, 2000; O’Brien, 1994; Panos et al., 2001; Paul, Schieffer & Brown, 2004; Stickles et al., 2002). For example, Brown, Paul, Symington, and Dietrich (2005) compared 16 adults and adolescents (between 14 and 55 years of age) with complete ACC and normal intelligence to a control group of 31 adults and adolescents of similar age and IQ. Compared to the control group, the group with ACC had more trouble understanding the subtleties of jokes or social stories. The inability to understand subtleties is consistent with the finding that some individuals with ACC lack the comprehension of non-literal language. For instance, when a battery of language tests were administered to 10 male adults with complete ACC (ages 16–31) and a control group of 14 male adults (ages 15–28), all with IQ scores in the normal range, participants with ACC had difficulties recognizing proverb meaning and affective prosody (i.e., grasping non-literal language). However, there was no


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significant difference between the two groups with regard to comprehending literal items (Paul et al., 2003). In this case, an example of a non-literal sentence was “I’d like to give you a piece of my mind,” while “The dog’s trying to give her a ride on the wagon” was an example of a literal sentence (p. 317). While these deficits in social communication are likely to impact the peer interactions of adolescents and adults with ACC, parents have also reported that young children with ACC had difficulties initiating and sustaining conversation, and they did not understand social reciprocity and nonverbal communication of others (Badaruddin et al., 2007). Specifically, when 61 children with ACC were evaluated using DSM-IV diagnostic criteria for autism, 43% of these children were reported to have difficulty “showing social and emotional give-and-take,” while 36% had difficulty in “understanding nonverbal communication” (Badaruddin et al., 2007). In addition, investigators found other atypical communicative behaviors such as repetitive and/or meaningless talk among individuals with ACC (O’Brien, 1994; Panos et al., 2001).

4.3. Cognition As noted previously, some individuals with ACC have been observed to show difficulties in understanding complex social situations, social problem solving, and interpreting emotional content (Paul et al., 2003). These social difficulties may reflect the underlying deficits in complex reasoning and abstract thinking. In a study mentioned earlier (Brown & Paul, 2000), two individuals with ACC were given a battery of cognitive and psychosocial tests including the Categories Test, a concept formation task. To do well on this test, a person has to learn a theoretical principle successfully by trial and error. Both individuals had difficulties in recognizing abstract categories. They also performed poorly on Raven’s Colour Progressive Matrices, which is a measure of complex problem solving (Brown & Paul, 2000). Some researchers (e.g., Temple & Ilsley, 1994) also observed impairments in visual memory for complex spatial material such as the mental rotation task (Vandenberg & Kuse, 1978 When presented with two-dimensional pictures of arrays of cubes in different spatial orientations, children with ACC had trouble determining which pictures represented the same set of cubes viewed from different angles (Temple & Ilsley, 1994). Not surprisingly, individuals with ACC also face other challenges in the area of cognition. For instance, these children often experience problems with maintaining attention (e.g., Badaruddin et. al., 2007; Doherty et al., 2006; Moutard et al., 2003; Panos et al., 2001; Schilmoeller & Schilmoeller, 2000). As reported in some studies, they

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have trouble sitting still and are easily distracted and may experience academic difficulties and require additional services. In many of the published case studies, individuals with ACC may have normal intelligence, but often in the low to average range. Despite normal IQ scores, they frequently face academic challenges in subjects such as math and language performance (Finlay et al., 2000; Moutard et al., 2003; Sorensen, 1997; Stickles et al., 2002). For example, with the strong involvement and reinforcement of learning by the parents, the previously mentioned young girl with ACC and normal intelligence was able to achieve slow but steady academic progress over a 10-year period (Sorensen, 1997). Nevertheless, even though she successfully completed elementary school, the academic gap between her and her peers widened during the middle and high school years, particularly in the area of math. Other case studies have documented similar patterns (e.g., Stickles et al., 2002). These academic challenges typically accentuate during the adolescent years, presumably due to the heavy demand of attention and abstract reasoning required by coursework. Yet these challenges may also surface at an earlier age. In one study, 17 children prenatally diagnosed with isolated ACC were followed until 6 years of age (Moutard et al., 2003). Repeated intelligence testing during this period found no intellectual disability (i.e., mental retardation), but did reveal subtle speech delay and reasoning difficulties in some of the children by elementary school age. Overall, the IQ scores of most children in the study remained within the normal range, with a small decrease over time. Individuals with ACC sometimes show what has been called anosognosia—the virtual inability to recognize one’s own limitations. These individuals presume that they can accomplish things that are not within their skill abilities. In some cases, the anosognosia may put the individual in situations of danger to him or herself. For example, a young child assumes he can swim across a fast flowing river when he is incapable of swimming at all (K. Schilmoeller, personal communication, 2009). Some parents also report that their children with ACC engage in confabulation (Schilmoeller & Paul, 2003). These children tell incorrect or untrue stories but believe what they are saying is true. For instance, a child may accurately describe the scenery in a city (e.g., the location and surrounding of a sports stadium) and believe that he has been to the city, while in reality he has never traveled there and most likely has seen the scenery on a television program (K. Schilmoeller, personal communication, 2009). This pattern of telling incorrect information often is construed as lying by educators, parents, friends, or employers of the individuals with ACC and the consequences can be punishment or termination of employment. To date, no formal research has examined these phenomena.


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5. Therapeutic Interventions for Individuals with ACC Few researchers have investigated the types and effectiveness of therapies used with individuals with ACC. Indeed, given that ACC often occurs concurrently with other brain anomalies or other disorders, it is not possible to list specific therapeutic interventions applicable to all individuals. Some data do exist, however, that describe the specific therapies commonly used with these individuals (Schilmoeller & Schilmoeller, 2000, 2001) or track longitudinally the progress of single cases or small groups of individuals with ACC (Panos et al., 2001; Ritter, 1981, 1992; Sorensen, 1997; Stickles et al., 2002). Based on these data, several observations and suggestions regarding treatment choices and issues are relevant. In a survey of 596 families with a child with ACC that contacted an international family support network for ACC, parental reports indicated that 90% of the children received some form of therapy (Schilmoeller & Schilmoeller, 2000). The three most frequently reported therapies were physical therapy (63.6%), speech therapy (59.9%), and occupational therapy (56.3%). In a follow-up study of 678 families (Schilmoeller & Schilmoeller, 2001), parents corroborated these data, reporting that more than six in 10 children received one or more of these same therapies. Other commonly received therapies included early intervention, sensory integration therapy, and some form of academic (e.g., special education) counseling. Several researchers (Ritter, 1981, 1992; Sorensen, 1997; Stickles et al., 2002) followed the longitudinal course of academic and speech–language interventions with individual cases. The three individuals in these reports shared several common characteristics. Each had low average IQ scores on standardized tests. Each experienced language delays, especially in the area of pragmatics. With sustained intervention efforts, each was able to make academic progress (two were near the last years of high school and the third had graduated from high school and was taking college courses). Finally, all of the authors emphasized that therapeutic and intervention decisions were not driven primarily by results from standardized tests. Instead, interventions were required to fit the client’s needs based on specific academic and communicative challenges, each of which evolved over time.

5.1. Common themes Informed by these studies, literature reviews, and personal conversations with parents and professionals, we summarize some common themes for families and professionals to consider.

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5.1.1. Physical delays Physical delays often are prevalent in the early years of life. Many children with ACC show delays in early milestones (rolling over, sitting up unassisted, walking, toilet training) but eventually gain proficiency in these areas. By the school years, many of these children show no significant delays compared with their typically developing peers. In these early years, physical therapy and occupational therapy, especially with attention to sensory integration issues, often are the therapies most indicated. 5.1.2. Nonverbal learning disabilities Some parents of children with ACC have found helpful the information on nonverbal learning disabilities (NLD; Rourke, 1995; Thompson, 1997). Others do not agree that issues for NLD completely match the learning experiences of those with ACC (e.g., Panos et al., 2001). However, even in the event that the match between NLD and ACC is not always congruent, many parents report that a list of suggestions that Rourke includes as an appendix in his book provides a good beginning point in terms of looking at educational issues for children with ACC. 5.1.3. Receptive versus expressive language People with ACC tend to absorb more information than they can demonstrate to others in educational settings that emphasize writing or verbal explanations. For example, in a case study mentioned earlier (Sorensen, 1997), the speech–language therapists working with the girl often reported that she appeared to know more than the formal tests revealed. Thus, teachers should be creative in finding ways for children to demonstrate their knowledge. The following anecdotal report by a parent is a good example in that regard (K. Schilmoeller, personal communication, 1999). A 5-year-old child with ACC was asked to draw a picture of a man. He took a crayon and scribbled across the page. The teacher concluded that the child had no concept of what a person looked like. However, shortly afterward when the child was provided different shapes of paper and pieces of masking tape, without prompting from an adult, that same child produced a face complete with eyes, nose, and mouth, and announced “This is me.” The child clearly understood what a face looked like. He simply could not manipulate a crayon well enough to demonstrate his knowledge. 5.1.4. Delays evident only at later ages Some families report that their children do not show any observable delays initially, commenting that their children with ACC are asymptomatic.


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However, often communicative, attention, and reasoning challenges are diagnosed for these same children when they are in formal educational settings (Moutard et al., 2003). This is especially the case in terms of abstract reasoning (e.g., math—Ritter, 1981, 1992; Sorensen, 1997; Stickles et al., 2002; certain types of more abstract humor such as irony or word play as well as social reasoning—Brown & Paul, 2000). Thus, early and close monitoring by both parents and professionals should be ongoing, even when the child with ACC shows no obvious delays. Perhaps the strongest recommendation regarding the selection of therapeutic interventions is to urge all teachers, therapists, and parents working with individuals with ACC to be diligent observers of the individual’s learning patterns. Rather than reaching for standardized curricula or models of therapy, let the uniqueness of the individual with ACC guide decisions about what works and what does not work.

6. Conclusion and Future Directions The precise impact of a disorder of the corpus callosum on the daily life and learning of a child or adult with the condition may be difficult to define. Yet, as researchers continue to untangle the mysteries of brain structure and function, we are learning more about the corpus callosum and the potential challenges for those living with the disorder. We should note that many of the psychosocial, communication, and cognitive studies on callosal disorders are case studies or studies with small samples drawn from clinical groups often without a comparison group. Even though these studies often documented subtle or significant developmental abnormalities, it has been suggested in some cases that ACC may be asymptomatic (Sauerwein & Lassonde, 1994; Wisniewski & Jeret, 1994), though some argued that more comprehensive studies need to be conducted in order to test this hypothesis (O’Brien, 1994). Given this lack of clarity, we therefore caution readers to take care in generalizing the findings summarized in this chapter when encountering cases of ACC.

6.1. Summary Not too long ago, the corpus callosum was believed to simply connect the two cerebral hemispheres. It was accepted that the effect of congenital absence or malformation of the corpus callosum on a person was the same as someone who had the corpus callosum severed later in life. It is now clear that such a belief is false (Lassonde et al., 2003; Paul et al., 2007). While we have learned a lot more about ACC in the past decades, much of this condition remains unknown.

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First of all, prevalence estimates of ACC vary greatly, ranging from 1.8 to 70 per 10,000 in the overall population. The rates are higher (220 or greater per 10,000) among the MR/DD population. Given that some individuals with ACC are undiagnosed, it is possible that the rates may be even higher. This is particularly relevant for those who have MR/DD, but with no discernable cause. Hopefully, with the increasing use of the imaging technologies and the advancement in the field, the condition may be more likely to be identified early and the question of prevalence may be better answered. Many individuals with ACC exhibit developmental delays and have other concurrent conditions. Consequently, the therapeutic and service needs of each individual change over time and vary from person to person. Depending on the person, the delays or abnormalities may be observed in areas such as behavior, cognition, speech/language, social, or motor development and may involve difficulties with daily living and school. For example, young children with ACC may be delayed in achieving some developmental milestones such as walking, speaking, and toilet training. Compared to their typically developing siblings, they may be more likely to have sleep problems (e.g., bedwetting, frequent nighttime waking) and have more temper tantrums and rapid mood changes. Children with ACC may also be more likely to show stubbornness and be less flexible in dealing with changes in routines. However, these children are generally described as friendly, happy, and enjoying interactions with others. Even though some may display certain autistic-like behaviors such as repetitiveness, the behaviors are of less severity and lower frequency than children with autism. Similarly, some children with ACC may have trouble paying attention or sitting still, but dual diagnosis with ADD or ADHD is not common. As a child with ACC enters elementary school, with proper monitoring and appropriate services (particularly in the areas of speech and language), he or she can usually meet academic expectations. However, some may have problems in pragmatic skills and may have “out of place” or “meaningless” talk. As they progress into middle and high schools, the difficulties may accentuate and so may the gaps between these children and their peers. These gaps can be social and academic. Some adolescents with ACC have impairments in complex and abstract reasoning and may experience difficulty with visual memory and spatial materials. These deficits may make learning subjects like math more challenging. Socially, some are described as unsophisticated and immature for their age and thus may have difficulties in establishing close friendship with same-age peers. There are also other sensory and cognitive abnormalities observed in individuals with ACC, most of which have not been studied extensively. For example, some infants with ACC exhibit delays in vision and auditory maturation. Case studies of these children revealed that they were initially


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diagnosed with severe hearing loss and vision impairment at birth, but developed normal hearing and vision within a few years. Other sensory abnormalities include an unusually high tolerance for pain among some individuals with ACC. The phenomenon of high pain tolerance seems particularly paradoxical in light of high sensitivity to touch among some children with ACC. It also raises concern for the physical safety of these children. In a similar sense, cognitive abnormalities like anosognosia can also cause concern for parents and caregivers of children with ACC. The virtual inability to recognize one’s own limitations can easily put oneself or others in physical danger. The phenomenon of confabulation suggests that individuals with ACC may have difficulties in sorting out the source of information they received. This abnormality may have some serious social and legal implications as individuals with ACC may be convinced that certain events are real when they are not. To outside observers, they may be viewed as being dishonest or untruthful. More research is needed to investigate these phenomena.

6.2. Future directions Regarding the directions for future research, we suggest the following areas. 6.2.1. Effectiveness of therapies and early intervention Given the limited data on the type and effectiveness of therapies for individuals with ACC, more research is needed. Very few studies (e.g.,, Panos et al., 2001) evaluate children with ACC using the model of NLD (Rourke, 1995; Thompson, 1997). Thus, the question remains as to whether the learning experiences of those with ACC match with the issues for NLD. It would be helpful for the parents and practitioners working with children with ACC if more information were available to determine the relationship between ACC and NLD, so that they may be able to utilize the educational resources and intervention strategies developed for those with NLD. The same holds true for other potentially relevant interventions. 6.2.2. Isolated ACC Given that many studies in the literature either use participants with comorbid conditions or are based on small samples with no comparison groups, it is difficult to tease out the unique effect of only missing the corpus callosum on brain function and development. Thus, more research with a focus on isolated ACC is needed to fully understand the impact of the disorder. Particularly with the advancement and increasing availability in imaging technology, more cases of callosal disorders are being diagnosed prenatally, some of which are isolated ACC. It is now possible to closely

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monitor these children and assess their development beginning in the early stages of their lives and, ideally, through adulthood. 6.2.3. Adult living As children with ACC develop into adulthood, other issues such as relationships, money management, and personal safety start to take center stage. Yet for many of these adults and their families, adult living is uncharted territory. Given that some individuals with ACC may be socially na¨ıve and unsophisticated in self-perception, and have problems with abstract reasoning, ansognosia, and confabulation (e.g., Brown & Paul, 2000; Ritter, 1992; Schilmoeller & Paul, 2003; Sorensen, 1997; Stickles et al., 2002), independent adult living may sometimes be hazardous. Families of some individuals with ACC face choices of whether to obtain legal guardianship or conservatorship of finances, and individuals with ACC may need support for obtaining and sustaining employment. Adults with ACC may face more challenges in forming intimate relationships, marrying, and being parents than do their peers. Studies are needed to examine how adults with ACC currently manage their daily living and how their experiences can serve as a roadmap for others in the future. There really is no single “best” way to support and/or educate individuals with ACC. For young children with the diagnosis, our recommendation to parents, educators, and service providers is to closely monitor the child, obtain appropriate evaluations, and start intervention services early. While some children with ACC exhibit impairments in certain areas at an early age, others may appear to develop typically until they reach school age. In addition to physical, occupational, speech and language therapies, social skills training may be beneficial in helping the child navigate the increasing challenges of school. The key is to monitor the child closely, even if he or she seems to initially be doing well, and to identify the problems early before the child falls too far behind academically or behavioral issues emerge.

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C H A P T E R E I G H T

Autism Intervention Research: From the Reviews to Implications for Practice Stephanny F. N. Freeman,* Tanya Paparella,* and Kelly Stickles† Contents 1. Introduction 2. Synthesizing the Reviews of Autism Treatments 2.1. Introduction 2.2. Comprehensive treatments 2.3. Targeted treatments 2.4. Summary 2.5. Nonresponders: one size does not fit all 2.6. Conclusions: research to practice/practice to research 3. UCLA’s Early Childhood Partial Hospitalization Program: A Pilot Study 3.1. Introduction 3.2. Pilot study: method 3.3. Pilot study: results 3.4. Pilot study: discussion 4. Integrated Summary Acknowledgments References

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Abstract Early intervention for children with autism is currently a focus in both the research literature, popular literature, and in practice. A number of reviews exist that examine studies of comprehensive interventions and targeted interventions, including those that examine environmental variables, participant and interventionist variables, methods of instruction, and nonresponders. All seek an understanding of the best interventions to ameliorate symptoms, identify treatment by individual characteristics, improve the quality of life, and demonstrate positive short- and long-term outcomes for young children with autism. * †

Department of Child Psychiatry, UCLA Graduate School of Education & Information Studies, UCLA



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There is little question that early intervention is beneficial, particularly for improving functional skills and developmental skills, improving core-deficits such as play, and reducing maladaptive behaviors and symptoms. General evidenced-based guidelines for treatment have been formulated (National Research Council, 2001) but few well-designed (randomized control) studies exist. Indeed, methodologically the studies are incomparable, the variability in the population and the samples are overwhelming, and the results are complex and difficult to interpret from both a research and practical standpoint. This chapter aims to help practitioners better understand how to use evidencedbased practice by synthesizing literature reviews on early intervention, presenting an example of an early intervention program that uses an evidenced-based yet clinically eclectic approach to treatment, and discussing implications for further intervention studies and treatment programs.

1. Introduction In recent years, the explosion of children diagnosed with autism has fueled an increased public focus. In turn, the need for research on effective treatment in the area of early intervention became an immediate concern. Early intervention is integral to understanding the manifestations of the disorder and maximizing change as early as possible. Despite the critical demand for effective treatment, a growing list of extremely variable intervention options, and an extensive intervention literature over recent years, many scholars claim that few well-designed and sound intervention studies exist for parents and practitioners to make conclusions about treatment. Why are conclusions about treatment difficult to make? First, autism is an extremely heterogeneous disorder with enormous variability in characteristic expression in different children, yet intervention studies have mostly approached the samples as one homogenous group. In extant traditional experimental designs, the outcomes are identified by group differences and it is unclear how the participants may have responded differentially to treatment based upon individual or treatment-by-individual characteristics. We cannot say with confidence that a certain type of intervention should be used for a child or a group of children with autism, a concept that is at the core of what we consider “evidence-based practice.” Second, from study to study the methodology is extremely variable, the results are mixed, comparisons are impossible, and little attention is given to the nonresponders. This state of affairs presents a serious dilemma for practitioners in that treatments do not work for every child, yet practitioners are expected to effectively treat every child. Many practitioners desperately seek a comprehensive “how to” manual on treatment of children with autism, even as the literature indicates that studying treatment effectiveness from a “one size fits all” approach is not effective. The result in

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practice is confusion as to what the literature concludes, clinical decisionmaking (with the hopes that the clinician is knowledgeable) on treatment choices, the development and use of interventions without evidence, and an array of choices that boggle the minds of parents and practitioners alike. Where, then, does autism intervention research stand? This chapter will synthesize reviews of autism research over the past 5 years, with a goal to better understand what can lead to better treatment for children. Reviews of both comprehensive and targeted treatments will be examined (e.g., play, social skills, self-management techniques). The chapter will summarize implications for treatment and limitations related to treatments within each content area. Sadly, given the multitude of variables involved in effective intervention in a heterogeneous population, ultimately, the reader will come away struggling with a complex picture and little clarity of how to implement evidence-based practice for children on the autism spectrum. It is clear from the research that no one approach works for all children and children respond differentially to different treatment approaches. Thus, the chapter goes on to offer a different way of approaching treatment research—that is, to focus on the intervention process rather than the approach. Thus, the last section of the chapter describes the Early Childhood Partial Hospitalization Program, a model treatment program for young children on the autism spectrum. It is unique in that we implement a multiapproach, dynamic, and evaluative delivery model for each child resulting in optimum outcomes.

2. Synthesizing the Reviews of Autism Treatments 2.1. Introduction The following section provides an overview of autism intervention research reviews to date. We begin with reviews of comprehensive treatments. Comprehensive treatments aim to intervene in all areas of a child’s development. Despite different treatment philosophies, comprehensive programs do not appear to differ significantly from one an other in terms of intervention effectiveness. What makes comprehensive treatments similar is that they share a set of common elements that seem to contribute to the success of programs overall. However, many of the studies that implement and investigate the effects of comprehensive treatments have been criticized for methodological flaws. Also, the variability in research design, hypotheses, target populations, and outcomes make it difficult to use comprehensive treatment findings as a basis for making decisions regarding effective, evidence-based treatment. We therefore extend this review to also examine targeted treatments that focus on particular aspects of

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individual functioning or on aspects of the environment. Targeted treatment research, while limited in scope of intervention, aims to identify the specific variables that contribute to significant change in core areas and critical change in more comprehensive areas for children with autism.

2.2. Comprehensive treatments Comprehensive treatment programs seek to improve the overall functioning of individuals, thereby changing the nature and course of their outcome. They have been defined as “treatments that address core deficits in autism including language, social, cognition, and play” and “analyses of child progress using general measures of children’s language or intellectual development as aspects of the outcome measure” (Rogers & Vismara, 2008). Comprehensive treatments typically report on both the content (“what”) of the treatment as well as the treatment approach (“how”). For example, Lovaas’ (1987) behavioral approach described both the skills targeted for change in each developmental area as well as how discrete trial instruction was delivered. Ten comprehensive programs were described in 2001 (National Research Council, 2001). The majority of programs were derived from a behavioral orientation, two programs ascribed to a developmental treatment approach, and one program (TEACCH) was described as eclectic, with characteristics of both behavioral and developmental orientations. Common elements among the early intervention models included targeted curriculum content, individualized interventions, a highly supportive teaching environment, a focus on generalization, strategies to increase predictability, a functional approach to maladaptive behaviors, planned transitions, and active family participation (Dawson & Osterling, 1997). Additional similarities included highly trained staff and close supervision (Anderson & Romanczyk, 1999). Nearly all of these programs reported their outcomes descriptively in the form of parent or trainee satisfaction data (Mesibov, 1997), measures of children’s progress in comparisons before and after intervention (e.g., McGee, Daly, & Jacobs, 1994, 2000) and case reports (e.g., Koegel, Koegel, Shoshan, & McNerney, 1999; Strain & Hoyson, 2000). Virtually no data existed to compare the relative merit of one model over another. Up until fairly recently, randomized controlled treatments for children with autism had not been conducted. However, in a recent review of evidenced-based comprehensive treatments, Rogers and Vismara (2008) identified five randomized controlled design studies. They also reported on several other nonrandomized controlled design studies of comprehensive treatments with varying treatment approaches (e.g., discrete trial instruction, developmentally based intervention, and a mixed methods approach based upon clinical judgment). Overall, the comprehensive treatments presented positive and remarkably similar findings. In sum, it appears that a majority of children participating in comprehensive interventions make


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significant progress in multiple developmental domains (Hoyson, Jamieson, & Strain, 1984; Koegel et al., 1999; Rogers & DiLalla, 1991; Rogers & Lewis, 1988, 1989; Rogers and Vismara, 2008; Strain & Hoyson, 2000; Schafer & Moersch, 1981). Support for comprehensive treatments, particularly those with a behavioral theoretical background, was emphasized by two other recent reviews. The first meta-analysis identified both large and moderate effect sizes of comprehensive Early Intensive Behavioral Interventions calling it the “intervention of choice for children with autism” (Eldevik et al., 2009). Eikeseth (2009) focused on outcomes of comprehensive programs and found strong support for Applied Behavioral Analysis by evaluating the magnitude of treatment effects. Despite the promising increase and the positive findings in published comprehensive treatments in the last 5 years, it is still problematic to use comprehensive treatments as a basis for making conclusive decisions regarding effective evidenced-based treatment. First, comprehensive intervention models improve particular core deficits (e.g., language, social skills) of autism but have not been able to demonstrate a complete remediation of autism symptoms. Second, randomized control trials (RCTs) designs are not designed to provide answers regarding all aspects of treatment. Thus, outcomes regarding fundamental characteristics may be reported [e.g., language outcomes, intelligence quotient (IQ)], but the multifaceted treatment needed to achieve change in these outcomes is not captured with enough specificity to provide informed clinical information to replicate the outcomes [e.g., specific treatment approaches in individual speech and language therapy, small group pragmatics, larger group pragmatics, targeted social skills development, motivational techniques, direct instruction (DI)]. Third, in addition to small sample sizes, RCT studies vary significantly in many aspects of research design. For example, studies vary widely in their treatment intensity, in length of treatment, and in measurement of outcomes. Ultimately, the fundamental differences in study approach do not allow for systematic comparison of one methodology against another. Thus, general overall improvement is achieved, but by their nature, comprehensive treatments do not identify the critical variables targeted for change in outcome. As a result, some intervention researchers have turned to targeted treatments to identify the specific variables that contribute to significant change in core areas for children with autism.

2.3. Targeted treatments Targeted treatments involve any treatment approach that has a specific method for implementation with a narrow outcome (e.g., developing symbolic play). Studies of targeted interventions have a number of significant advantages. The results can greatly contribute to a more refined

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understanding of salient intervention content/curriculum. Second, the content can be implemented with tighter fidelity and standardization. Third, experimentally extraneous variables (such as time, environment, people involved) can be reasonably controlled. While their strength lies in identifying specific variables for change, targeted treatments do not treat many symptoms of autism. For example, a play intervention may contribute significant treatment information in play, but will not directly address the child’s delays in preacademics or maladaptive behaviors. Targeted treatment research often has methodological flaws, specifically with experimental control. Experimental control is not systematically exercised in treatment implementation and the admission of participants. Thus, while they play an informative role in intervention research, targeted treatments, by nature, contribute only partially toward an understanding of what constitutes effective treatment for a child with autism. Several types of targeted treatments are depicted in Table 8.1 and described below. First, we describe domain-specific review studies that focus on targeted interventions within a specific developmental domain (e.g., social skills). Reviews of method-specific interventions are next described. These reviews focus on a specific method of intervention to achieve an outcome, for example, the use of video modeling. Lastly, we conclude with a short description on reviews of environment-specific and interventionist-specific treatment designs. 2.3.1. Domain specific (Table 8.1) Extant literature reviews focus on intervention studies treating both core-deficit areas and global domains (areas of difficulty found in autism but not defining of the disorder, e.g., academic skills; Chiang & Lin, 2007). In the following section, we describe intervention review studies in the domains of play, social skills, self-management, challenging behaviors, and communication. These targeted treatments are directed toward specific developmental domains or symptoms. The studies within the reviews have varied methodologies and can consist of, for example, single-subject multiple baseline designs (e.g., Whalen & Schreibman, 2003), small sample treatment designs (Koegel et al., 1999; Strain & Hoyson, 2000), and in rare cases, randomized control group designs (Kasari, Freeman, & Paparella, 2006). 2.3.1.1. Social and play skills Targeted treatments of social and play skills (Table 8.1) in children with autism are significantly on the rise (Barton & Wolery, 2008; Matson, Matson, & Rivet, 2007; White, Keonig, & Scahill, 2007). Social skills can be defined in behavioral terms as, “interpersonal responses with specific operational definitions that allow the child to adapt to the environment

Table 8.1 Domain specific content

Authors and review characteristics Symbolic play

Social skills

Issues

Conclusions

Barton and Wolery (2008)

Generalization difficult • variability of treatment • inconsistent definitions of pretend play

With targeted treatment (specifically techniques of adult modeling and prompting), symbolic play improves

16 studies focused all on teaching pretence

Focus of reviewed literature was on improvements in pretense behaviors rather than overall play levels or global engagement

Specific teaching strategies lacking

Matson, Matson and Rivet (2007)

Multiple intervention approaches: peer modeling, peer mediated, social stories, reinforcement schedules, and uncategorized

Modeling and reinforcement treatments were most popular models

79 studies focused on eye contact, appropriate content of speech, speech intonation, appropriate facial affect, initiations

Methodological issues: haphazard selection of target variables, lack of social validity, and lack of group research designs

Operant or Social Learning Theory basis

Concerns: lack of rigorous diagnoses of the participants and cross-content investigations were nonexistent (Continued)

Table 8.1 (Continued )

Authors and review characteristics White et al. (2007)

14 studies focused on direct group social skill training interventions

Selfmanagement

Lee, Simpson, and Shogren (2007) 11 single subject design studies targets within social (sharing, nonverbal behavior, eye contact), adaptive (schedules and independent living)

Issues

Conclusions

Extreme variability in methodology, outcome measurement (Social Skills Rating System most frequently used parent-report measure), identification of sample, techniques used, and in manual development Missing: sound theoretical framework and appropriate measurements for the construct of social skills

Reviewers concluded: several promising interventions were identified

The philosophies of the described interventions ranged from behavior (using token systems to reward behaviors), to direct instruction approaches, to social cognitive/development (using photos and visuals, discussion of target skills)

All treatments effective but not for all children

Behavior

Horner, Carr, Strain, Todd, and Reed (2002)

Intervention effectiveness was not mediated by a diagnosis of autism

41 studies reviewed eight procedures: stimulus-based procedures, direct instruction, extinction, punishment, reinforcement, systems change, pharmacology, and unclear procedures

Functional analysis of behavior was most related to success Generalization remains one of the biggest challenges

Conroy, Dunlap, Clarke and Alter (2005) 23 studies examining only positive behavioral interventions (PECS, functional analysis, antecedent/ consequence, and selfmanagement)

All but one design used a singlesubject approach

A trend toward more stimulusbased and instruction-based interventions occurred over time (less reinforcement/ punishment) Systems change concept seemed to be developing as a more targeted methodology High efficacy — 80% behaviors were reduced

Instruction- and stimulus-based methodologies used the most Frequent use of examination of systems surrounding behaviors 40% indicated a direct link between functional analysis outcomes and interventions implemented (Continued)

Table 8.1 (Continued )

Authors and review characteristics

Communication

Machalicek, O’Reilly, Beretvas, Sigafoos, and Lancioni, (2007) 26 studies with a school focus. Intervention procedures = manipulating antecedents, changing the instructional context, differential reinforcement, and selfmanagement techniques Goldstein (2002) 60 studies characterized by methodology; sign language, discrete trial, milieu based, language development to decrease problem behaviors, social and scripted language interventions, and parent/ classroom approaches Ogletree, Oren, and Fischer (2008) Synthesized Discrete Trial Training, CABA, and DSP

Issues

Conclusions

Studies encountered: • high environmental variability • high behavioral variability • frequent use of other treatments

No cohesive guidance for successful behavioral intervention in the school setting

Different theoretical frameworks Heterogeneous samples Quasi-experimental No control groups

Equivocal There was no particular treatment that facilitated language growth over another (direct comparisons were not made)

Strong description of several characteristics of effective communication interventions. These include prompting, modeling, time delay, reinforcement, routines, and visual supports

One approach not more effective than another


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through verbal and nonverbal communication.” Examples of play and social skills that were targeted for intervention included pretense abilities, eye contact, pragmatic speech, appropriate facial affect, and initiations toward others. Missing from most studies was, first, a sound theoretical framework established as beneficial for children with autism. An alarming symptom of a lack of underlying philosophy was a seemingly haphazard selection of target variables. A second concern was a lack of appropriate measurements for the construct of social skills. Third, generalization was not adequately addressed. Therefore, not only were the majority of the studies fraught with methodological issues (e.g., the lack of group research designs), but they also lacked social validity. Nonetheless, across reviews, several promising interventions were identified as effective in improving the social interactions of children with autism both with adults and peers: pivotal response treatment (PRT), adult prompting, environmental modifications, social skills groups, social stories, video modeling, and peermediated instruction (Hwang & Hughes, 2000; Rogers, 2000). 2.3.1.2. Self-management Although not defined as a “core deficit,” a number of studies have focused on self-management techniques and found effective results for a portion of the participants. Self-management can be viewed as a skill that embodies particular deficits in social skills, joint attention skills, and play/interaction—as well as independence. Interventions in self-management skills contribute to the likelihood of successful integration (i.e., it is generalizable and transportable), to positive outcomes, and to the facilitation of change in multiple domains of development for some children. 2.3.1.3. Challenging behaviors The prevalence of challenging behaviors in children with autism is well documented (Emerson, 1995; Research Units on Pediatric Psychopharmacology Autism Network, 2002). Although maladaptive behavior is not a problem specific to children with autism, it is clearly an area that is descriptive of the disorder (albeit not exclusively), extremely disruptive to effective education and social development (Horner, Diemer, & Brazeau, 1992), and puts children at risk for exclusion and isolation (National Research Council, 2001). Thus, there is a significant need by all educators, therapists, professionals, and parents for effective treatments to target maladaptive behaviors. Three recent reviews (Conroy et al., 2005; Horner et al., 2002; Machalicek et al., 2007) were conducted that address challenging behavior in children with autism. The philosophical approach underlying most behavioral intervention programs was behavioral theory. Within behavioral theory, the most accepted and successful approach was to use a functional

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assessment and analysis of behavior to drive and monitor behavior change (in both positive and punishment-based interventions). However, only 40% of the positive behavior interventions were directly linked to use of a functional analysis of behavior. Overall conclusions from the reviews suggested trends toward a more thorough examination of the systems surrounding disruptive behaviors, stimulus-based and positive instructionbased interventions, more programmed intervention protocols and less clinical judgment, and less simple reinforcement/punishment procedures. Again, generalization remains one of the biggest challenges. 2.3.1.4. Communication Like maladaptive behavior, communication deficits are not solely descriptive of autism, although the disorder is characterized by social communication deficits and in many cases, significant delays in functional language. The intervention work on communication skills is particularly difficult to navigate because first, the theoretical frameworks differ widely. Second, underlying philosophies vary from a communication/language disorder intervention approach, to behavioral approaches like discrete trial teaching, and developmental approaches (e.g., sign language, milieu-based instruction, social and scripted language interventions, the picture exchange communication system). Third, many studies are not specific to autism. Although all participants in reviewed studies have a communication disorder, children with a variety of diagnoses are included. Thus the samples are heterogeneous and provide little autism specific directions for treatment. In sum, no particular treatment seems to facilitate language growth over another. Direct comparisons were not made from one treatment to another; thus, conclusions were impossible. Further, it was impossible to identify which approach might work better for certain types of children (e.g., children in need of complex language systems, children with rudimentary language skills, and nonverbal children). Indeed, the literature provides little direction in terms of service delivery models or the intensity of services that are more likely to maximize communication intervention efforts. What seems to emerge are several characteristics of effective communication interventions. These include prompting, modeling, time delay, reinforcement, routines, and visual supports. With regard to methodology in the communication research, many of the studies are quasi-experimental. Thus, although overall positive change was reported, the findings remain significantly limited, without control group comparisons to demonstrate true treatment effects. Thus, despite the generally positive conclusions, communication research to date is lacking in rigorous evidence-based study designs that demonstrate ecological validity.


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2.3.2. Method specific Unlike the previous section, where the focus of the treatment was on a specific area of development (e.g., play), method-specific interventions examine a particular method or treatment approach to achieve an outcome. We review two interventions that focus on the method to achieve change, namely, alternative communication and video modeling. 2.3.2.1. Augmentative and alternative communication Despite the popular argument that using augmentative and alternative communication (AAC) inhibits the growth and development of oral language, the research is fairly consistent in disputing this claim. In a metaanalysis of 23 studies between 1975 and 2003, Millar, Light, and Schlosser (2006) concluded that a significantly high percentage (94%) of participants experienced a positive impact on their speech production with the use of AAC. Not surprisingly, this body of treatment research had drawbacks. First, few (26%) established rigorous experimental control such that one could make confident conclusions that AAC techniques were related to speech production. Second, methodological issues were commonly identified: insufficient diagnostic criteria for all disorders, sample sizes that represented a wide range of age and ability level, and only 31% of the participants in the studies had a diagnosis of autism. Third, although all interventions described used AAC, they nevertheless used a wide range of treatment techniques and varied considerably by treatment intensity. Finally, a number of studies did not report data on treatment integrity. 2.3.2.2. Video modeling Video modeling is a specific teaching technique that has been used to cover a variety of content domains. It involves watching modeled behaviors (the desired intervention outcomes) on television or a computer program. Interventionists have used Bandura’s Social Learning Theory (1977) and capitalized on media to provide opportunities for children with autism to learn from the modeled behavior of others in a controlled manner. For example, several recent studies have examined video modeling as a technique to teach play (Hine & Wolery, 2006; Paterson & Arco, 2007), social development (Parsons, 2006), to increase positive behaviors (Banda, Matuszny, & Turkan, 2007), to teach functional skills (Murzynski & Bourret, 2007), and to model language. Unlike the comprehensive programs (one methodology used to treat a variety of domains), video modeling is considered a targeted intervention in that none of those carrying out the intervention feel that it is a methodology to be used comprehensively. There seems to be consensus that video modeling can be used to teach social communication and social skills (e.g., perspective taking) and to increase appropriate functional skills and behaviors (Ayres & Langone,

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2005; Bellini & Akullian, 2007; Delano, 2007). A variety of methodologies were used such as positive self review, video feed forward with and without hidden supports, and the editing of target content. Different video models can also be used (peers, siblings, adults, etc). Further, skills maintain and video modeling seems to generalize well in that it is transportable (to homes, school, and community) and is user-friendly. Key points that likely contribute to video modeling effectiveness include the following: video modeling integrates visually cued instruction with modeling; attention is maximized by removal of other nonimportant stimuli that are not relevant to the goal; a reduction of the anxiety and stress associated with real-life situations for many children with autism; and a child’s motivation is increased as many enjoy video media (Ayres & Langone, 2005; Bellini & Akullian, 2007; Delano, 2007). Unfortunately, all the studies did not identify the critical features of video modeling that strongly contributed to improvements and efficient use of the techniques. Was it that the video modeling broke down the task into manageable and usable learning segments? Was it that the video provided a nonthreatening situation that facilitated learning for individuals with autism? All studies struggled with controlling external variables, other confounding interventions and variability in subject characteristics. Further, social validity and intervention fidelity were not measured and the studies varied methodologically. 2.3.3. Environment specific In addition to domain- and method-specific targeted interventions, another factor that contributes to intervention efficacy is environment, that is, the variability in environments where intervention can take place (e.g., center based, home based, school based). No studies have systematically analyzed one environment compared to another nor has any study isolated one environment as a significant independent variable related to treatment effectiveness (Harris, Handleman, & Jennett, 2005). One review consolidated the literature that examined environment of intervention but unlike the other reviews described in this chapter, the findings were not promising. Social skill interventions conducted in school settings (Bellini, Peters, Benner, & Hopf, 2007) were found to be minimally effective for treatment effect and generalization, regardless of participants, materials, and even if the skill was broken down into smaller targets. Some trends were identified: older subjects gained more from social-skill interventions, individual interventions were more successful than group-based interventions, but no significant differences were found regarding intensity (but average intensity in the studies was significantly lower than what Gresham (2002) recommended = 30 h over 10–12 weeks). Further, the designs posed a unique set of problems


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in that “pull out” programs may have hindered results, treatment intensity is compromised, and fidelity and intervention specificity were weak. 2.3.4. Interventionist specific Further confounding the ability to make informed conclusions is whether effectiveness and outcomes are affected by who delivers the intervention. Although studies have examined the effectiveness of teaching parents a particular intervention technique (Rocha, Schreibman, & Stahmer, 2007), and generalizing therapist-based training to parents (Schreibman, 1988; Smith, Groen, & Wynn, 2000), to date, there are no reviews that consolidate these interventions. Thus, the question remains ambiguous whether therapists or parents are best to deliver treatments. McConachie and Diggle (2007) did, however, review all parentimplemented/control sample interventions for children with autism. These studies are very recent, as a parent-implemented intervention methodology is a relatively new focus of treatment research. Reviewers identified that only four of the 12 studies were fully randomized controlled trials, while others had methodological uncertainties or weaknesses. Child benefits were not as strong with parent training, whereas therapist taught interventions improved IQ, behavior, and language. Parents reported significant direct benefits such as decreased stress, improved ability to identify their child’s communicative attempts, and increased knowledge about the disorder and teaching techniques. Interaction benefits included increased synchrony between parent and child and improved parent–child interactions in unstructured periods (e.g., play and disruptive behaviors). In sum, then, it seems that increasing parent participation has clear benefits for parents, but we still do not know if parents implementing interventions can be better than or at least equal to trained therapists in achieving positive child outcomes.

2.4. Summary A practitioner seeking “evidenced-based techniques” can become easily overwhelmed by the trends identified thus far. At first glance, it appears that many studies have identified positive results, yet the variety of philosophies, methodologies, techniques, and outcomes make conclusions difficult. Further, reports of poor generalizability and poor ecological validity were significant limitations prevalent throughout the reviews. This is an enormous limitation, as autism intervention is only as good as a child’s ability to effectively use it in his/her everyday life. The question remains: How do practitioners intervene? The answer: One must take the best elements of each study and consider how treatment

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specifics work for certain children. Begin with a comprehensive intervention and integrate targeted interventions considering individual profiles and characteristics. Recall that the comprehensive intervention reviewers that reported positive outcomes embraced a set of common elements that played a considerable role in program effectiveness for most children. A team of the most prominent researchers, practitioners, and physicians in the field came together to disseminate those important characteristics of comprehensive interventions as written guidelines to determine appropriate intervention services: intervention should be provided at the earliest possible age; intervention must be intensive; parent training and support should be a component of the program; the curriculum should focus on the social and communication domains; instruction should be systematic with individualized goals and objectives; and particular emphasis should be put on teaching for generalization (National Research Council, 2001). These characteristics ensure both depth and breadth of intervention and ultimately provide a structural framework to ensure a quality program. These guidelines were a landmark attempt to provide some protocol to describe a quality program. However, it remains a significant challenge to identify a framework that informs success for all children (Goldstein, 2002). Solely relying on comprehensive research is problematic because one then relies on one main treatment philosophy (e.g., behavioral, developmental) to address multiple areas of development. Although comprehensive treatments offer a “one stop” answer for families, they may not actively pursue or use a variety of cutting edge treatments to affect critical change in targeted ways on discrete domains of development. Comprehensive programs do achieve positive outcomes, but as mentioned, not for all children and not in all domains. Thus, treatment could be much more informed if they considered to a much larger extent critical variables that relate to better outcomes in different areas of development, environment, and individual characteristics. For example, within a primarily behavioral comprehensive program, perhaps a particular approach (e.g., milieu-based intervention) may be beneficial for developing symbolic play skills, while a parentdriven intervention may be more effective in ensuring generalized self-management strategies in a child. In contrast, targeted treatments do seek cutting edge knowledge to identify precise variables for change. Thus, they embody a dynamic approach to informing the field. The weakness of considering targeted treatments alone is a limited contribution to the overarching complexity of what is needed to change the pathway of development in children with autism. The answer is to use an “informed eclectic” approach that is grounded in a comprehensive philosophy for overall development and remediation of symptoms, but also uses targeted interventions for discrete domains of


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development. Consider the literature findings described above to clinically determine what approach is best for certain domains, individual children, the setting, and the interventionist. Unfortunately, however, just when one thinks that the answer is now clear, clinical practice muddies the water. Even if one believes that the treatment choice made is most effective (positive comprehensive and targeted outcomes for participants) some children do not respond to the initial chosen protocol.

2.5. Nonresponders: one size does not fit all Beyond our consideration of the types of treatments available, when one considers the big picture of autism intervention there is a further element that undeniably requires attention: those children who do not respond to a particular intervention. In every treatment approach and in every study reviewed, there are children that simply do not meaningfully respond, that is, who do not show clinically or statistically significant gains after treatment. Throughout this chapter, all of the reviews specified the response rate of the participants within each intervention. No intervention is effective for all children. Although the investigation of nonresponders is somewhat recent, it is not new to individualize education and match educational and therapeutic resources to specific needs, especially in the early intervention years (Gabriels, Hill, Pierce, Rogers, & Wehner, 2001; Prizant & Wetherby, 1998). The key is to merge these two bodies of literature to identify why some children respond while others do not. While as yet no reviews exist, studies have identified pretreatment characteristics that relate to successful outcome in individual intervention studies. These include child IQ, language skills, visual–spatial skills, social relatedness (joint attention, play, eye contact), behavior, and parent–family factors (Harris & Handleman, 2000; Ozonoff & Cathcart, 1998; Rogers, 1998). Further, characteristics of those children who respond well to particular interventions have been identified (e.g., Rogers et al., 2006). Thus higher response rates to a particular intervention might be characterized by better joint attention, better imitation skills, and lower scores on diagnostic measures. Only one study has exclusively examined the characteristics of children who do not respond to intervention (Sherer & Schreibman, 2005). The authors developed a predictive profile to identify nonresponders. This profile included children with low rates of toy play, verbal self-stimulatory behaviors, low approach behaviors, modest rates of nonverbal self-stimulatory behaviors, and avoidance behaviors. The nonresponders were not additionally treated after insignificant intervention findings. In theory, practitioners could make evidence-based clinical decisions to identify particular interventions that might fit particular children. Practical

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reasons, however, often make implementation difficult. For example, clinicians may not be able to alter or change an intervention for a particular child because of program philosophy, group versus individual interventions, a lack of clinician expertise, family dynamics, and difficulty coordinating with other therapies (Ozonoff & Cathcart, 1998). One must believe that certain intervention types or approaches can work even if another does not as we can never give up on intervention for children with autism. It is important to know who responds well to a given intervention, what the characteristics of the participant are, who does not respond, what characteristics define the nonresponder, and to what intervention they did eventually respond. This research path can inform what treatment types are the most beneficial to try initially for which children—an endeavor that is advantageous in improving child outcomes, and is economically beneficial as well.

2.6. Conclusions: research to practice/practice to research 2.6.1. Research to practice We revise the answer to the question of how to effectively treat children with autism (given nonresponders): Use an “informed eclectic” approach that is grounded in a comprehensive philosophy for overall development and remediation of symptoms but that also uses targeted interventions for discrete domains of development. Consider the literature findings described above to clinically determine which approach is best for certain domains, individual children, the setting, and the interventionist. Systematically evaluate the progress of the participants and, after a set protocol of time, if a portion of the sample is nonresponsive, change intervention and measure again. Continue this procedure until all have achieved relative but significant progress. Indeed, comprehensive and targeted treatments have unique strengths and limitations, with the result that neither model can stand on its own to guide best practice—use the best components of both. At the core of the complexity is that autism is a developmental disorder requiring intervention across multiple core areas of development. Further, even with our limited knowledge on intervention effectiveness, it is clear that early intervention can significantly impact a child’s developmental outcome. Yet autism is not a homogenous disorder. Children present with a wide spectrum of symptoms, which creates a complex challenge for determining what constitutes effective treatment—make treatment flexible, as data and clinical observations indicate. 2.6.2. Practice to research Treatment research in this field has increased enormously in recent years. However, it is still struggling to deliver well-designed studies that show


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meaningful effectiveness. Contributing to the lack of clarity is that intervention research has mostly approached autism as a homogenous disorder, and has not adequately taken into account that children within the autism spectrum respond differentially to treatment in both the laboratory setting and the clinical setting. Even more confusing, children respond differentially even within different intervention targets. For example, it is not enough to describe a child as responding to behavioral intervention. It may be that in the domain of language development that the same child responds much better to a different type of intervention. Further, what may be effective for one child may not be effective for another child. Given this multilayered puzzle that occurs in clinical practice, how then, does one begin to think about research? How do we begin to determine what works for a particular child, how do we measure change, and how do we report it within the larger picture of autism as a whole? How can we come to conclusions regarding what constitutes effective treatment? We propose rethinking how effective intervention is measured. Perhaps it is not the intervention approaches that should be further examined, but the model and the process in which the intervention is carried out and how service providers work towards mastery for all children. More specifically, it could be that the elements of an intervention program matter more. Perhaps a focus of intervention should shift to how intervention is implemented and the critical factors involved in ensuring that all children make progress. This method allows for incorporating the multilayered complexity that is necessary for treating children on the autism spectrum. First, it allows for using both a comprehensive approach and targeted treatments simultaneously, thus maximizing the strengths of both. Second, it provides flexibility to treat children with a range of symptom presentation, that is, the heterogeneous nature of the disorder. Third, it builds in flexibility to try different approaches for nonresponders, discarding the notion of a “one size fits all” approach. As an introduction to this method we describe the Early Childhood Partial Hospitalization Program (ECPHP) as a comprehensive program that achieves significant outcomes (gains beyond what would be considered typical developmental progress based on maturity) for all children. It is considered a comprehensive program, yet it incorporates a variety of targeted treatments depending on the individual characteristics of each child. The ECPHP embraces a dynamic approach whereby a variety of treatments are evaluated to determine effectiveness for each child. It aims for a significant response rate in all children and achieves this by modifying intervention approaches depending on the child’s response to treatment. Thus, the ECPHP embraces the heterogeneous nature of autism. The program is unique in its treatment flexibility and strong outcomes. In addition to describing the program’s unique model and treatment process, an initial set of one component (skills progress) of ECPHP outcome data is

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presented that demonstrates significant change in preacademic skills in children pretreatment/posttreatment—data that have ensured success by all children in the program.

3. UCLA’s Early Childhood Partial Hospitalization Program: A Pilot Study 3.1. Introduction 3.1.1. Program description 3.1.1.1. Environment The ECPHP is a day treatment program for children with autism and behavioral disorders. Currently, ECPHP enrolls a maximum of 12 children at any one time. The core of the program centers around three preschool classrooms. Classroom 1 is for 2-year-olds of all ability levels. Classroom 2 is intended for children who are 3 years and older, and require one-to-one supervision. Classroom 3 also serves children 3 years and older and has a one-to-two teacher to client ratio. Children in this class are more capable of participating in group activities with inconsistent one-to-one supervision. Each child is placed in one of the three classrooms based on their initial assessment, but changes can be made during the program if necessary. Children who attend the program receive intervention 5 days a week, 6 h per day in a structured environment for a maximum of 12 weeks. The environment of the classroom simulates a typical preschool classroom but with less stimulation (less artwork and decoration) and with designated individual or small group workstations. 3.1.1.2. Staff The codirectors have Ph.D.’s and are licensed psychologists with extensive experience treating children on the autism spectrum. They supervise all the staff, the administrative duties, the curriculum and programs, and oversee the children’s treatment and the family participation. Each child is assigned to one of the codirectors; they conduct assessments, develop and review treatment plans, communicate with all the therapists, continually evaluate the child’s progress, and communicate with the families and community providers. A licensed clinical social worker is provided for each family to provide support, parent training, to assist the family in obtaining community resources and in planning for discharge. Specialists on staff include a speech pathologist, an occupational therapist, and a recreational therapist. Specialists provide individual pull-out services as needed, provide in-class intervention, monitor progress and update developmental goals and techniques, and


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supervise the behavioral therapists as they implement their learning objectives. The registered nurse oversees all aspects of treatment concerning health (e.g., allergies, diets) and the development of self-help skills. The medical director (psychiatrist) implements medication management (if appropriate), psychiatric interviews, medical evaluations, and conducts insurance reviews as necessary. The trained behavior therapists are present consistently and implement the classroom instruction under the supervision of the codirectors and specialists. All staff receive regular in-service training and hands-on instruction/supervision by the codirectors. 3.1.1.3. Implementation All aspects of the program are planned, systematic, and interdisciplinary. After the initial comprehensive evaluation period, each child’s developmental profile, skills profile, specific learning and behavioral needs, and family lifestyle contribute to a written individual interdisciplinary treatment plan. This is implemented with ongoing reevaluation and revision based on daily observation, assessment, and weekly team meetings with all staff and families. The evaluation and treatment plan is developed from initial assessments and in conjunction with both parent and teacher input. Learning objectives and instructional approaches are defined, and careful data and records are kept on progress toward those goals. 3.1.1.4. Approach The ECPHP ascribes to an informed eclectic treatment approach consisting of both adult- and child-directed instructional techniques. Thus the program recognizes the strengths and contributions of behavioral, developmental, and blended intervention approaches in both the comprehensive and targeted areas. The adult-directed instruction is based on Applied Behavioral Analysis. Examples of behavioral methods used in the clinic include discrete trial teaching, and assessment of maladaptive behaviors using functional analysis of behavior methods. Discrete trial teaching includes concise instructions, clear prompt procedures, immediate reinforcement of correct responses, and repeated practice (Smith, 1999). A functional analysis of behavior includes a systematic evaluation to determine the underlying reasons that are contributing to a child’s behavior. An informed behavioral intervention plan is then created and implemented based on the results of the functional analysis. Depending on the ability of the child and the responsiveness to applied behavior analysis (ABA), the program will also use DI. This approach is an explicit, scientifically based model of effective instruction (Engelmann & Carnine, 1982). DI can be distinguished from other models of explicit instruction by its focus on curriculum design and effective instructional

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delivery. Teacher–student interactions include (1) active student participation (increasing opportunities for students to respond and receive feedback); (2) unison responding (increasing students’ responding by having them chorally respond); (3) signals (providing a cue to evoke unison oral responses); (4) pacing (promoting active student engagement with brisk teacher pacing); (5) teaching to mastery (ensuring firm responding over time); (6) error corrections (minimizing student errors by carefully sequencing instruction; when errors do occur, using careful error correction procedures—model, lead, test, retest); and (7) motivation (enhancing motivation through high levels of student success). DI is supported by independent reviews of research with particular focus on students with special needs (e.g., Carnine, Silbert, Kame’enui, & Tarver, 2004). This approach was used to develop academic, cognitive, and social skills programs. Depending on child progress, the program might also use a pivotal response treatment (PRT) approach. PRT is a comprehensive service delivery model that uses both a developmental approach and ABA procedures. PRT aims to provide opportunities for learning within the context of the child’s natural environments (Koegel & Koegel, 2006, Pivotal Response Treatments for Autism). This approach was used for some of the children’s programs when their attention and motivation were particularly difficult to maintain. The child-directed instruction is based upon principles of milieu teaching (Koegel & Koegel, 1995; Warren & Kaiser, 1986). This includes strategic environmental setup, following the child’s lead, imitating and expanding the child’s actions and communication, and overlaying contingent language onto an activity. The type of instructional approach varies across children and content domains, that is, depending on what is most effective for a particular child within a specific content area. For example, a particular child may respond best to a child-directed approach when learning how to play symbolically. The program also makes use of a variety of other empirically supported targeted instructional techniques as appropriate. Examples include “The Picture Exchange Communication System” (PECS) (Bondy & Frost, 1994, 2001), visual schedules (MacDuff, Krantz, & McClannahan, 1993), and Fast for Word (Scientific Learning Corporation, 2004a, 2004b). The ECPHP draws on multiple instructional approaches and targeted interventions to develop areas of core deficit specific to the disorder of autism (e.g., language, social engagement, joint attention, play). Indeed, areas of core deficit, and related abilities, receive particular attention within each child’s comprehensive treatment plan. Such content areas include the development of social relationships, social problem solving, emotion recognition and empathy, perspective taking, creative thinking, and initiation and organization of thought.


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3.1.1.5. Intervention targets Intervention is comprehensive and multiple domains of development are targeted. These include (1) Cognitive and Academic Skills: Children work individually and/or in small groups as appropriate to their developmental level; (2) Social development: Goals addressed include joint attention, social problem solving, emotion recognition, empathy and perspective taking, symbolic and social play, and peer interactions; (3) Self-help: This can include toilet training, self-care (e.g., brushing teeth, washing, dressing), and eating; (4) Speech and Language: Goals are to improve the communication abilities (receptive, expressive, and pragmatic language) of each child, which may include alternative forms of communication (e.g., PECS, assistive technology) if necessary; (5) Organization of behavior, sensory integration, visual and fine motor skills, and gross motor skills. These areas are targeted in Occupational and Recreational Therapy as well as throughout the day in the interdisciplinary classroom milieu; (6) Behavior Management: Both appropriate and inappropriate behaviors are evaluated. Interventions are individually implemented to increase appropriate behaviors and decrease inappropriate behaviors using approaches that are ecologically sound; and (7) Home/Family targets: All in-program interventions are described in meetings and in writing to family members. The program views parents as important partners in their child’s treatment. ECPHP offers parent support groups, parent education seminars, weekly individual meetings to address all aspects of the child’s treatment, parent training, and frequent classroom observations. Indeed, the ECPHP clearly meets all the criteria of a comprehensive model program as set forth by the National Research Council (NRC, 2001). These include (but are not limited to) individual comprehensive treatment plans, teaching approaches, and content. Treatment includes a focus on core deficits, and the analysis and treatment of maladaptive behaviors from a multimodal and functional perspective. Targeted treatments are regularly incorporated as necessary for each individual child depending on their presentation and response to treatment. Our intervention environment is strongly multidisciplinary, and highly specialized. Further, given the time constraints, we monitor progress and if children do not make measurable gains within a 3-week period (for cognitive, academic, and specialized programs), we try another intervention method. In areas of behavior, we evaluate progress weekly (in frequency and duration of maladaptive behaviors). Parents are key participants in establishing effective treatment plans for their children. Family support and parent education targeting autism in general and family-specific needs is also a key element. ECPHP specializes in working collaboratively with community agencies to facilitate appropriate and planned transitions and treatment continuity. As discussed earlier, comprehensive models should also provide evidence of outcome and change. Thus, we conducted a small pilot study examining child, preacademic outcomes, and parent satisfaction in children who attended ECPHP.

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3.2. Pilot study: method 3.2.1. Participants Participants in the pilot study included 145 children, 119 males and 26 females, who attended ECPHP from 2001 to 2008. All children came to the program with a prior clinical diagnosis of autism. If there were any questions about the accuracy of the diagnosis, the child was reevaluated using diagnostic assessments (e.g., the Autism Diagnostic Interview, the Autism Diagnostic Observation Scale, and clinical history) and all participants diagnoses were validated. To enroll in ECPHP, children were placed on the waiting list if they were between 2 and 7 years old, were diagnosed with autism by a licensed psychologist or psychiatrist, required intensive outpatient treatment (but not hospitalization), and/or similar services were not available in the community. Children were then admitted on a first come, first serve basis as space permitted. Once the child was “admitted” to ECPHP, a staff member not associated with the study provided information about data collection. Parents signed consent forms for their data to be included in the study. There was an 87.3% positive response rate. Admittance by families was due to a failure to progress or participate in school or community settings. The total length of stay for each child was determined by a combination of three variables: acute needs, goals met, and insurance coverage. Essentially, if children presented with more serious needs, were slow to make progress and/or had insurance coverage that continued to cover their stay, the child remained in the program. Conversely, reduction in the need for services, achievement of developmentally appropriate goals, and/or lack of continued insurance coverage for the program could each be potential reasons for leaving the program. The average stay was 96.56 days (SD = 45.91), or approximately 3.5 months, and the average age of the children was 52.35 months (SD = 18.90). 3.2.2. Procedure Within the first week of admission to the program, all children received a battery of assessments, including cognitive and preacademic testing. The preacademic assessments were readministered in their last week attending the program. Assessments were completed independent of personnel involved with the child’s treatment/progress. Additionally, different individuals administered different types of assessments; that is, cognitive and achievement tests were given at separate occasions by different individuals who were unaware of the child’s prior test results. This created a blind study in terms of assessment. Also at admission, parent questionnaires (a Problem Behavior Checklist and the Child Behavior Checklist—CBCL) were distributed and completed in a home environment. Only those who

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consented to participate in this study were used for the analyses. Measurable goals were established to improve the skills within the program areas appropriate and individualized for each child. Again, the focus of this pilot study was on preacademic and academic skill development. Every 3 weeks at the weekly team meeting, we determined as a clinical team whether to change the instructional format based upon the data from the child’s progress. Figures 8.1 and 8.2 describe the instructional methods used for the pilot study and for the program in general. It is to be noted, that children who stayed for longer than 3 months in the program were approved due to extreme maladaptive behaviors—not related to their cognitive and academic progress. 3.2.2.1. Assessments Table 8.2 describes the child assessments used in the study (cognitive and preacademic/achievement). Note that the WISC and WPPSI-III were treated as one cognitive variable as they both yielded standard scores on identical scales for identical cognitive domains (Verbal, Performance, and Full Scale). Table 8.3 describes the adult assessments used in the study. The

Week 1 All children: Assessments Week 2–5 All children: Structured Systematic Approach Adult Driven, DTT

Week 6–9 Continue DTT as children were 80% accurate for specific targets in unprompted structured and unstructured situations

Week 10–13 Continue DTT

Week 6–9 Non-responders: Change to PRT Motivation is a pivotal element on how content is taught more naturalistic/child-directed

Week 10–13 Continue PRT as children were 80% accurate for specific targets in unprompted structured and unstructured situations

Week 10–13 Nonresponders: Change to milieubased instructional format

Week 14 and on: Continue with instructional format and final assessments upon discharge

Figure 8.1 Group 1 (Classrooms 1 and 2) Instructional format.

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Week 1 All children: Assessments Week 2–5 All children: Direct Instruction Approach individual or small group settings

Week 6–9 Continue DI as children were 80% accurate for specific targets in unprompted structured and unstructured situations

Week 10–13 Continue DTT

Week 6-9 Non-responders: Change to PRT Motivation/interest pivotal element more individually specialized more naturalistic/child-directed

Week 10–13 Continue PRT as children were 80% accurate for specific targets in unprompted structured and unstructured situations

Week 10–13 Nonresponders: Change to DTTbased instructional format, adult directed

Week 14 and on: Continue with instructional format and final assessments upon discharge

Figure 8.2

Group 2 (Classroom 3) Instructional format.

Partial Hospital Program Patient Satisfaction Survey was provided by the hospital, after the program had ended, it was anonymous, and parents were not monitored for return.

3.3. Pilot study: results Based on the type of treatment plan, analysis was conducted based on two groups: higher and lower functioning. The lower functioning group (Group 1—the Classroom 1 and Classroom 2 children) required the PEP-R to assess their functioning while the higher group completed the WJIII (Group 2—Classroom 3 children). Descriptive data for each group are presented in Tables 8.4 and 8.5. 3.3.1. Initial confounding variables: cognitive scores, behavior report by parents, age, length of stay Correlations were conducted to assess for any confounding variables (initial cognitive scores, behavior report by parents, age, and length of stay) related

Table 8.2

Child assessments

Assessment name Cognitive Weschler Primary Preschool Scales of Intelligence—III (WPPSI-III)—(Wechsler, 2001) Wechsler Intelligence Scale for Children—III (WISC-III)— (Wechsler, 1991) Mullen Scales of Early Learning (Mullen, 1995)

Age range

Scales

Score

Form 1 = 2:6–3:11 Form 2 = 4:0–7:3

Verbal Performance

IQ (stand and score) M = 100, SD = 15

6:0–16:11

Verbal Performance

IQ (stand and score) M = 100, SD = 15

Birth—5:9

Visual Reception Fine Motor Receptive Language Expressive Language

Age equivalent

(Continued)

Table 8.2

(Continued )

Assessment name Achievement Woodcock Johnson III Test of Achievement WJ-III (Mather & Woodcock, 2001)

Psycho Educational Profile-Revised (Schopler, Reichler, Bashford, Lansing, & Marcus, 1990)

Age range

Scales

Score

2—mid-twenties

Letter Word Identity (LWI) Understanding Directions (UD) Calculation (C) Math Fluency (MF) Spelling (S) Passage Comprehension (PC) Applied Problems (AP) Word Attack (WA) Quantitative Concepts (QC) Sound Awareness (SA) Perception Imitation Cognitive Verbal Cognitive Performance

Standard scores

6 months—7 years

M = 100, SD = 15

Age equivalent

Table 8.3

Adult questionnaires

Assessment name

Subscales

Scoring

The Problem Behavior Inventory (Willis, Lavigna, & Donellan, 1993)

Muscular Problems and Habitual Mannerisms Feelings and Emotions Attention and Activity Total behavioral score

Child Behavior Checklist (CBCL) (Achenbach, 1991)

Withdrawn Somatic Complaints Anxious/Depressed Social Problems Thought Problems Attention Problems Delinquent Behavior Aggressive Behavior Total Problem Score

Scale from 1 to 7 (several times/months–every few minutes) 0–77 score range Total behavior score = Total score/ Frequency of responses Internalizing Problems Scale Externalizing Problems Scale Standard Score

(Continued)

Table 8.3

(Continued )

Assessment name

Subscales

Scoring

Partial Hospital Program Patient Satisfaction Survey

Met Needs Desire/Likelihood of Returning Rating of Child’s Condition at Discharge Staff Level of Concern How Well Staff Explained Treatment Rating the Staff Skill Satisfaction How Does Program Compare

Likert scale 1–4 Likert scale 1–4 Likert scale 1–5 Likert scale 1–5 Likert scale 1–5 Likert scale 1–5 Likert scale 1–5 Likert scale 1–5

Table 8.4 Group 1 descriptives

Achievement (PEP-R) Mean in months (SD) Imitation Perception Verbal Performance Total Pep-R IQ (Mullen) Mean in months (SD) Visual Reception (n = 72) Fine Motor (n = 70) Receptive Language (n = 72) Expressive Language (n = 71)

Admission

Change

Exit

N = 86 26.19 (11.64) 29.60 (13.78) 24.85 (11.93) 22.72 (9.49) 25.84 (10.25)

N = 86 12.36 (8.28) 14.81 (13.10) 9.77 (7.08) 10.85 (7.64) 11.95 (6.67)

N = 86 38.55 (13.25) 44.41 (14.53) 34.62 (13.43) 33.57 (11.01) 37.79 (11.43)

25.25 (7.29) 24.79 (8.25) 21.80 (10.47) 20.06 (10.93)

– – – –

– – – –

(Continued)

Table 8.4

(Continued )

Behavior (CBCL) Mean T-score (SD) Internalizing Externalizing Total Behavior Anxiety (n = 69) Behavior (Problem Behavior) Mean (SD) Muscular Problems and Habitual Mannerisms Feelings and Emotions Attention and Activity

Admission

Change

Exit

N = 70 62.29 (7.63) 58.67 (9.13) 32.49 (8.60) 56.27 (7.92)

– – – –

– – – –

N = 72 2.49 (0.90)

–

–

2.26 (0.90) 3.24 (1.47)

– –

– –

Table 8.5

Group 2 descriptives

Achievement (WJ) Mean standard score (SD) Understanding Directions Applied Problems Word Attack Quantitative Concepts Sound Awareness Total WJ IQ (WPPSI) Mean standard score (SD) Performance IQ Verbal IQ Full Scale IQ

Admission

Change

Exit

N = 58 100.17 (14.71) 95.29 (15.68) 106.97 (24.20) 101.81 (17.59) 88.69 (27.94) 98.93 (15.68)

N = 58 5.51 (10.05) 7.71 (9.40) 11.03 (17.57) 11.10 (13.52) 15.56 (20.59) 10.12 (9.77)

N = 58 105.68 (13.10) 103.00 (16.40) 118.00 (22.70) 112.92 (18.11) 104.25 (23.11) 109.05 (15.83)

N = 56 95.61 (22.97) 89.88 (22.06) 93.21 (19.15)

– – –

– – – (Continued)

Table 8.5

(Continued )

Behavior (CBCL) Mean T-score (SD) Internalizing Externalizing Total Behavior Anxiety Behavior (Problem Behavior) Mean (SD) Muscular Problems and Habitual Mannerisms

Admission

Change

Exit

N = 43 66.98 (10.70) 62.68 (10.32) 67.40 (9.93) 65.93 (12.54)

– – – –

– – – –

N = 52 2.49 (0.90)

–

–

– –

– –

Feelings and Emotions

2.26 (0.90)

Attention and Activity

3.24 (1.47)

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to the outcome variables (change on the PEP-R or Woodcock Johnson). For Group 1, scores on the Mullen Scales of Early Learning were not related to change scores on the PEP-R. Additionally, scores on the WPPSI were not related to change scores on the Woodcock Johnson for Group 2. No relationships were found between the behavioral variables for either group and the outcome variables. This seems to indicate that improvements were made regardless of initial behavioral impairments. Length of stay in the program did not relate to change scores for either group. Age at admission did not relate to change scores on the PEP-R for Group 1. For Group 2, age at program entry was related only to the change score on the Word Attack subscale of the Woodcock Johnson (r = –0.31, p = 0.02). 3.3.2. Pre- and posttreatment change Eighty-six participants were included in Group 1. The average age was 3.5 years and the average length of stay was 3 months. Group 2 comprised of 58 participants. The average age was 5.7 years and average length of stay was 3 months. 3.3.2.1. Change from entry to exit The means on the PEP-R subscales of Perception, Performance, Verbal, and Imitation including an overall score were greater posttreatment than pretreatment. A paired-samples two-tailed t-test was performed for pre- and posttreatment PEP-R scores on the subscales, showing that the means were significantly different (see Table 8.6). The total average increase was 11.95 months across all subscales. The means on the WJ subscales were computed as pre- and posttreatment; Understanding Directions (UD), Applied Problems (AP), Word Attack (WA), Quantitative Concepts (QC), and Sound Awareness (SA). For all variables, the posttreatment mean was higher than the pretreatment mean score (see Table 8.5). A paired-samples two-tailed t-test was performed on the pre- and posttreatment subscores; all of the subscores were significant (see Table 8.7). The total average improvement was 10.12 standard score points across all subscales. Table 8.6

cN t-Value df

Group 1 PEP-R t-test

Imitation

Perception

Verbal

Performance

86 12.98** 85

86 10.40** 85

86 12.80** 85

86 13.17** 85

* p < 0.05, **p < 0.01.

Table 8.7

Group 2 Woodcock Johnson t-test

N t-Value df p < 0.05,

*

p < 0.01.

**

Understanding

Applied

Word

Quantitative

Sound

Directions 58 4.43** 57

Problems 58 6.14** 57

Attack 58 4.73** 57

Concepts 58 6.27** 57

Awareness 58 5.83** 57

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3.3.3. Treatment pathway 3.3.3.1. Group 1 Using the average score from admission to discharge on the PEP-R, as can be seen in Fig. 8.3, 45% of children made gains using DTT, 30% made gains using PRT, 25% made gains using milieu-based instruction. All children made better-than-age appropriate progress by discharge. 3.3.3.2. Group 2 As can be seen in Fig. 8.4, 33% of children made gains using DI, 33% made gains using PRT, 33% made gains using DTT instruction. All children made better-than-age appropriate progress by discharge. 3.3.4. Satisfaction Out of the 75 families who received the satisfaction survey, 42 questionnaires were returned (56%). All the targeted questions were answered by all 42 families except for “How does this program compare with other similar services or programs you’ve experienced in the past?” On this question, only 35 families responded. For the seven that did not respond, families had stated that they had not had “similar services” or “prior services.” As can be seen from Figs. 8.5 and 8.6, parents felt that the program met all their needs and that their child’s condition was much improved. Further, the staff’s skills, concern, and explanations of treatment were excellent. Finally, the families were very satisfied and felt the program provided much better service than prior experiences.

45

40

35 Group 1 Group 2

30

Group 3 25

20 Week 0

Figure 8.3

Week 3

Week 6

Classroom 1 progress by group.

Week 9

Week 12

232


120 115 110 Group 1

105

Group 2 Group 3

100 95 90 Week 0

Figure 8.4

Week 3

Week 6

Week 9

Week 12

Classroom 2 progress by group. 3.95

Mean score

4

3.69

3.5 3 2.5 2 Met needs

Figure 8.5

Likelihood of returning

Parent Satisfaction Questionnaire: Mean score for four point questions. Mean score

5

4.786

4.977

4.977

4.977

4.901

4.943

Staff concern

Staff explanations

Staff skill

Satisfaction

Program comparison

4.5 4 3.5 3 2.5 2 Rating of child's condition

Figure 8.6

Parent Satisfaction Questionnaire: Mean scores for five point questions.


233

3.4. Pilot study: discussion During the time in which they attended ECPHP, the children attained statistically significant improvements on preacademic achievement measures from pre to post treatment. These changes are remarkable, given that the children were in the ECPHP for an average of 3.22 months (SD = 1.53). This is greater than chronological age improvement (for children who had shown delayed improvement in the past). The parents also felt that the improvement was tied to the comprehensive intervention and they felt highly satisfied. Altogether, the data support the ECPHP’s comprehensive interdisciplinary program in terms of improving children’s preacademic functioning. Thus, it seems possible that effectiveness of this intervention lies in its informed eclectic response to treatment approach within a defined treatment period incorporating the behavioral and complementary methods, as well as the recommendations proposed by Rutter (1985) and the National Research Council (2001). This pilot data is limited in that it does not employ a randomized control design, and also does not report on other outcome measures (e.g., social development, behavior), although this is the next step. While the within-group pretreatment/posttreatment design, however, is an acceptable approach to program evaluation in the field of early childhood special education (Fewell & Sandall, 1986), we do recognize that such designs do not demonstrate causality unambiguously. Rather, we view this pretreatment/posttreatment methodology as a first step toward more rigorous controlled intervention research. We also acknowledge that we have not included formal data on the contribution of other interventions and therapies in which children participated; however, given the intensive and comprehensive nature of the ECPHP, the majority of children do not receive other concurrent interventions.

4. Integrated Summary The chapter reviewed the recent review literature on both comprehensive intervention programs and targeted treatments. Despite the growing focus on intervention and a respectable number of treatment studies, it is increasingly clear that the heterogeneous nature of the disorder does not allow for a simple “one size fits all” treatment approach, nor for straightforward measurement of treatment effectiveness. Based on the synthesis of intervention review research in this chapter and the pilot clinical treatment outcomes from the Early Childhood Partial Program at UCLA, we present a different way of studying treatment effectiveness. Rather than focusing on intervention approach only, and

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measuring effectiveness of one approach, we suggest measuring intervention effectiveness in terms of the model, that is, how intervention is delivered to ensure achievement of specified goals for all children. If some children are nonresponders, change the methodology. Very recent statistical methodology designed for the treatment of drug addiction has demonstrated this approach by employing “adaptive treatment strategies.” These strategies are guided by a patient’s responses to prior treatment and thus change within the design of the study (see SMART design; Scott, Levy, & Murphy, 2007). Although our pilot study was not randomized (as in the SMART design), the idea of examining efficacy of individual treatments within a sequence, and the efficacy of the sequences themselves, offer an enticing future design for conceptualizing interventions for children on the autism spectrum. The ECPHP was described as an effective comprehensive treatment program that uses systematic targeted treatments on an individual basis. The elements of the model are unique: the interdisciplinary team, the active and dynamic decision-making, the attention by knowledgeable clinicians to children’s treatment, contribution of recent research, intensive teacher training, unchanging intensity (but with a different focus for each child to achieve gains), and generally fixed program length. These are all critical variables in contributing to change. But perhaps the most powerful variable of the program treatment model is the constant evaluation of child progress in determining if the treatment approach being used is effective. If not, after a period of time (3 weeks), based on child characteristics and child response to treatment, treatment approach is changed and reevaluated. Thus the focus of intervention shifts from a single treatment approach (“what”) to the design of a multiapproach treatment delivery model (“how”). We suggest that this shift in thinking best meets the needs of autism treatment, from both clinical and research perspectives. It embraces the heterogeneity and complexity of autistic disorder and achieves a best possible outcome for all children.

Acknowledgments The authors would like to thank Dorwin Birt for his assistance with the database creation and management. We thank Connie Kasari, Robert Hodapp, and Steven Forness for their feedback on drafts of this chapter. We also thank Agnieszka Blazejko and Nisha Bansal for their help with entering data. Most important, we thank all the staff, families, and children, of the Early Childhood Partial Hospitalization Program.


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C H A P T E R N I N E

Epidemiology of Mental Illness and Maladaptive Behavior in Intellectual Disabilities Johannes Rojahn* and Lisa J. Meier* Contents 1. Introduction 2. Intellectual Disability 3. Psychopathology in Intellectual Disability 3.1. Mental illness 3.2. Maladaptive behavior 3.3. Maladaptive behaviors as behavioral equivalents 4. Epidemiology 4.1. Prevalence 4.2. Incidence 5. Risk Factors and Correlates 5.1. Levels of intellectual disability 5.2. Chronological age 5.3. Sex 5.4. Comorbidity with autism spectrum disorder 5.5. Genetic disorders—behavioral phenotypes 5.6. Social–environmental factors 5.7. Psychological coping skills 6. Conclusions References

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Abstract The following chapter discusses a literature review on the epidemiology of psychopathology in intellectual disabilities (ID), with a focus on research published since 2000. Substantial differences in prevalence and incidence *

Department of Psychology, George Mason University



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Johannes Rojahn and Lisa J. Meier

estimates across publications due to methodological discrepancies are a concern but some of the more recent studies from Australia and some European countries (foremost the UK) show noticeable technical improvements. In summary, prevalence of global mental illness across all ID levels, sex, and age ranged from roughly 16% to 54%. Psychopathology in children with ID was 4.8 and 4.5 times higher than in typically developing comparison groups. Maladaptive behavior prevalence ranged from 0.1% to 23% (aggressive behavior 6% to 32%, self-injurious behavior 4% to 21%, and destructive behavior 2% to 19%). While the presence of a mental illness does not seem to be associated with cognitive functioning levels, maladaptive behavior is strongly negatively correlated, especially self-injurious and stereotyped behavior. Age does not affect the presence of mental illness while challenging behaviors increase from childhood to early or mid adulthood, before diminishing as people age. While sex does not predict general mental illness, evidence points to a male predominance in aggressive and destructive behavior. Comorbid autism spectrum disorder (ASD) is a risk factor for concurrent mental illness and challenging behaviors. There is growing evidence that certain forms of psychopathology may be phenotypic for certain chromosomal conditions. Vulnerability for psychopathology in ID, as in the general population, is a function of complex interactions of often still poorly understood factors that include neurobiological substrates, personal characteristics, and socioeconomic circumstances.

1. Introduction For centuries, psychiatric disorders in individuals with intellectual disability (ID) were ignored. Maladaptive behaviors, especially severe ones, of course were never overlooked but it took a long time before the possibility of a conceptual link between some of those behaviors and psychiatric disorders began to dawn. Indeed, mental illness was considered by some to be incompatible with ID due to insufficient ego strength or lack of cognitive abilities (Matson, 1988a). It was arguably not until the pivotal Isle of Wight total population prevalence study by Rutter, Graham, and Yule (1970) that professionals and researchers began to appreciate and acknowledge the fact that major mental illnesses such as mood disorders, anxiety disorders, and schizophrenia could occur alongside ID. Rutter et al.’s (at that time) surprising discovery has since been corroborated. However, the issue of psychopathology is complex, particularly when it is comorbid with ID. The purpose of this chapter is to review the literature on the epidemiology of psychopathology in ID, to summarize what we know at this point, to identify gaps in our methodology, and to point out future directions. First, key concepts of ID and psychopathology must be defined.

Epidemiology of Mental Illness and Maladaptive Behavior

241

2. Intellectual Disability There is a broad international consensus1 about the three core characteristics of ID: (1) significantly subaverage intellectual functioning as determined by an intelligence quotient (IQ) score of two standard deviations below the mean (70–75 or below); (2) significant deficits in adaptive functioning, and (3) the onset before the age of 18 years. Definitions do diverge slightly as to the classification of functional levels or needed supports. The range of severity is typically defined as mild (IQ 50–55 to 70), moderate (IQ 35–40 to 50–55), severe (IQ 20–25 to 35–40), and profound (IQ below 20–25). Most epidemiological studies show prevalence rates of 1–1.5%. For instance, the 1994/1995 National Health Interview Survey— Disability Supplement found a prevalence rate of ID below 1% of the U. S. population (University of Minnesota, 2000, April).

3. Psychopathology I n Intellectual Disability In this chapter, the term psychopathology is used as an umbrella term that covers all major forms of mental illness, as well as maladaptive behaviors, whether or not they are symptoms of a psychiatric disorder.

3.1. Mental illness Mental illness includes the clinical disorders (Axis I) and the personality disorders (Axis II) of the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Text Revision (DSM-IV-TR; American Psychiatric Association, 2000). While the multiaxial DSM-IV-TR has widely accepted diagnostic criteria for the Axis II condition of mental retardation/intellectual disabilities, its usefulness for diagnosing other psychiatric disorders in the ID population has been questioned. Recently, NADD (National Association for the Dually Diagnosed) published the Diagnostic Manual-Intellectual Disability: a Textbook of Diagnosis of Mental Disorders in Persons with Intellectual Disability (DM-ID; NADD, 2007). In association with the American Psychiatric Association, NADD modified and adjusted DSM-IV-TR criteria for major psychiatric disorders so that the diagnostic criteria are more appropriate for individuals with ID and also suggests strategies on how to use the multiaxial system with this special population. Its usefulness and psychometric 1

Including the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Text Revision (DSM-IV-TR) (American Psychiatric Association, 2000), the Definition, Classification and System of Supports adopted by the American Association on Intellectual and Developmental Disabilities (AAMR/AAID) (Lucksson et al., 2002), the Diagnostic Criteria for Psychiatric Disorders for Use with Adults with Learning Disabilities/Mental Retardation (DC-LD; Royal College of Psychiatrists, 2001), the International Classification of Diseases and Related Health Problems (ICD-10; World Health Organization, 1994), and the International Classification of Functioning, Disability, and Health (ICF; World Health Organization, 2001).

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properties are yet to be evaluated. A similar attempt had been made by the Royal College of Psychiatrists (2001) with the development of the Diagnostic Criteria for Psychiatric Disorders for Use with Adults with Learning Disabilities/ Mental Retardation (DC-LD), which was designed to complement the ICD-10 for individuals with mild ID (World Health Organization, 1992).

3.2. Maladaptive behavior As a group, individuals with ID are prone to develop some form of maladaptive behavior during their lifetime. The most common maladaptive behaviors in this group include aggression, destructive behavior, selfinjurious behavior, and stereotyped behavior, oppositional behavior, overly demanding behavior, eloping (wandering), and sexually inappropriate behavior. Such behaviors can escalate to a point when they become serious safety concerns. In a clinical context, behavior problems are often defined informally and ad hoc because terms such as “aggressive behavior” seem to have high face validity. As we shall demonstrate, however, it can make a difference how behavior problems are defined when trying to determine their prevalence and their relationship to psychiatric disorders. Especially for research purposes, it is important to use standardized instruments and operationalized definitions (for a more in-depth discussion on the assessment of maladaptive behavior, see Rojahn, Hoch, Whittaker, & Gonza´lez, 2007). This is especially true for epidemiological research. The Royal College of Psychiatrists (2001) established criteria that need to be met before a behavior can be diagnosed as a “behavior problem”: (1) it has to be serious enough as to require clinical assessment and interventions, (2) it is not a direct consequence of other psychiatric disorders (e. g., pervasive developmental disorders), drugs or physical disorders (which is not always easy to determine), (3) it has either a significant negative impact on someone’s quality of life, or presents health and/or safety risks, and (4) it is persistent and pervasive across a range of personal and social settings.

3.3. Maladaptive behaviors as behavioral equivalents One hypothesis as to why maladaptive behaviors are so prevalent in individuals with ID is that some of them may be a behavioral equivalent, or an atypical manifestation of psychiatric disorders. The term was coined by Sovner and Hurley (1982a,b). They argued that because individuals with ID function differently from those of average or above intelligence, the clinical features of their psychological illnesses could also present differently. For example, some have suggested that self-injury, aggression, or screaming might be behavioral equivalents of depression in individuals with severe or profound ID (Marston, Perry, & Roy, 1997; Reiss & Rojahn, 1993). Charlot


243

(2005) pointed out that, in order to decide whether a behavior should be considered a behavioral equivalent, one has to follow the course of the behavior and the other symptoms of the psychiatric disorder. In mood disorders, for example, onset and duration of the behavior equivalents must be synchronized with the episodic onset and duration of the mood shifts. Although the idea of behavioral equivalents is intriguing and clinically plausible, strong empirical evidence for their existence is still lacking. A few studies have supplied data consistent with the general notion of behavioral equivalents—namely that challenging behaviors are differentially associated with one or more specific psychiatric conditions (e. g., Felce, Kerr, & Hastings, 2009; Hemmings, Gravestock, Pickard, & Bouras, 2006; Holden & Gitlesen, 2003; Kishore, Nizamie, & Nizamie, 2005; Rojahn, Matson, Naglieri, & Mayville, 2004), while other did not find such associations (e. g., Tsiouris, Mann, Patti, & Sturmey, 2003). But even among those studies that found associations between behavior problems and psychiatric conditions, considerable inconsistencies were found across studies. In addition, studies that showed the strongest association between behaviors and specific psychiatric conditions were those in which the behaviors were part of the diagnostic criteria for that disorder (e. g., stereotyped behavior linked to pervasive developmental disabilities). The relationship between psychiatric disorders and behavior problems might be stronger for individuals with lower levels of functioning (Felce et al., 2009). At this stage, the safest conclusion would be that some maladaptive behaviors can be nonspecific indicators of emotional distress associated with psychiatric conditions, rather than atypical symptoms of specific disorders.

4. Epidemiology Although most experts acknowledge that people with ID are vulnerable to a wide range of psychiatric disorders, reliable epidemiological data remain elusive. This is in part due to a host of quandaries such as (1) the difficulty in reaching consensus on definitions and classifications of many psychiatric disorders in individuals with ID, (2) the unique challenge of diagnosing individuals with limited capacity to reflect upon and report their thoughts, feelings and experiences, (3) difficulty in drawing representative samples that allow comparisons between studies, and (4) the screening and assessment differences across epidemiological studies that result in very different results and make it difficult to compare outcomes. Differences between studies can be found in sampling, assessment instruments, and data reporting. For instance, among 23 publications that we surveyed for this chapter, 22 different assessment instruments were used either as single instruments or in various combinations (see Table 9.1). Striking evidence

Table 9.1 Summary of international epidemiological studies on psychopathology in intellectual disabilities since the year 2000 listed in alphabetical order by author

1.

2.

Authors

Reference population and sampling procedure

Definition of the concept and assessment tools

Resultsa

Cooper et al. (2009a)

Location: Glasgow, UK Sampling method: Individuals with ID living within the Greater Glasgow Health Board area. Phase 1: 2002–2004 Phase 2: 2004–2006 Nphase 1 = 1023 Nphase 2 = 651 Ages: 16 years and older ID: mild to profound Location: Glasgow, UK Sampling method: Individuals with ID living within the Greater Glasgow Health Board area 2002–2004. Nt1 = 184 (time 1) Nt2 = 131 (2 years later) Ages: 16–82 years

PAS-ADD Checklist (screen) PPS-LD:

Point prevalence of self-injurious behavior: 4.9% Two-year incidence of self-injurious behavior: 0.6% Two-year remission rate: 38.2%

Cooper et al. (2007)b

• DC-LD

PAS-ADD Checklist PPS-LD (which includes four different types of diagnosis): • Clinical diagnosis • DC-LD

Point prevalence of mental illness at time1 (as a function of diagnostic measure): 52.2%—clinical diagnosis 45.1%—DC-LD 10.9%—DCR-ICD-10 11.4%—DSM-IV-TR

ID: profound

• DCR-ICD-10 • DSM-IV-TR

Point prevalence of problem behavior at T1 (as a function of diagnostic measure): 39.1%—clinical diagnosis 33.7%—DC-LD 0.0%—DCR-ICD-10 0.0%—DSM-IV-TR Two year incidence of mental illness (varied by measure): 13.0%—clinical diagnosis 12.2%—DC-LD 6.1%—DCR-ICD-10 2.3%—DSM-IV-TR Two-year incidence of problem behavior (as a function of diagnostic measure): 6.1%—clinical diagnosis 6.1%—DC-LD 0.0%—DCR-ICD-10 0.0%—DSM-IV-TR (Continued)

Table 9.1

3.

(Continued )

Authors



Resultsa

Cooper et al. (2009b)

Location: Glasgow, UK Ages: 16 years and older Sampling method: Individuals with ID living within the Greater Glasgow Health Board area. Phase 1: 2002–2004 Phase 2: 2004–2006 Nphase 1 = 1,023 Nphase 2 = 651 Ages: 16 years and older ID: mild to profound

PAS-ADD Checklist (screen) PPS-LD:

Point prevalence of aggressive behavior Phase 1: 9.8%

• DC-LD

• 6.3 %—physically aggressive • 3.0%—destructive • 7.5%—verbally aggressive Two-year incidence of aggressive behavior: 1.8% • 0.6 %—physically aggressive • 0.6%—destructive • 1.4%—verbally aggressive Two-year remission rate: 27.7% • 31.5 %—physically aggressive • 29.4%—destructive • 32.1%—verbally aggressive

4.

Cooper et al. (2007)b

Location: Glasgow, UK Sampling method: Individuals with ID living within the Greater Glasgow Health Board area between 2002 and 2004 N = 1,023 Ages: 16 years and older ID: mild to severe

5.

Crocker et al. (2006)

Location: Quebec, Canada Sampling method: Individuals with ID receiving services from three rehabilitation agencies N = 3,165 Ages: 18 years and older ID: mild to profound

PAS-ADD Checklist (screen) PPS-LD (which includes four different types of diagnosis):

Point prevalence of mental illness (as a function of diagnostic measure): 40.9%—clinical diagnosis 35.2%—DC-LD 16.6%—DCR-ICD-10 15.7%—DSM-IV-TR Point prevalence of problem • Clinical diagnosis behavior (as a function of • DC-LD diagnostic measure): • DCR-ICD-10 22.5%—clinical diagnosis • DSM-IV-TR 18.7%—DC-LD 0.1%—DCR-ICD-10 0.1%—DSM-IV-TR MOAS (modified Prevalence of aggressive behavior to include over 12 months: sexual 53.9%—total aggressive behavior aggression) • 25%—physical • 37.6%—verbal • 25%—property • 25%—self • 9.8%—sexual (Continued)

Table 9.1

6.

(Continued )

Authors



Resultsa

Dekker and Koot (2003)

Location: Holland, Netherlands Sampling method: Random sample from schools for children with ID in the province of Zuid N = 474 Ages: 7–20 years ID: borderline to moderate

DISC-IV-P PDD-MRS

Prevalence of mental illness: (over 1 year) 38.6%—any DSM-IV disorder 21.9%—anxiety disorder 4.4%—mood disorder 25.1%—disruptive behavior disorder • Any ADHD 14.8% • ODD 13.9% • Conduct disorder 3.0% 14.2%—more than one DSM-IV disorder 40.7%—DSM-IV disorder comorbid with PDD

7.

8.

de Ruiter et al. (2007)

Einfeld et al. (2006)

Location: Holland, Netherlands Sampling method: Random sample from schools for children with ID in the province of Zuid ID Samples: Nt1 = 953 (1997) Nt2 = 463 (1998) Nt3 = 509 (2002) Ages: 6–18 years Mean Ages: T1 = 11.7 years T2 = 12.8 yaers T3 = 15.3 years ID: borderline to moderate Location: Australia rural and urban agencies Sampling method: Longitudinal study with four measurements (about every 3 years) between (1991 and 2003) N = 507 Ages: 5–19 years (wave 1) ID: mild to profound

CBCL • Withdrawn • Delinquent behavior • Social problems • Anxious/ depressed • Aggression • Thought problems • Somatic complaints • Externalizing • Attention problems • Internalizing DBC subscales: • Disruptive • Self-absorbed • Communication Disturbance • Anxiety • Social Relating Disturbance • Total Behavior Problem Score

Prevalence of mental illness: Not reported Mean CBCL scores:

Age in years

ID

Non-ID

6–8 9–11 12–14 15–18

39.1 33.2 33.0 29.2

20.5 20.2 17.7 16.6

Prevalence of mental illness: 41%—significant DBC-type psychopathology at first measure 31%—at fourth measure

(Continued)

Table 9.1

9.

(Continued )

Authors



Resultsa

Emerson (2003)

Location: Great Britain Sampling method: Retrospective analysis of 1999 data files from Office for National Statistics on mental health of British children Ntotal = 10,438 N1 = 264 with ID N2 = 10,174 without ID Ages: 5–15 years ID: not specified

DAWBA (ICD-10 and DSMIV criteria)

Prevalence of mental illness in children with and without ID: Children/adolescence with ID 39%—any psychiatric disorder 9.5%—any emotional disorder 8.7%—any anxiety disorder 1.5%—depression 25.0%—any conduct disorder 7.6%—PDD 8.7%—hyperkinesis Children/adolescence without ID 8.1%—any psychiatric disorder 4.1%—any emotional disorder 3.6%—any anxiety disorder 0.9%—depression 4.2%—any conduct disorder 0.1%—PDD 0.9% hyperkinesis

10.

Emerson and Hatton (2007)

11.

Emerson et al. (2001)

Location: Great Britain Sampling method: Retrospective analysis of 1999 and 2004 data files from Office for National Statistics on mental health of British children N1 = 641 with ID N2 = 17,774 without ID Ages: 5–15 years ID: not specified Location: District Health Authorities in UK Sampling method: 1. All ID service programs identified (residential, day, respite, etc.) 2. Screening of each setting 3. Personal information on people with maladaptive behavior N1995 = 2,189 Age: not reported; children and adults ID: not reported

DAWBA (ICD-10 and DSM-IV criteria)

CBS-IS

Point Prevalence of mental illness: 36%—significant psychopathology in those with ID 8%—significant psychopathology in those without ID

Prevalence of maladaptive behavior: 12.1%—overall maladaptive behaviors • • • •

6.7%—aggressive behavior 3.4%—self-injurious behavior 4.0%—destructive behavior 9.8%—other maladaptive behavior Prevalence data calculated to represent percentage of total N screened (Continued)

Table 9.1

(Continued )

Authors



Resultsa

12.

Gustafsson and Sonnander (2004)

RSMB PIMRA

Prevalence of mental illness (varied by measure): RSMB—37% of total sample PIMRA—54% of total sample

13.

Holden and Gitlesen (2003)

Location: Sweden Sampling method: Mental Health Services Register to identify adults with ID also receiving out or inpatient psychiatric care N = 294 Ages: 18 years and older ID: mild to severe Location: Norway Sampling method: All lived at home or local facilities referred for services in two counties of Norway Ntotal = 165 Nwith maladaptive behavior = 105 Nwithout maladaptive behavior = 60 Ages: 18 years and older ID: mild to profound

PASS-ADD Checklist DSM-IV Type of Maladaptive Behavior

Prevalence of mental illness: Total sample: 21.3%—anxiety 30.0%—depression 8.4%—hypomania 15.5%—psychosis With maladaptive behavior 27.1%—anxiety 27.1%—depression 9.4%—hypomania 19.8%—psychosis

• • • •

aggression destruction self injury other

14.

15.

Holden and Gitlesen (2006)

Hove and Havik (2008a)

Location: Hedmark, Norway Sampling method: All people, both children and adults, with administratively defined mental retardation, living in Hedmark, Norway N = 904 Ages: 0–89 years ID: mild to profound

CBS-IS

Location: Western Norway Sampling method: Identified as receiving community based services for ID in 32 municipalities N = 593 Ages: 18 years and older ID: mild to profound

P-AID (based on DC-LD)

Without maladaptive behavior 11.9%—anxiety 32.2%—depression 6.8%—hypomania 8.5%—psychosis Prevalence of maladaptive behavior: 11.1%—overall maladaptive behavior Forms of maladaptive behavior: • • • •

6.4%—attacking others behavior 4.4% self-injurious behavior 2.3%—destructive behavior 7.1%—other difficult, disruptive or socially unacceptable behavior

Three-month point prevalence of mental illness: 34.9%—mental disorders 43.0%—mental disorder or behavior problem 15.9%—anxiety disorders 12.3%—affective disorders 4.6%—psychoses 20.2%—problem behavior specifically (Continued)

Table 9.1

(Continued )

Authors



Resultsa

16.

Jones et al. (2008)b

PAS-ADD Checklist DC-LD criteria for problem behavior

Point prevalence of problem behaviors varied by criteria: 22.5%—problem behavior by psychiatrists’ opinion 18.7%—problem behavior by DC-LD

17.

Lowe et al. (2007)

Location: Glasgow, UK Sampling method: Individuals with ID living within the Greater Glasgow Health Board area 2002–2004. N = 1,023 Ages: 16–83 years ID: mild to profound Location: South Wales, UK Sampling method: All services providers in seven unitary authorities in the for people with intellectual disabilities were screened N1 = 1,458 individuals screened with Setting Questionnaire N2 = 901 after attrition administered CBS-IS (e. g., no consent) Ages: 5–93 years ID: not reported

CBS-IS

Prevalence of serious maladaptive behavior: 10%—of LD population had seriously maladaptive behavior Out of N2 = 901 32%—aggressive behavior 21%—self-injurious behavior 19%—destructive behavior 39%—disruptive behavior

18.

Morgan et al. (2008)

19.

Myrbakk and von Tetzchner (2008)

Location: Western Australia Sampling method: Cross referenced Mental Health Information System and Intellectual Disability Register N = 245,749 Ages: 23–37; 38–52 ID: all levels including borderline Location: Norway Sampling method: Administrative sample of individuals with ID receiving support from municipality Ntotal = 181 with ID N1 = 71 with behavior problems N2 = 71 without behavior problems Ages: 14–70 years ID: Mild to profound

ICD-9 Data extracted from registers in 2003

Prevalence of mental illness: 31.7%—with ID had a psychiatric disorder (1.8%—with a psychiatric disorder had ID)

ABC (moderate or severe behavior problems) RSMB Mini PAS-ADD ADD DASH-II

Prevalence of psychiatric symptoms in ID individuals with and without behavior problems: 69%—psychiatric symptoms in the group with behavior problems 29%—psychiatric symptoms in the group without behavior problems

(Continued)

Table 9.1

(Continued )

Authors



Resultsa

20.

Smiley et al. (2007)b

Location: Glasgow, UK Sampling method: Individuals with ID living within the Greater Glasgow Health Board area. (Follow up from Cooper, 2007) N = 651 Ages: 16 years and older, time 1 18–80 years, time 2 ID: mild to profound

PPS-LD PAS-ADD Checklist Clinical diagnosis DC-LD ICD-10 DSM-IV-TR

21.

Tsakanikos et al. (2006)

ICD-10 Location: London, UK Psychiatric interview Sampling method: 20-year retrospective record review for those referred for mental health services Ntotal = 590 Nmen = 295 Nwomen = 295 Ages: adults ID: mild to severe

Two-year incidence of mental illness varied by measure: 16.3%—clinical diagnosis 14.7%—DC-LD 8.4%—DCR-ICD-10 6.8%—DSM-IV-TR Two-year incidence of problem behavior varied by measure: 4.6%—clinical diagnosis 3.5%—DC-LD 0.0%—DCR-ICD-10 0.0%—DSM-IV-TR Prevalence of mental illness: 49% of total sample had a psychiatric disorder 47% of men had a disorder • 17.3% schizophrenia spectrum • 9.2% personality disorder • 7.5% anxiety • 5.8% depressive disorder • 4.1% adjustment reaction • 1.7% dementia 52% of women had a disorder

22.

Tsakanikos et al. (2006)

Location: London, UK Sampling method: Individuals with ID referred to Specialist Mental Health Service of London N1 = 605 ID without Autism N2 = 147 ID with Autism Ages: 16–84 years ID: mild to severe

ICD-10 PAS-ADD Checklist

• 16.3% schizophrenia spectrum • 4.7% personality disorder • 6.4% anxiety • 7.8% depressive disorder • 7.8% adjustment reaction • 5.8% dementia Prevalence of mental illness in ID individuals with and without Autism: ID with Autism: 16.4%—schizophrenia spectrum 2.9%—personality disorder 4.3%—anxiety 6.4%—depressive disorder 5.0%—adjustment reaction 1.4%—dementia ID without Autism: 18.5%—schizophrenia spectrum 9.0%—personality disorder 8.1%—anxiety 9.0%—depressive disorder 6.5%—adjustment reaction 4.2%—dementia (Continued)

Table 9.1

23.

a

(Continued )

Authors



Resultsa

Tyrer et al. (2006)

Location: Leicestershire, UK Sampling method: Individuals with ID listed on the Leicestershire register N = 3,065 Ages: 19 years and older ID: mild to profound

DAS

Prevalence of physical aggression towards others was 14%

Unless otherwise specified, percentage in the results section stands for the percentage among those individuals with ID who were identified as cases among those who were actually screened for caseness. Indicates research based on same initial database.ABC = Aberrant Behavior Checklist (Aman & Singh, 1986). ADD = Assessment of Dual Diagnosis (Matson & Bamburg, 1998). AIRP Assessment and Information Rating Profile (Bouras, 1995). CBCL = Child Behavior Checklist (Achenbach, 1991). CBS-IS = Challenging Behavior Survey—Individual Schedule (Alborz, Emerson, Kiernan, & Qureshi, 1994). DAS = Disability Assessment Schedule (Holmes, Shah, & Wing, 1982). DASH-II = Diagnostic Assessment of the Severely Handicapped (Matson, 1995). DAWBA = Development and Well Being Assessment (Goodman, Ford, Richards, Gatward, & Meltzer, 2000). DBC = Developmental Behavior Checklist (Einfeld & Tonge, 2002). DC-LD = Diagnostic Criteria for Psychiatric Disorders for use with Adults with Learning Disabilities/Mental Retardation (Royal College of Psychiatrists, 2001). DCR-ICD-10 = Diagnostic Criteria for Research - International Classification of Diseases Tenth Revision (World Health Organization, 1993). DISC-IV-P = Diagnostic Interview Schedule for Children-Parent Version (DSM-IV) (Shaffer, Fisher, Lucas & Comer, 2000; Shaffer, Fisher, Lucas, Dulcan & Schwab-Stone, 2000). DSM-IV-TR = Diagnostic and Statistical Manual of Mental Disorders: Fourth Edition-Text Revision (American Psychiatric Association, 2000). ICD-10 = International Classification of Diseases Tenth Revision (World Health Organization, 1992) Mini PAS-ADD = Mini Psychiatric Assessment Schedule for Adults with Developmental Disability (Prosser, Moss, Costello, Simpson, & Patel, 1997). MOAS = Modified Overt Aggressive Scale (Kay, Wolkenfeld, & Murrill, 1988). P-AID = Psychopathology Checklist for Adults with Intellectual Disability (Hove & Havik, 2008b). PAS-ADD = Psychiatric Assessment Schedule for Adults with Developmental Disabilities (Moss et al., 1998). PDD-MRS = Scale of Pervasive Developmental Disorder in Mentally Retarded Persons (Kraijer, 1997). PIMRA = Psychopathology Inventory for Mentally Retarded Adults (Matson, 1988b). PPS-LD = Present Psychiatric State for Adults with Learning Disabilities (Cooper, 1997). RSMB = Reiss Screen for Maladaptive Behavior (Reiss, 1988). Setting Questionnaire (Kiernan & Qureshi, 1986).

b


259

of the dependence of prevalence rates on the definitions of the target phenomena, classifications systems, and assessment instruments was provided by Cooper, Smiley, Morrison, Williamson, and Allan (2007; Study 4, Table 9.1) and Cooper et al. (2007; Study 2, Table 9.1). Prevalence estimates of psychiatric disorders ranged from 15.7 to 45.1%, depending on whether DC-LD criteria (Royal College of Psychiatrists, 2001), were used as opposed to criteria developed for the intellectually typical section of the population (DSM-IV-TR and ICD-10), or clinical diagnoses. Accurate diagnoses of mental illness rely heavily upon gaining access to the patient’s covert processes such as recurrent thoughts, impulses, or mental images (Mikkelsen, Charlot, & Langa, 2005). A patient’s ability to report on covert events deteriorates with declining intellectual abilities and the clinician is often left with interpreting the individual’s observable behavior (or interpreting reports of the person’s behavior by a third party.) As a result, psychiatric diagnoses become increasingly problematic as the intellectual functioning of the individual diminishes (Rush & Frances, 2000). To review the recent epidemiological research on psychopathology in ID, we were not aiming at an exhaustive review. Instead, we primarily selected studies that (1) screened for a broad range of psychiatric disorders and behavior problems (exceptions were a few excellent recent studies on specific types of maladaptive behavior), (2) screened individuals with unspecified ID, (3) were published in the new millennium, and (4) used large national, regional, or total population sampling frames. We thus identified 23 research publications (abstracted in Table 9.1) all of which were conducted in countries with central health services registries (Australia, Canada, Holland, Norway, Sweden, England, Scotland, and Wales). Several manuscripts were separate reports based on the same comprehensive database [Cooper et al., 2009a (Study 1, Table 9.1); Cooper et al., 2007 (Study 2, Table 9.1); Cooper et al., 2009b (Study 3, Table 9.1); Cooper et al., 2007 (Study 4, Table 9.1); Smiley et al., 2007 (Study 20, Table 9.1)], while others used accumulating data sets at different points in time [Tsakanikos, Bouras, Sturmey, & Holt, 2006 (Study 21, Table 9.1); Tsakanikos et al., 2006 (Study 22, Table 9.1)]. In addition to the systematic summary of these 23 publications presented in Table 9.1, we also discussed other recent epidemiology studies, but without systematically abstracting them in a table format.

4.1. Prevalence In the context of this chapter, prevalence refers to the total number of cases (e. g., individuals with ID who also have some form of mental illness and/ or severe challenging behavior) in the reference population (e. g., the population of individuals with ID that was screened) at a given point in

260


time. It is important to distinguish between prevalence (proportion of cases in the reference population) and the proportion of cases with an additional criterion (e. g., proportion of cases in a certain age group). 4.1.1. Mental illness According to the studies presented in Table 9.1, the prevalence rate for mental illness of any kind in individuals across all ID levels, sex, and age ranged from 15.7% [Cooper et al., 2007 (Study 4, Table 9.1)] to 54.0% [Gustafsson & Sonnander, 2004 (Study 12, Table 9.1)]. Table 9.2 shows the variability of prevalence estimates of overall psychopathology as a function of assessment criteria. The estimates vary drastically, depending on the criteria and assessment methods alone. This is best illustrated by Cooper et al. (2007; Study 2, Table 9.1) with point prevalence of mental illness ranged from a high of 52.2% when clinical diagnoses were the criteria to a low of 11.4% when DSM-IV-TR criteria were applied in the screening. The prevalence rates of specific mental illness conditions also varied widely. Anxiety disorders ranged from 1.5% (Salvador-Carulla, RodriguezBlazquez, Rodriguez de Molina, & Perez-Martin, 2000) to 24.4% (Felce et al., 2009); depression from 1.5% (Emerson, 2003) to 30.0% (Holden & Gitlesen, 2003); psychotic disorders to 4.6% (Hove & Havik, 2008a) to 26.93% (Salvador-Carulla et al., 2000); and personality disorders from 4.5% (Felce et al., 2009) to 9.0% (Tsakanikos et al., 2006). 4.1.2. Maladaptive behavior Because maladaptive behavior, particularly severe aggression and selfinjury, are likely to lead to costly out-of-home placement and to have a significant impact on the individual, accurately assessing the prevalence and causes as well as effective treatment is critical to serving this population. Cooper et al. (2007; Study 4, Table 9.1) found that prevalence rates for maladaptive behavior of any kind ranged from 0.1% when DSM-IV-TR or DCR-ICD-10 criteria were used to 22.5% when clinical diagnoses were used. Again, estimates vary drastically, depending on the screening criteria and assessment methods. Table 9.3 shows the variability of overall behavior problems as a function of assessment criteria. The range of prevalence rates for specific types of behavior problem were as follows: Aggressive behavior ranged from 6.4% [Holden & Gitlesen, 2006 (Study 14, Table 9.1)] to 32% [Lowe et al., 2007 (Study 17, Table 9.1)]; self-injurious behavior ranged from 4.4% [Holden & Gitlesen, 2006 (Study 14, Table 9.1)] to 21% [Lowe et al., 2007 (Study 17, Table 9.1)]; and destructive behavior ranged from 2.3% [Holden & Gitlesen, 2006 (Study 14, Table 9.1)] to 19% [Lowe et al., 2007 (Study 17, Table 9.1)].

Table 9.2 Varying prevalence rates of overall psychopathology across studies and assessment methods

Assessment criteriaa

a

Authors

Study # in Table 9.1

Clinical diagnosis

DC-LD

DCRICD-10

DSMIV-TR

Cooper et al. (2009a) Cooper et al. (2007) Dekker and Koot (2003) Einfeld et al. (2006) Emerson (2003) Emerson and Hatton (2007) Gustafsson and Sonnander (2004) Hove and Havik (2008a) Morgan et al. (2008) Tsakanikos et al. (2006)

2

52.2

45.1

10.9

11.4

4

40.9

35.2

16.6

15.7

5

PIMRA

37

54

41

7 8

39 36

10

34.9

16

31.7

19

49

For the full name of the assessment criteria or instruments, see the legend in Table 9.1.

RSMB

38.6

6

13

DBC

39 36

Table 9.3 Varying prevalence estimates of problem behaviors across studies and assessment methods

Assessment criteriaa

a

Authors

Study # in Table 9.1

Clinical Diagnosis

DC-LD

DCR-ICD-10

DSMIV-TR

Cooper et al. (2009a) Cooper et al. (2007) Emerson et al. (2001) Holden and Gitlesen (2006) Hove and Havik (2008a) Jones et al. (2008) Lowe et al. (2007) Smiley et al. (2007)

2

39.1

33.7

0

0

4

22.5

18.7

0.1

0.1

CBS-IS

9

12.1

12

11.1

13 14 15 18

20.2 22.5

18.7

4.6

3.5

10

For the full name of the assessment criteria or instruments, see the legend in Table 9.1.

0

0


263

4.2. Incidence Incidence in this context means the number of newly emerging cases of mental illness or maladaptive behavior during a given time period in individuals with ID. 4.2.1. Mental illness To our knowledge, there are no lifelong incidence data on psychopathology in ID. For obvious reasons of cost and practicality, longitudinal incidence studies on mental illness in ID are very rare and, those that do exist cover relatively short time periods. For instance, Smiley et al. (2007; Study 20 in Table 9.1) used a prospective cohort design to measure the incidence of episodes of mental illness in adults with ID over a 2-year period in Greater Glasgow, U. K. Incidence of any type of psychopathology varied between 6.8% when DSM-IV-TR criteria were used to16.3% when clinical diagnoses were considered. It is unclear, however, whether these estimates referred to newly emerging cases, or active episodes of mental illness. Although not a true incidence study, Einfeld et al. (2006; Study 8, Table 9.1) followed a group of 578 children and adolescents with ID between 1991 and 2003 as part of the Australian Child to Adult Development Study. Data were collected at four points in time and complete data were obtained for 507 individuals. At the first measurement, point prevalence of psychopathology was 41%. Interestingly, prevalence subsequently decreased to 31% at fourth measure, despite the fact that only 10% of individuals had received any mental health treatment during that time. More specifically, of the five subscale scores of the Developmental Behavior Checklist (DBC; Einfeld & Tonge, 2002), four decreased over time (disruptive behavior, self-absorbed, communication disturbance, and anxiety). The severity of psychopathology decreased more in boys than in girls (boys started with higher baseline scores) and decreased more in individuals with mild ID compared to those with severe or profound ID. Although the prevalence of psychopathology decreased slowly over time, for most it persisted well into young adulthood. Another important discovery was that the overall seriousness of psychopathology was consistent across the range of ID. De Reuter, Dekker, Verhulst, and Koot (2007, Study 7, Table 9.1) conducted a longitudinal, multiple-birth-cohort study on a large sample of children and adolescents ages 6–18 years using a Dutch version of the Child Behavior Checklist (CBCL; Achenbach, 1991) comparing children with and without ID. Children with ID had higher CBCL scores on all subscales than typically developing children across all age groups.

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At this point, our knowledge about the incidence of psychopathology in individuals with ID is still rudimentary. Longitudinal studies such as the one described by Smiley et al. (2007) are needed, preferably studies that are conducted over longer time periods, involving young children that would allow us to learn about first onset of mental illness in ID. The findings of Einfeld et al. (2006) and de Ruiter et al. (2007) of a decrease of psychopathology in children and adolescents with ID over time are interesting but waiting to be replicated. 4.2.2. Maladaptive behavior Smiley et al. (2007; Study 20, Table 9.1) also examined the incidence of problem behaviors in adults with ID and found again a considerable variation as a function of the assessment criteria. Over a 2-year period, incidence of problem behaviors was reported as ranging from 0% (with DCR-ICD10 or DSM-IV-TR) to 3.5% (according to DC-LD criteria), and finally 4.6% (when determined by clinical diagnosis). The study by de Reuter et al. (2007, Study 7, Table 9.1) is also relevant for the issue of maladaptive behavior. While children with ID had higher overall CBCL scores than the control children, the CBCL score differences decreased over time, most noticeably in social problems and aggression. Again, more longitudinal research, especially starting at a very early age, will not only help to identify the process of early development of maladaptive behavior (e. g., Richman & Lindauer, 2005; Symons, Sperry, Dropik, & Bodfish, 2005) but also guide early intervention to prevent the development of maladaptive behavior (Richman, 2008).

5. Risk Factors and Correlates In any field of human health and well-being, a main interest is to identify risk factors that may contribute to later pathology. However, there is often confusion about the correct terminology of related terms “risk factors,” “correlates,” and “risk markers” (Witwer & Lecavalier, 2008). Kazdin, Kraemer, Kessler, Kupfer, and Offord (1997) defined a risk factor as a precursor event that is associated with a higher probability (risk) of a certain outcome over the probability of that outcome in individuals who were not exposed to that event. Risk factors have a causal relationship with an outcome. Identifying a risk factor is often accomplished first by recognizing a correlational relationship of an event with the outcome (correlate) when measured at the same point in time. Evidence that an event is not only correlated with the outcome but is causing it (i. e., that it is a risk factor) has to be established by longitudinal studies which demonstrate that the event actually predicted the later outcome. The literature on


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psychopathology in ID has only a few established risk factors in that stringent sense. Risk markers, on the other hand, are biological or neuropsychological traits that indicate a genetic tendency toward developing an outcome (Compton, 2005). Risk markers will be discussed in the section on behavioral phenotypes. Even though prevalence rates for psychiatric and problem behaviors are not consistent across studies, the general impression is that individuals with ID show a higher rate of psychiatric and behavior disorders than the general population [e. g., Emerson, 2003 (Study 9, Table 9.1)]. There are multiple possible biological, psychological, developmental, emotional, social, and environmental factors that may contribute to this increased prevalence, either uniquely or in interaction with other factors. Harris (2006) proposed that it is the combined and interactive nature of a number of risk factors and markers that contribute to psychopathology in ID, including neurobiological causes of both psychopathology and ID; cognitive impairments leading to fewer adaptive coping responses; poor academic performance that contributes to low self-esteem; unrewarding peer relationships; the amount of stress that the care of an individual with ID can put on the family; increased abuse and exploitation; and the lack of appropriate diagnosis and treatment. More rarely, there may also be a negative circular relationship among severe psychosocial deprivation, poor nutrition, and lack of stimulation leading to further cognitive, social, and psychological impairment. This notion is also reflected in the generalized model of dynamic influences on individual health, adaptation and development as shown in Fig. 9.3. Although we realize the complex interactive nature of risk factors and risk markers and the difficulty of isolating the impact of single risk factors, we will discuss one by one the available evidence for some salient factors such as level of functioning, age, and sex.

5.1. Levels of intellectual disability Cognitive functioning as a potential risk factor of psychopathology is highly associated with language development, communication, coping strategies with stress, and other adaptive behavior skills and other critical developmental variables. Although some have deemphasized the importance of ID levels as a dimension of classification (Luckasson et al., 2002), it is one of the most robust predictors of developmental outcome in the population with ID. 5.1.1. Mental illness With regard to mental illness in general, there is considerable inconsistency in the literature about the effects of the ID functioning level. One group of studies showed that cognitively higher functioning individuals with ID

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were more likely to have a mental illness diagnosis than lower functioning individuals [for instance, Morgan, Leonard, Bourke, & Jablensky, 2008 (Study 18, Table 9.1); Myrbakk & von Tetzchner, 2008b (Study 19, Table 9.1); Rojahn & Esbensen, 2005]. Koskentausta, Livanainen, and Almqvist (2007), on the other hand, found a curvilinear relationship between the presence of mental illness and level of functioning: the risk of psychopathology was higher in children with moderate ID as compared to mild or profound ID. Probably the most convincing series of epidemiologic studies in this field has been conducted by Cooper and her colleagues. They found no evidence that level of ID in adults was related to mental illness (when autism and behavior problems were excluded). The group with mild ID had the same prevalence of mental illness of 22.4% as the group with moderate to profound ID [Cooper et al., 2007 (Study 4, Table 9.1)] (see also Fig. 9.1). Obviously, more research needs to be conducted to clarify the relationship between level of functioning and mental illness in general as well specific disorders. One important concern is, as we had mentioned, that the reliability of psychiatric diagnoses decreases with declining levels of function (Rush & Frances, 2000). It may also be that mental illness in the lower functioning portion is manifested mostly by atypical symptoms (i. e., behavioral equivalents). Since the concept of behavioral equivalents is still conceptually vague and lacks strong empirical support, many cases of mental illness may still be overlooked in the lower function group of individuals with ID. 30 25 20 15

Mild ID (n = 389)

10

Moderate to profound ID (n = 625)

5 0 Problem behavior

Mental illness (excluding behavior problems and autism)

Figure 9.1 Prevalence of problem behaviors and clinical diagnoses of mental illness as function of level of intellectual disabiliy (figure based on data presented by Cooper et al., 2007).


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5.1.2. Maladaptive behavior While the evidence is still patchy as to whether and to what extent the intellectual functioning level may present a risk factor for mental illness, the picture is clearer with regard to behavior problems (Rojahn & Tasse´, 1996; Witwer & Lecavalier, 2008). A host of studies show that level of ID is strongly correlated with the prevalence of challenging behaviors in general [e. g., Cooper et al., 2009a (Study 1, Table 9.1); Cooper et al., 2007 (Study 4, Table 9.1); Holden & Gitlesen, 2003 (Study 13, Table 9.1); Jones et al., 2008 (Study 16, Table 9.1)]. Figure 9.1 shows the differential relationship between level of functioning and the prevalence of mental illness versus the prevalence of maladaptive behavior that was discovered using the same general methodology by Cooper et al. (2007; Study 4, Table 9.1). There is also some evidence that the relationship between level of functioning and problem-behavior prevalence may differ depending upon the type of behavior problem. For instance, Emerson et al. (2001) and Cooper et al. (2009b, Study 3, Table 9.1) found that outwardly directed aggression was more likely to be shown by individuals with less severe disabilities. Holden and Gitlesen (2003; Study 13, Table 9.1) also found that aggression against others and temper tantrums was more common among those with mild to moderate disability compared to those with more severe disability while self-injurious behavior was more common in those with profound ID. Yet these findings are offset by the results presented by Crocker et al. (2006; Study 5, Table 9.1) and Tyrer et al. (2006; Study 23, Table 9.1) that individuals with severe/profound ID were more likely to engage in aggression than those with mild/moderate ID. Figure 9.2 shows the prevalence of different types of severe problem behavior by level of functioning in a population of 135,282 individuals (ages 0–45 years of age) who received developmental disabilities services from the states of New York and California (described in Rojahn, Borthwick-Duffy, & Jacobson, 1993). It illustrates that prevalence of maladaptive behavior in general was positively correlated with level of functioning, but that aggressive behavior, relative to self-injury for instance, was more prevalent in the cohort with mild ID, and less prevalent in those with profound ID.

5.2. Chronological age The next question we want to address is to what extent chronological age might be a potential risk factor in ID. 5.2.1. Mental illness While there have been studies published on the prevalence of mental illness in ID at different age levels, we have not found a study that compared

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30 25 20 Aggression 15

Destruction SIB

10 Stereotypy 5 0 Mild

Moderate

Severe

Profound

Figure 9.2 The prevalence of different types of problem behaviors by level of functioning (n = 135,282; from state agency administrative data sets from New York and California described in Rojahn et al., 1993).

prevalence risks across the lifespan. A study by Dykens, Shah, Sagun, and King (2002) did examine changes in mental health problem behaviors in a group of 211 children and adolescents with Down syndrome. They found that of four age groups (4–6, 7–9, 10–13, 14–19) internalizing behaviors, and specifically withdrawn behaviors, were significantly higher in the age groups 10–13 and 14–19 years compared to younger children. In the 14–19 age group, age and gender interacted significantly such that females more than males scored significantly higher in this age group. Externalizing behaviors were more prevalent in the 10- to 13-year-old age group. Age was not found to be a predictor of mental illness prevalence in adults [Cooper et al. 2007 (Study 2, Table 9.1)] or in children [Dekker & Koot, 2003 (Study 6, Table 9.1)]. In an institutional sample of ID adults 18 years and older, the highest prevalence of mental illness was found in the broad age span of 51–94 years, with 77% of those identified with mental illness coming from this age group. Moss and Patel (1993) found that prevalence of mental disorders was 11.4% in people age 50 years and up with moderate to profound ID. Two studies were found that included age as a factor for prevalence of specific psychiatric disorders in children. In one study [Emerson, 2003 (Study 9, Table 9.1)] children under the age of 11 years were more likely to be diagnosed with a pervasive developmental disorder, whereas children age 11 and over were more likely to be diagnosed with depression. In another study [Emerson & Hatton, 2007 (Study 10, Table 9.1)], children between the ages


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of 5 and 10 years were more likely to be diagnosed with “hyperkinesis” compared to children age 11–16 years, who were more likely to be diagnosed with an emotional disorder. De Reuter et al. (2007) found that while children with ID demonstrated a greater risk for psychopathology on a parent rating scale compared to children without ID across all ages, the developmental course between ages 6 and 18 was similar. Interestingly, attention problems decreased more sharply in children with ID, and in males, social problems decreased more sharply in children with ID. 5.2.2. Maladaptive behavior Emerson et al. (2001; Study 11, Table 9.1) studied the age distribution of maladaptive behavior in two subsequent surveys. In their 1988 survey, the majority (64%) of the individuals with challenging behavior were between the ages of 12 and 35 and in the 1995 sample 60% were in that age bracket. In the 1995 survey sample, prevalence increased up until and then peaked in the mid-teens, while it increased until the mid-twenties (1988 sample) before leveling off. Lowe et al. (2007; Study 17, Table 9.1) found that the greatest proportion of individuals with severe maladaptive behavior were young adults between 16 and 39 years (40%), followed by those between 40 and 59 (34%) years, 17% were children younger than 16 years, 9% were 60 years or older. Compared to Emerson et al. (2001), however, Lowe et al. (2007) found the peak prevalence of general maladaptive behavior to occur at a somewhat older age (between the ages of 30 and 60 years). Holden and Gitlesen (2006; Study 14, Table 9.1) distinguished between more or less severe problem behaviors and found an interaction effect between age and behavior problem severity: Demanding challenging behavior increased between the ages of 10 and 20 years and was highest between the ages of 20 and 40 years. After that the prevalence of demanding maladaptive behavior decreased. Less demanding maladaptive behavior, however, was most common in individuals under 20 years, with little change after that. Maladaptive behavior of any severity was least common in individuals over 60 years of age. de Ruiter et al. (2007) found that aggressive behavior in boys with ID decreased sharply between ages 6 and 18 years. Chronological age clearly is related to the prevalence of general maladaptive behavior, but like with most correlates or risk factors the available data vary. One constant finding is that prevalence of challenging behavior increase until early or mid adulthood, and slowly decreases into old age—a trend that has been confirmed by several earlier studies that are not presented in Table 9.1, such as Borthwick (1994), Oliver, Murphy, and Corbett (1987), Rojahn et al. (1993), and Saloviita, 2000).

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5.3. Sex 5.3.1. Mental illness Sex as a potential risk marker has been examined in some studies. Cooper et al. (2007; Study 4, Table 9.1) reported that the prevalence rates of mental illness of any type based on clinical diagnosis (excluding maladaptive behavior) were 29.2% for men and 27.3% for women. Tsakanikos, Bouras, Sturmey, and Holt (2006; Study 21, Table 9.1) found sex differences in psychiatric comorbidity in adults with mild to severe ID, both in terms of specific diagnoses and in patterns of referral for specialized mental health services. Based on ICD-10 criteria, personality disorder was the most common diagnosis among men, whereas dementia and adjustment reaction were most common among women. No statistically significant sex differences were found in depression, anxiety, or schizophrenia. Lunsky (2003) interviewed 51 men and 48 women with borderline intellectual functioning through moderate ID and found that women reported significantly higher levels of depression than men on the adapted Birleson Depression Scale (BDS; Birleson, 1981), which is consistent with the trend in the general population. However, when depression was assessed by a rater using the Reiss Screen for Maladaptive Behavior (RSMB; Reiss, 1988) and the informant version of the BDS, the difference between men and women was not significant. Informant ratings were significantly lower than self ratings for men and for women. The difference between self and other report scores may suggest that observers were less able to accurately assess internal symptoms of depression than are the individuals themselves, even in relatively high functioning individuals. Witwer and Lecavalier (2008) concluded from their literature review that adolescent girls had more internalizing symptoms than males, but these sex differences were not seen in younger children. As for Attention-Deficit/ Hyperactivity Disorder (ADHD), males tend have higher scores than females on behavior rating scales. In other words, most of the research that examined the effects of sex on the prevalence of mental illness in ID seems to be consistent with the relationship between sex and mental illness in the general population. 5.3.2. Maladaptive behaviors McClintock, Hall, and Oliver (2003) summed up their meta-analysis by concluding that men were more likely to show aggression than females. Likewise, based on their literature review, Witwer and Lecavalier (2008) surmised that boys and male adolescents were more likely to show disruptive, antisocial and problem behaviors than their female counterparts. Male predominance in challenging behavior in general was also found by Lowe et al. (2007; Study 17, Table 9.1), while Tyrer et al. (2006; Study 23,


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Table 9.1) found that men were more likely to engage in physical aggression than women. Crocker et al. (2006; Study 5, Table 9.1) found sex differences in adults with mild to profound ID for certain types of aggression, but no sex differences in verbal and physical aggression. Men were found to be more likely to show property destruction and sexual aggression. However, other credible studies found opposite sex effects on maladaptive behavior. For instance, Cooper et al. (2009a; Study 1, Table 9.1) and Cooper et al. (2009b; Study 3, Table 9.1) reported that being female predicted aggressive and self-injurious behavior. Jones et al. (2008; Study 16, Table 9.1) also found that being female predicted problem behaviors. Hemmings et al. (2006) challenged the notion of sex contributing to the prediction of any challenging behaviors altogether (aggression, destruction, self-injury, tantrums, etc.). In other words, the question of whether sex is a risk marker for maladaptive behavior has no clear answer as of yet.

5.4. Comorbidity with autism spectrum disorder Autism spectrum disorders (ASDs) are a group of complex developmental disabilities characterized by problems with social interaction and communication, as well as by routines or repetitive behaviors. 5.4.1. Mental illness An earlier literature review suggested that ASD was correlated with an increased risk for psychopathology (Witwer & Lecavalier, 2008) and more recent studies are still consistent with that summary. For instance, inpatients with ASD and an ID were found to have more mood, anxiety, and other related symptoms than inpatients with ID but without ASD (Charlot et al., 2008). Higher rates of psychiatric symptoms in individuals with ID combined with ASD were found in other studies as well (Hill & Furniss, 2006; LaMalfa et al., 2007). Hill and Furniss (2006) and LaMalfa et al. (2007) found that individuals with pervasive developmental disorder/autism had higher scores on subscales for organic disorder, anxiety, and mania of the Diagnostic Assessment for the Severely Handicapped-II (DASH-II; Matson, 1995). The more severe the autistic symptoms, the higher the DASH-II subscale scores were. Tsakanikos et al. (2006; Study 22, Table 9.1), however, found no evidence that adults with ASD had an increased risk for psychopathology. 5.4.2. Maladaptive behaviors As stated above, repetitive actions are core symptoms of ASD and are also referred to as stereotyped behavior, which can also include self-injurious

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behavior. Therefore, since stereotypies and self-injury are by definition associated with ASD we would expect higher levels of those types of in individuals with a diagnosis of ASD and ID as compared to individuals with ID without autism. Several studies have been conducted to investigate whether individuals with ASD do indeed have higher levels of maladaptive behavior than those without. Bodfish, Symons, Parker, and Lewis (2000) compared matched groups of adults with ID with or without autism. Although the difference was not statistically significant, there was a trend for the group of individuals with ASD to have a higher proportion of selfinjurious behavior cases (45%) compared to the non-ASD group (25%). In 2006, Hill and Furniss found that children and young adults with severe ID and with ASD had higher scores on the DASH-II subscales for stereotypies than the comparison group without ASD. Felce et al. (2009, p. 249) also found that “the triad of social impairment characteristic of autism spectrum disorder” contributed independently to the prediction of maladaptive behavior, and Matson, Wilkins, and Macken (2009) reported that the severity of ASD was positively related to the number and the intensity of challenging behaviors. Those with severe ASD exhibited significantly higher levels of problem behaviors (including self-injury, stereotypies, aggression, and destruction) than children with mild or moderate ASD. Matson, Ancona, and Wilkins (2008) also identified increased sleep disturbance in individuals with both ID and ASD as compared to ID alone and Ashworth, Martin, and Hirdes (2008) found that individuals with ASD had a higher rate of pica than those with ID alone. In contrast, no relationships between self-injurious behavior and ASD were found by Cooper et al. (2009a; Study 1, Table 9.1) nor between aggressive behavior and ASD by Tyrer et al. (2006; Study 23, Table 9.1). Lowe et al. (2007; Study 17, Table 9.1) did not find relationships between any problem behaviors and ASD.

5.5. Genetic disorders—behavioral phenotypes The concept of the behavioral phenotype was introduced by Nyhan (1972; cf. Harris, 2006) in his presidential address to the Society for Pediatric Research and refers to the observation that specific chromosomal, genetic, or neurodevelopmental disorders may be associated with particular patterns of behavior. 5.5.1. Mental illness Chromosomal, genetic, or neurodevelopmental disorders that are associated with ID can also be risk markers for specific psychiatric disorders, or phenotypic mental illness. For instance, persons with Down syndrome


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are at higher risk of Alzheimer’s dementia and depression in later life (Dykens, 2007). Many individuals with fragile X syndrome meet criteria for anxiety disorders and in particular social anxiety or shyness (Dykens & Hodapp, 1997; Sullivan, Hooper, & Hatton, 2007). Signs of depression are often found in women with fragile X (Freund, Reiss, Hagerman, & Vinogradov, 1992; Sobesky, Hull, & Hagerman, 1994). Adult females with fragile X also show social deficits including shyness, social anxiety, and withdrawal and may meet criteria for schizotypal and avoidant personality disorders. Prader–Willi syndrome is associated with high rates of affective disorders with psychotic features (Beardsmore, Dorman, Cooper, & Webb, 1998). Clarke (1998) estimated the prevalence of psychotic disorders to be about 6.3% in a population of adults with Prader–Willi syndrome. Soni et al. (2007) found that individuals with Prader–Willi syndrome had psychiatric illness that most closely resembled an affective disorder, Specifically, those with the maternal uniparental disomy genetic subtype had a more severe course of illness than those with the deletion genetic subtype in terms of risk of recurrence of an affective disorder, more episodes, and higher prevalence. Individuals with Williams syndrome are described as quite sociable and interpersonally sensitive, suggesting a vulnerability to the experience of loss and sadness. Anxiety, hyperactivity, and inattentiveness are common in Williams syndrome, including generalized anxiety or worry about future events (Dykens & Hodapp, 1997). Einfeld, Tonge, and Florio (1997) found that individuals with Williams syndrome were more likely to be diagnosed with a psychiatric disorder when compared to a group individuals matched on age, sex, and level of ID. Increased rates of anxiety, hyperactivity, preoccupations, and inappropriate interpersonal relating were found as well as sleep disturbance and hyperacusis. Furthermore, individuals with ID also have a higher prevalence of medical concerns that may have the same cause as the ID, such as heart conditions in Down syndrome (Gabriel, Loschen, Reeve, Sanderson, & Charlot, 2007). Because individuals with ID are often poor reporters of their own health concerns, physical discomfort might contribute to disruptive behaviors or development of depression or anxiety (Gabriel et al., 2007). 5.5.2. Maladaptive behaviors There are several genetic conditions that are associated with severe behavior problems. For instance, individuals with Lesch–Nyhan syndrome all show ferocious self-biting and other self-injurious and aggressive behaviors (Nyhan, 2002). Other genetic or chromosomal syndromes that are known to be associated with behavior problems include Cornelia de Lange syndrome

274


(Hyman et al., 2002; Moss et al., 2005; Oliver et al., 2009; Sarimski,1997), Smith Magenis syndrome (Finucane, Dirrigl, & Simon, 2001; Dykens, Hodapp, & Finucane, 2000; Dykens & Smith, 1998; Taylor & Oliver, 2008), Rett syndrome (Mount, Hastings, Reilly, Cass, & Charman, 2003; Sansom, Krishnan, Corbett, & Kerr, 1993), Prader–Willi syndrome (Einfeld, 2005; Oliver, Woodcock, & Humphreys, 2009; Reddy & Pfeiffer, 2007; Stein, Keating, & Zar, 1993) and fragile X syndrome (Einfeld, 2005; Einfeld, Tonge, Turner, Parmenter, & Smith, 1999; Hagerman, 1990; Hagerman & Silverman, 1991). Behavior problems that are linked to a specific genetic cause may be less responsive to general behavior modification strategies (Einfeld, 2005). The most prominent feature of Prader–Willi syndrome is overeating, which is very common in older children and adults (Dykens & Hodapp, 1997). Infants may show failure to thrive. The onset of overeating or hyperphagia is associated with an increase in other behavior problems such as non-food-related repetitive, compulsive, and ritual behaviors, including skin picking and hoarding, impulsivity, and low activity levels. Obsessive– compulsive symptoms are common as are other non-food-related repetitive, compulsive, and ritual behaviors such as skin picking and hoarding (Beardsmore et al., 1998; Dykens, Leckman, & Cassidy, 1996; Dykens & Shah, 2003). Sleep disorders are also common in Prader–Willi syndrome, which are generally related to obesity and sleep apnea. In a comparison of severity of behavior problems in four common genetic disorders associated with ID, Einfeld et al. (1999) found that individuals with Prader–Willi syndrome and Williams syndrome had higher rates of behavior problems compared to individuals with Down or fragile X syndromes.

5.6. Social–environmental factors This section briefly summarizes the available data on the potential influence of social–environmental disadvantage such as poverty, single-parent family, poor family functioning, maternal poor mental health, and negative life events (Emerson & Hatton, 2007) on psychopathology in ID. 5.6.1. Mental illness In a risk factor study with children and adolescents aged 6–18 years with ID in the Netherlands, Wallander, Dekker, and Koot (2006) found that family dysfunction, parental mental health, and child physical health have been shown to predict newly emerging psychopathology. Emerson and Hatton (2007; Study 10, Table 9.1) looked for associations between three psychiatric disorders (conduct disorder, emotional/anxiety disorder, and hyperkinesis) and sex, age, and social/environmental factors. Social/environmental risk factors included poverty, single-parent family, poor family functioning, maternal poor mental health, and negative life events. Cumulative exposure


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to social disadvantage was related to increased risk of all three types of disorders for both children with ID and typically developing children; children with ID had a higher exposure rate to the social/environmental risk factors than did typically developing children. They also found that if they statistically controlled for the increased exposure to all forms of social disadvantage in children with ID, the increased risk of psychiatric disorders among these children decreased significantly. Socioenvironmental risk factors for psychiatric disturbance in children with ID were also investigated by Koskentausta et al. (2007) on a sample of 75 children in Finland with ID between the ages of 6 and 13 years. They found that the risk of psychopathology was increased in children of low socioeconomic status and children in single-parent homes. Other factors were associated with psychopathology was moderate ID (compared to mild or profound), limitations in adaptive behavior, impairments in language development, and poor socialization. Another family factor that can contribute to mental illness (depression) is maternal depression (Esbensen, Mailick Seltzer, & Greenberg, 2006). 5.6.2. Maladaptive behaviors Prevalence of problem behaviors in general [Jones et al., 2008 (Study 16, Table 9.1)] are related to out-of-home care, as is self-injury [Cooper et al., 2009a (Study 1, Table 9.1)] and aggression [Cooper et al., 2009b (Study 3, Table 9.1); Crocker et al., 2006 (Study 5, Table 9.1)]. Tyrer et al. (2006; Study 23, Table 9.1) found that aggression was less prevalent in individuals who lived independently than those who did not.

5.7. Psychological coping skills As mentioned earlier, Harris (2006) speculated that one obvious reason why the ID population may be especially vulnerable to psychopathology are cognitive impairments that result in a lack of adaptive coping responses and are likely to increase perceived stress. Coping skills is likely to be a concept highly related to social adaptive behavior. 5.7.1. Mental illness In the only study of its kind to date, Hartley and MacLean (2008) found a significant negative correlation between the use of coping efforts and psychological distress in individuals with ID (without ASD). A significant negative correlation was discovered between active coping strategies and psychological distress, while a trend toward a positive correlation was observed between distress and avoidant coping. These findings support the idea that inadequate coping by individuals with ID may be related to increased symptoms of anxiety and depression, and that increasing active

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coping efforts in response to stress may help to decrease vulnerability to anxiety and depression. Active coping was defined as the effort to gain control over a stressful situation or control over one’s own emotions, and actively using other people as resources to seek solutions or manage affect. Avoidant coping was defined as avoiding or disengaging from a stressful situation (behavioral avoidance coping) or wishing that the stressful situation did not happen or avoiding thinking about it (cognitive avoidance coping). In an earlier study, adults with mild ID reportedly engaged in more avoidant coping, which was related to depression and anxiety (Hartley & MacLean, 2005). More research is needed in the area of coping skills as a preventive strategy and about ways to enhance those skills. 5.7.2. Maladaptive behaviors No study was identified that examined the relationship between behavior problems and coping skills per se. However, coping skills are likely related to social adaptation, communication, and intellectual functioning. The relationship between level of functioning and behavior problems has been discussed earlier in this chapter.

6. Conclusions Although it had long been ignored, it is now generally accepted that individuals with ID can and do experience mental illnesses such as anxiety, depression, and schizophrenia. Indeed, we now know that the population with ID is particularly vulnerable to mental illness and behavior disorders. Having said that, however, due to a plethora of methodological difficulties in epidemiological research, we are still far from having a firm grip on how much more vulnerable individuals with ID are than the population at large. At a minimum, only studies that directly compared an ID population with a typical population using the same research methodology can make such comparative statements. We found only three recent studies that met this criterion [de Ruiter et al., 2007 (Study 7, Table 9.1), Emerson, 2003 (Study 9, Table 9.1), Emerson & Hatton, 2007 (Study 10, Table 9.1)]. All three of them focused on children and adolescents (from 5 to 18 years). Emerson (2003) and Emerson and Hatton (2007) showed that the prevalence of significant psychopathology in children with ID was 4.8 and 4.5 times as high as in the comparison group without ID (39 vs. 8.1% and 36 vs. 8%, respectively). Psychopathology caseness in both studies was based on the DAWBA (Goodman et al., 2000), which is based on DSM-IV and ICD-10 criteria. This is noteworthy in so far as studies that used multiple assessment and screening criteria indicated that prevalence estimates of mental illness that were based on DSM-IV and ICD-10 criteria were consistently lower


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than clinical diagnoses or screenings based on specially ID-adapted diagnostic criteria such as the Diagnostic Criteria for Psychiatric Disorders for use with Adults with Learning Disabilities/Mental Retardation (Royal College of Psychiatrists, 2001). Prevalence rates of global mental illness across all ID levels, sex, and age in the studies summarized in Table 9.1 ranged from approximately 16 to 54%. A coarse and very general comparison benchmark may be the prevalence of active psychiatric disorders of any kind in the general U. S. population that was found to be 20% (life time prevalence of 32%) according to the Epidemiologic Catchment Area Study (Robins, Locke, & Regier, 1991). As with mental illness, prevalence estimates for maladaptive behavior varied considerably as a function of assessment criteria. Prevalence rates from studies listed in Table 9.1 for global maladaptive behavior ranged from 0.1 to 23%. For specific types of maladaptive behavior, the range for aggressive behavior was 6–32%, for self-injurious behavior 4–21%, and for destructive behavior from 2 to 19%. Obstacles for obtaining reliable estimates of maladaptive behavior prevalence include the lack of standardized categorical definitions and cutoff criteria in terms of behavior problem severity. No direct comparisons of the prevalence of maladaptive behavior in individuals with ID and in those without ID have been found. Given the wider ranges of psychopathology prevalence estimates, it is not surprisingly that the information on potential risk factors and markers is also quite erratic. In summarizing the main findings, arguably, the methodologically strongest studies found no relationship between cognitive functioning level and mental illness. The prevalence of maladaptive behavior, on the other hand, was strongly negatively correlated with ID level. This relationship may differ depending upon the type of behavior problem: for instance, aggressive behavior may be less negatively correlated with level of functioning than self-injury and stereotyped behavior. Chronological age was not found to be a predictor of mental illness in adults or in children. On the other hand, age clearly is related to the prevalence of general maladaptive behavior. But like with most other correlates or risk factors in this population, the available data vary widely across studies. One constant trend is that the prevalence of challenging behavior increases from childhood to early or mid-adulthood, and then slowly decreases as people age. Sex does not seem a strong predictor of mental illness in general. Some relationship was found between sex and specific mental disorders. For instance, personality disorders seem more common in men, while dementia and adjustment disorders might be more prevalent in women. No sex differences were reported in anxiety or schizophrenia and some contradicting information has been found for depression. Among adolescents

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girls, internalizing behaviors are more prevalent than in males. Whether sex is a risk marker for maladaptive behavior is not clear as of yet. Male predominance in aggressive and destructive behavior seems likely, but the available results are conflicting. It appears as if ASD is a risk factor for other forms of mental illness. Some studies suggested that individuals with ID and ASD were more prone to mood disorder, anxiety, and other related symptoms than those without ASD, while other studies did not. As far as maladaptive behavior is concerned, one would expect ASD to be a risk factor given that stereotyped behavior is symptomatic for ASD. Indeed, some studies suggested that the severity of ASD was positively correlated to the number and the intensity of such challenging behaviors as self-injury, stereotypies, aggression, and destruction, and that individuals with ASD exhibited significantly higher levels of problem behaviors. However, other credible studies did not find such a relationship. There is also growing evidence that certain forms of mental illness may be phenotypic for certain genetic or chromosomal conditions. Fragile X syndrome, for instance, seems related to social anxiety, while Prader–Willi syndrome is associated with affective disorders with psychotic features. Anxiety, hyperactivity, and inattentiveness are also common in Williams syndrome. Research that links specific genetic markers, such as the repetition of a specific nucleotide sequence in fragile X or the microdeletion or mutation of Smith–Magenis to certain types of mental illness will add not only to our understanding of the incidence of psychiatric disorders in individuals with ID, but also in the general population. Evidence also points to the fact that there are probably interactions between chromosomal conditions and sex, especially in X-linked chromosomal conditions. For example, depression was often found in women with fragile X. Phenotypes of maladaptive behavior have been known for some time such as in Lesch– Nyhan syndrome, Cornelia de Lange syndrome, Smith–Magenis syndrome, Rett syndrome, and Prader–Willi syndrome. Children with ID, especially in the mild and moderate ID range, have a greater exposure rate to the social/environmental risk factors than typically developing children. Some evidence suggests that social disadvantage also increased their risk of psychiatric disorders. Inadequate coping may be related to increased risk of anxiety and depression. Although risk factors and markers were discussed in this chapter one by one, we do not imply that mental illness or maladaptive behavior can be best predicted by single factors. Rather, vulnerability for psychopathology is most likely a function of complex interactions of mostly poorly understood factors. Such factors may include neurobiological substrates, behavioral characteristics, and socioeconomic conditions (Harris, 2006; Sturmey, Lindsay, & Didden, 2007). Figure 9.3 presents a hypothetical biosocial–behavioral path model that depicts the multivariate and

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Prenatal

Postnatal

Resource variables

Personal and psychosocial resources, family function, supports

Cognitive functioning, environmental stimulation, life events, stress

Proximal Variables Catalytic variable

Distal Predisposing Variables

correlational link possible causal link causal link

Material resources

SES, demographics, maternal mental health

Outcome (psychosocial adaptation, mental health)

Genetic, chromosomal, biological (teratogens)

Figure 9.3 Generalized model of dynamic influences on individual health, adaptation and development (adapted from Baumeister, 1988).

interdependent nature of prenatal and postnatal influences that contribute to a person’s psychosocial adaptation in general and to mental health or illness in particular (adapted from the “new morbidity” model by Baumeister, 1988). Genetic endowment, behavioral, and mental health characteristics of the mother and socioeconomic status tend to determine material resources, which in turn influence access to support services. These factors also influence the quality of a nurturing and safe environment. In reviewing the literature, many of the methodological difficulties inherent in accurately determining the prevalence rate of psychopathology in individuals with ID became apparent. Nineteen different diagnostic and screening instruments were identified in a review of 23 research publications on prevalence of psychopathology in individuals with ID. The spread of prevalence rates for specific syndromes even within a single study had an odds ratio of 1/4.8 [10.9–52.2%; Cooper et al., 2007 (Study 2, Table 9.1)] depending upon whether the diagnosis was by clinical opinion or strictly based on ICD-10 or DSM-IV-TR criteria. Use of classification systems developed for individuals with normal intellectual functioning, such as the ICD-10 or the DSM-IV-TR, may lead to significant under diagnosis of mental illness in this population. Supplemental systems such as the DM-ID (NADD, 2007) and the DC-LD (Royal College of Psychiatrists, 2001) may

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help to improve the diagnostic accuracy in individuals with below average intellectual functioning (Mikkelsen et al., 2005). Whether an entirely new system for identifying and labeling psychopathology in individuals with ID is developed, or an existing system such as the DM-ID or DC-LD is used, it is important that researchers and practitioners to reach consensus on which instruments and which classification system identifies and best meets the needs of this population. Some of the problems in diagnosing psychiatric disorders in individuals with ID are the same as diagnosing psychiatric disorders in the general population, including the controversy around validity of psychiatric diagnoses, usefulness of diagnostic classification systems, and where the line between “normal” and “abnormal” functioning (Widiger & Coker, 2003). Progress in assessing, diagnosing, understanding, and treating emotional and behavioral disorders in individuals with ID continues to be slow but steady. It is hoped that those of us who work with this vulnerable population will continue to address the multiple obstacles that create barriers for identifying, understanding, treating, and preventing the threat of mental illness.

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INDEX

A ABA see Applied behavior analysis ABILITIES Index, 102 ACC see Agenesis of corpus callosum Active coping, 275 Adolescents: emerging concept, 4 with intellectual disabilities, 42–3 transition to adulthood, 4 developmental task, 14 employment pattern, 14 families’ reaction to, 19–20 friendships and intimacy with people outside of family, 14 high school exit and, 10–11 individual differences in families, 22 individuals with ASD experience, 11 markers of, 12–13 multidimensional evaluation of, 15–16 objective indices prediction, 17–18 other intellectual and developmental disabilities, 16–17 residential independence, 11, 14 school, family and outcome research, integration, 27–8 secure attachment bond during infancy and early childhood, 9 stressors and worries, 20–2 subjective indices prediction, 18 unsuccessful, 26 work activities, 14 Agenesis of corpus callosum, 173, 187–9 associated abnormalities and syndromes, 176 cognitive characteristics, 183–4 diagnosis of, 174–5 emotional and behavioral problems in, 181 and emotional state, 180–1 future research, directions for, 189 on adult living, 190 effectiveness of therapies and early intervention, 189 focus on isolated ACC, 189–90 language development, delay in, 181–2

physical/health-related issues in, 179 daily living issues, 178 developmental delays, 178–9 physical and structural abnormalities, 176–7 seizure disorders in children, 177 sleep problems, 178 prevalence of, 175 risk factors for, 174–5 social communication, deficits in, 182–3 social traits of individuals, 180–1 themes to consider in, 185 delays evident only at later ages, 186–7 nonverbal learning disabilities, 186 physical delays, 186 receptive and expressive language, 186 therapeutic interventions for, 185–7 Agent Orange and injuries from war, 98 American Psychiatric Association, 241 Anosognosia, 184 Applied behavior analysis, 215 Attachment theory: autonomy seeking, 8–9 parent–child relationships, 7 storm and stress, 9 type of attachment relationships, 7–8 young infants and primary attachment, bond between, 8 Augmentative and alternative communication (AAC) technique, 207 Australian Child to Adult Development Study, 263 Autism intervention research, 195–234 Early Childhood Partial Hospitalization Program, 213 reviews of, 197 comprehensive treatment programs, 198–9, 210–11 informed eclectic approach, 210, 212 nonresponders, investigation of, 211–12 practice to research, 212–14 research to practice, 212 targeted interventions see Targeted treatments Autonomy seeking, 8–9 Avoidant coping, 275

289

290

B Bandura’s Social Learning Theory, 207 BDS. see Birleson Depression Scale Behavior problem: behavior functional analysis, 215 criteria given by Royal College of Psychiatrists, 242 Birleson Depression Scale, 270 Brain: function of corpus callosum, 172 structural development in DS, 155–6 Broad-based health knowledge and health care, 39 Buddhist beliefs and disabilities, 97–8

C Canada: Canadian sample study, 14 intellectual disabilities studies, 39 Categories Test, 184 CBCL. see Child Behavior Checklist CHAP. see Comprehensive Health Assessment Program Child Behavior Checklist, 263 Child-directed support, 113 Children with ASD: friendships and intimacy with people outside of family, 14 IQ scores and early language skills, 17 mothers and fathers, similarities and differences between, 108 relational bond with caregiver, 9 meta-analysis study, 10 seizure disorders in, 177 transition planning within schools, 18 China: individuals with disabilities in: Autism Diagnostic Interview-Revised (ADIR), 72 categories and percentages, 71 Chinese Classification of Mental Disorders, 72 concept of family and, 77–8 current statistics on, 70–3 disclosure of, 79 family isolation, 79 fear of stigma, 78–9 heightened stress and limited support, 81 herbal medicine and acupuncture, 79 households, 71 intellectual disability, 72 learning in regular classrooms, 74

Index

legislation and, 73–4 literature on, 80 mother–child relationship, 86 parental roles in child’s education, 81–2 parents and children, interdependence between, 77 people’s attitudes for, 78 percentages of enrollments of children with, 76 percentages of unenrolled school-aged children with, 77 relationship-focused coping caregivers, 86 Second China National Sample Survey on Disability, 70 shame and guilt, 79 siblings of, 87–8 special education services for, 74 special schools, establishment of, 82 tests, 72 unemployment rate for parents of children with, 80 Collectivism, 127 Comprehensive Health Assessment Program: efficacy of, 57–8 health review, 45 Compulsory Education Law of People’s Republic of China, 73 Confabulation, 184, 189 Confucianism, 77 see also China Convention on Rights of Persons with Disabilities, 34 Cornelia de Lange syndrome, 273, 278 Corpus callosum, 172 disorders of, 173–4 location and structure of, 172 prenatal development of, 173

D DBC see Developmental Behavior Checklist Denver Developmental Screening Test, 72–3 Developmental Behavior Checklist, 263 Diagnostic and Statistical Manual of Mental Disorders-IV: Chinese version, 72 Text Revision (DSM-IV-TR), 241–2 Diagnostic Assessment for Severely Handicapped-II (DASH-II), 271 Diagnostic Criteria for Psychiatric Disorders for Use with Adults with Learning Disabilities/ Mental Retardation (DC-LD), 242 Dimensions of Identity Development Scale, 25

291

Index

Direct instruction (DI), 199, 215, 220, 231 Disability: defined, 36 in functioning of individuals, 36 health care, 43 as health-related demographic characteristic, 59–60 and intellectual disabilities: access to health care, 43–4 building on evidence base, 44–6 disparities, 40–1 social determinants, 41–3 and legislation in China, 73–4 quality of life, 39–40 Second National Sample Survey on Disability, 70 special education classes for children with, 74 Disabled Persons Employment Promotion and Vocational Rehabilitation Act, 130 see also Korea Disabled Persons Welfare Act, 129 see also Korea Disconnection syndrome, 173 Discrete trial teaching, 215 Down syndrome (DS), 23, 46–7, 116, 268, 272, 273 action and object recognition, 149–51 cerebral glucose metabolic rate in, 156 emotion perception and interpersonal interactions, 151–2 imitation and gestures, 152–5 influence of action on perception and cognition, 160–2 and intervention strategies, 162–5 mirror neuron system, role of, 157–8 motor delays, impact of, 162 neurobiology, 155–7 erception–action coupling in, 158–60

E Early Childhood Partial Hospitalization Program, 213, 234 adult questionnaires, 223–4 child assessments used in study, 221–2 Group 1 (Classrooms 1and 2) instructional format, 219 Group 2 (Classroom 3) instructional format, 220 Group 1 descriptive, 225–6 Group 2 descriptive, 227–8 Group 1 PEP-R t-test, 229 Group 2 Woodcock Johnson t-test, 230

intervention effectiveness, 233–4 participants in pilot study, 218 procedure, 218–19 program description: approach, 215–16 environment, 214 implementation, 215 intervention targets, 217 staff, 214–15 result analysis, 220 initial confounding variables, 220–9 pre-and post treatment change, 229 treatment pathway: Group 1, 229, 231 Group 2, 229, 231 satisfaction survey, 231 ECPHP see Early Childhood Partial Hospitalization Program Emotional support, 112, 113 Employment: and disabilities, China in, 72 pattern and adults with ASD, 14 Environment impact on person’s functioning, 36 Erikson’s psychosocial theory, 5 European Union (EU) funded public health projects: Pomona and Pomona II, 46 Extended Objective Measure of Ego Identity Status II, 25

F Family (ies): caregivers, 56 directed support, 113 of individuals with ASD, 7 studies in Vietnam, 99 family needs assessment project, 100–101 issues related to, 101–103 mothers and fathers, comparison between, 107–110 parental stress in, 103–106 social support and, 106–107 surveys in Vietnam, 115–16 Fear of unknown places and investigations, intellectual disabilities, 43 Flourishing mental state, 38 Fragile X syndrome, 273, 274, 278

G Gender roles in parenting within Vietnamese families, 108–109, 116

292

H Health: and disability, 36 gain: complexity of improving, 47 illness and chronic conditions, 48 investigations into, 47 person-centered strategy, 48 research and, 48–9 shared responsibility of stakeholders, 48 and intellectual disabilities: access to health care, 43–4 building on evidence base, 44–6 disparities, 40–1 social determinants, 41–3 social and economic circumstances, 41 socioeconomic position on, 42 Herbal medicine and acupuncture in China, 79 High school exit and children with ASD, 10–11 Homogeneity, 134 Human: development stages: identity and identity confusion, 5–6 intimacy and isolation, 6 motion, 150 Hyperactivity and intellectual disabilities, 43 Hypoplasia, 173

I ICF see International Classification of Functioning IDEA see Individuals with Disabilities Education Act Identity and identity confusion, 5–6 Imitation, 152–5 children with DS: deferred imitation task by, 153 imitative solutions by, 154 performance on subtests of apraxia battery, 153 visual-motor assessment batteries and, 152 nature in DS, 153 Inclusion movement in developed countries, 130–1 Inclusiveness, 58 practical initiatives, 59 public health and intellectual disabilities, 59–60 research strategies, 60–1 Individualized education plans (IEPs) for students with disabilities, 129 see also Korea

Index

Individuals with Disabilities Education Act, 11 Infants, acquisition of cognitive abilities by, 161–2 Informal support, 113 Informed eclectic treatment approach to autism, 210, 212 see also Early Childhood Partial Hospitalization Program Instrumental support, 112 Intellectual disabilities (ID), 240, 275–80 characteristics of, 240 disability, 40–1 evidence-based set of health indicators, 46 incidence, 263–4 invisibility of people with, 48–9 prevalence, 259–62 psychopathology in, 240 behavior problems, variability of, 262 biosocial–behavioral path model and, 278 epidemiological studies on, 243–50 maladaptive behavior, 242–3 mental illness, 241–2 psychopathology prevalence estimation, variability of, 261 risk factors and correlation: chronological age, 267–9 comorbidity with ASD, 271–2 genetic disorders, 272–4 levels of, 265–7 psychological coping skills, 275–6 sex, 270–1 social–environmental factors, 274–5 studies, 39 see also Maladaptive behavior; Mental illness Internal working models, 8 International Classification of Functioning: contextual factors, 36 disability and health, 34 Ireland: intellectual disabilities studies, 39 national policy document on men’s health in, 59–60

K Korea, 126 collectivist-oriented country, 127 individuals with disabilities: advocacy groups and, 143 discrimination due to, 134–5 employment related services, 138 family relationships, 135–7 financial support for, 139 five year policy development plan for, 144

293

Index

general education and, 141 government help to, 138 homogeneity and, 134 inclusion, 140–2 in-service training programs, 141 Korea Institute for Health and Social Affairs, 138–9 medical and housing services, 143 mothers of, 135 movies and television documentaries, 140 negative view of, 134 Prohibition Law of Discrimination against People with Disabilities, 135 public awareness, 139–40 research on, 143 school-aged students with, 131–2 sibling relationship, 136–7 social inclusion, 142 social services, 137–8 social support and services, 142–3 special education services, 132–4 special school and classes, 132 statistics, 131–2 stigma, 134–5 Korea Research Institute for Human Settlements, 127 literacy rate, 128 special education and family services: Act for Promotion of Special Education for Handicapped, 131 developing era, 129–30 Disabled Persons Employment Promotion and Vocational Rehabilitation Act, 130 Disabled Persons Welfare Act, 129 Dongdaemoon Public Elementary School, 129 Education Law, 129 establishing era, 128–9 Five Year Special Education Plan, 129 imposed employment rate, 130 inclusion in, 130–1 individualized education plans (IEPs) for students with disabilities, 129 Korean Braille point system, 129 Paralympics, role in, 129–30 quickening era, 128 transitional era, 129

L Lamina reuniens, 173 Language journals in China, 80 Languishing mental state, 38 LCR see Learning in regular classrooms

Learn At Play Program, 164 Learning in regular classrooms, 74–7 effectiveness, 75 percentages of unenrolled school-aged children with disabilities, 77 student with special needs in, 76 teacher training, 76 Lesch–Nyhan syndrome, 273, 278 Literature on Chinese families of individuals with disabilities, 80 characteristics of, 80–1 family-focused education, 87 parental roles: in child’s education, 81–2 extended family and support, 84 needs and social support, 82–4 practical parenting skills, 83 stress and coping, 84–7 training program, 87 Locomotion, onset of, and development, 161

M Magnetic resonance imaging, 156, 174 for detecting callosal disorders, 174 Mainstream health services and intellectual disabilities, 43 Maladaptive behavior, 242 and ASD, 271–2 as behavioral equivalents, 242–3 chronological age as risk factor, 269 genetic disorders associated with, 274 incidence of, 263–4 levels of ID and, 266 prevalence rate for, 260, 261 sex as risk marker, 270 socio-environmental risk factors for, 274–5 Marriage Law in China, 80 Material support, 113 Mental illness, 241–2 age as predictor of, 268 and ASD, 270 and coping skills, 275–6 effects of sex on, 270 genetic disorders with, 272 incidence of, 263, 264 levels of ID and, 265–6 prevalence rate for, 260 socio-environmental risk factors for, 274–5 Ministry of Labour, Invalids and Social Affairs, 95 Mirror neuron system, 157–8 MOLISA see Ministry of Labour, Invalids and Social Affairs

294

MRI see Magnetic resonance imaging MSPSS see Multidimensional Scale of Perceived Social Support Multidimensional Scale of Perceived Social Support, 108 psychometric properties of, 114 social support, Western and Vietnamese culture, 118

N NADD see National Association for Dually Diagnosed National Association for Dually Diagnosed, 241 National Healthcare Disparities Report: disabilities defined, 44 Neighborhood deprivation, 43 New York sample study, 14 NHDR see National Healthcare Disparities Report NLD see Nonverbal learning disabilities Nonverbal learning disabilities, 186, 189

O Object motion, 150 One-child policy in China, 78

P Parenting Stress Index, 104 Parents of individuals with ASD: protective strategies, 7 Past negative experiences and intellectual disabilities, 43 Person-centered strategies, 50 approaches and personalized supports, 51 carers: lifestyles of, 55–6 needs, 56 community action, 56 development and implementation of educational interventions, 53 health: literacy, 54–5 people’s perceptions and illness, 52 personnel and, 52 promotion, 51 individual need, identification of, 51 information and, 51 information technology, developments in, 53–4 mainstream health-care providers training, 57–8 peer models and advisers, 54 personal responsibility, 52 person-held health records, 52–3

Index

physical health, 51 public health services, 57 seamless health services and supports, 58 supporters and carers, 55 Pivotal response treatment, 216 Point-light stimuli, 149–50 Population-based samples of children with ASD in Sweden study, 15–16 Positive mental health status of individuals, 38 Prader–Willi syndrome, 47, 273, 274, 278 Professional work support, 113 Protection of Persons with Disabilities, Law of People’s Republic of China, 74 PRT see Pivotal response treatment PSI see Parenting Stress Index Psychometrically valid scales in Vietnamese population, 110 multidimensional scale of perceived social support, 114–15 social support scale for parents, 111–13 Vineland adaptive behavior scales, 111 Public health, 37–8

R Raven’s Colored Progressive Matrices, 184 Regulations on Education for Persons with Disabilities in China, 74 Reiss Screen for Maladaptive Behavior, 270 Relationship-focused coping strategies for individuals with disabilities, 86 Rett syndrome, 274, 278 Risk factors and markers, 265 RSMB see Reiss Screen for Maladaptive Behavior

S Second National Sample Survey on Disability, 70 Seizure disorders in children, 177 Self-injurious behavior and intellectual disabilities, 43 Self-management, 205 Sensory impairments and intellectual disabilities, 43 Severe intellectual disability, 85 Shouting and intellectual disabilities, 43 SID see Severe intellectual disability Single-parent families in intellectual disabilities, 80 SLS see Social Life Scale Smith–Magenis syndrome, 278 Social determinants of ill-health, 41–3 Social Life Scale, 108 Social skills, 205

295

Index

Social Support Scale for Parents of Children with Disabilities, 107 social support network, 118 Vietnamese version, 107–108 South Asian children: with intellectual disabilities, 42–3 Special education classes for children with disabilities, 74 SSSPCDD see Social Support Scale for Parents of Children with Disabilities Statewide-linked birth and public school record database, 41

T Targeted treatments, 199–209 domain-specific review studies, 200 on challenging behaviors, 205–206 on communication skills, 206 in self-management skills, 205 on social and play skills, 200–205 environment specific, 208–209 interventionist specific, 209 method-specific interventions, 207 augmentative and alternative communication, 207 video modelling, 207–208 Thematic Apperception Test, 180 Transition process: biological mothers and son/daughter, interview of, 22 integration of: school, family and outcome research, 27–8 parent satisfaction with, 22 and stress, 24 systematic investigation of time, 26–7 unsuccessful, 26 Typically developing (TD) children, 150

U United Kingdom: mental health services in, 59–60 sample study, 14 United States: Convention: disability, person’s dignity and autonomy, 38–9 Healthy People 2010, 60 intellectual disabilities studies, 39 statewide-linked birth and public school record database, 41 Universal Declaration of Universal Human Rights, 36

V VABS see Vineland Adaptive Behavior Scales Video modelling, 207–208 Vietnam, 94 Family Needs Assessment Project, 95 individuals with disabilities: disability concept, 98–9 ending of war and, 99 family studies, 99 issues related to, 101–103 Law on Education, 95 legal rights of, 95–6 mothers and fathers, comparison between, 107–110 parental stress in, 103–106 psychometrically valid scales in Vietnamese population, 110–115 social support and, 106–107 stigma associated with, 99 supernatural causes, 99 superstitious explanations for, 98 traditional view, 98 Vietnamese culture, tradition and practices, 96–8 Yin–Yang during pregnancy, 98 Vineland Adaptive Behavior Scales: Asian nations for, 118 Vietnamese version, 111

W Wechsler tests, 72 Western and non-Western cultures, 104–105 Williams syndrome, 273, 274, 278 World Health Organization (WHO) disability and health, framework on, 34 health definition, 37 health literacy, 54–5 individual and population levels, 36

Y Yin–Yang theory, 98 Young adults with ASD: employment for, 15 job placements, 23 friendships and intimacy with people outside of family, 14 residential placements and, 23 son/daughter exit from school system, 23


Contents of Previous Volumes

Volume 1 A Functional Analysis of Retarded Development SIDNEY W. BIJOU Classical Conditioning and Discrimination Learning Research with the Mentally Retarded LEONARD E. ROSS The Structure of Intellect in the Mental Retardate HARVEY F. DINGMAN AND C. EDWARD MEYERS Research on Personality Structure in the Retardate EDWARD ZIGLER Experience and the Development of Adaptive Behavior H. CARL HAYWOOD AND JACK T. TAPP A Research Program on the Psychological Effects of Brain Lesions in Human Beings RALPH M. REITAN

The Role of Input Organization in the Learning and Memory of Mental Retardates HERMAN H. SPITZ Autonomic Nervous System Functions and Behavior: A Review of Experimental Studies with Mental Defectives RATHE KARRER Learning and Transfer of Mediating Responses in Discriminating Learning BRYAN E. SHEPP AND FRANK D. TURRISI A Review of Research on Learning Sets and Transfer or Training in Mental Defectives MELVIN E. KAUFMAN AND HERBERT J. PREHM Programming Perception and Learning for Retarded Children MURRAY SIDMAN AND LAWRENCE T. STODDARD

Long-Term Memory in Mental Retardation JOHN M. BELMONT

Programming Instruction Techniques for the Mentally Retarded FRANCES M. GREENE

The Behavior of Moderately and Severely Retarded Persons JOSEPH E. SPRADLIN AND FREDERIC L. GIRARDEAU

Some Aspects of the Research on Mental Retardation in Norway IVAR ARNIJOT BJORGEN

Author Index-Subject Index Volume 2 A Theoretical Analysis and Its Application to Training the Mentally Retarded M. RAY DENNY

Research on Mental Deficiency During the Last Decade in France R. LAFON AND J. CHABANIER Psychotherapeutic Procedures with the Retarded MANNY STERNLIGHT Author Index-Subject Index

297

298 Volume 3 Incentive Motivation in the Mental Retardate PAUL S. SIEGEL Development of Lateral and Choice-Sequence Preferences IRMA R. GERJUOY AND JOHN J. WINTERS, JR. Studies in the Experimental Development of Left-Right Concepts in Retarded Children Using Fading Techniques SIDNEY W. BIJOU Verbal Learning and Memory Research with Retardates: An Attempt to Assess Developmental Trends L. R. GOULET


Social Psychology and Mental Retardation WARNER WILSON Mental Retardation in Animals GILBERT W. MEIER Audiologic Aspects of Mental Retardation LYLE L. LLOYD Author Index-Subject Index Volume 5 Medical-Behavioral Research in Retardation JOHN M. BELMONT Recognition Memory: A Research Strategy and a Summary of Initial Findings KEITH G. SCOTT

Research and Theory in Short-Term Memory KEITH G. SCOTT AND MARCIA STRONG SCOTT

Operant Procedures with the Retardate: An Overview of Laboratory Research PAUL WEISBERG

Reaction Time and Mental Retardation ALFRED A. BAUMEISTER AND GEORGE KELLAS

Methodology of Psychopharmacological Studies with the Retarded ROBERT L. SPRAGUE AND JOHN S. WERRY

Mental Retardation in India: A Review of Care, Training, Research, and Rehabilitation Programs J. P. DAS Educational Research in Mental Retardation SAMUEL L. GUSKIN AND HOWARD H. SPICKER Author Index-Subject Index Volume 4 Memory Processes in Retardates and Normals NORMAN R. ELLIS A Theory of Primary and Secondary Familial Mental Retardation ARTHUR R. JENSEN Inhibition Deficits in Retardate Learning and Attention LAIRD W. HEAL AND JOHN T. JOHNSON, JR. Growth and Decline of Retardate Intelligence MARY ANN FISHER AND DAVID ZEAMAN The Measurements of Intelligence A. B. SILVERSTEIN

Process Variables in the Paired-Associate Learning of Retardates ALFRED A. BAUMEISTER AND GEORGE KELLAS Sequential Dot Presentation Measures of Stimulus Trace in Retardates and Normals EDWARD A. HOLDEN, JR. Cultural-Familial Retardation FREDERIC L. GIRARDEAU German Theory and Research on Mental Retardation: Emphasis on Structure LOTHAR R. SCHMIDT AND PAUL B. BALTES Author Index-Subject Index Volume 6 Cultural Deprivation and Cognitive Competence J. P. DAS Stereotyped Acts ALFRED A. BAUMEISTER AND REX FOREHAND

299


Research on the Vocational Habilitation of the Retarded: The Present, the Future MARC W. GOLD Consolidating Facts into the Schematized Learning and Memory System of Educable Retardates HERMAN H. SPITZ An Attentional-Retention Theory of Retardate Discrimination Learning MARY ANN FISHER AND DAVID ZEAMAN

The Role of the Social Agent in Language Acquisition: Implications for Language Intervention GERALD J. MAHONEY AND PAMELA B. SEELY Cognitive Theory and Mental Development EARL C. BUTTERFIELD AND DONALD J. DICKERSON A Decade of Experimental Research in Mental Retardation in India ARUN K. SEN

Studying the Relationship of Task Performance to the Variables of Chronological Age, Mental Age, and IQ WILLIAM E. KAPPAUF

The Conditioning of Skeletal and Autonomic Responses: Normal-Retardate Stimulus Trace Differences SUSAN M. ROSS AND LEONARD E. ROSS

Author Index-Subject Index

Malnutrition and Cognitive Functioning J. P. DAS AND EMMA PIVATO

Volume 7

Research on Efficacy of Special Education for the Mentally Retarded MELVINE E. KAUFMAN AND PAUL A. ALBERTO

Mediational Processes in the Retarded JOHN G. BORKOWSKI AND PATRICIA B. WANSCHURA The Role of Strategic Behavior in Retardate Memory ANN L. BROWN Conservation Research with the Mentally Retarded KERI M. WILTON AND FREDERIC J. BOERSMA Placement of the Retarded in the Community: Prognosis and Outcome RONALD B. MCCARVER AND ELLIS M. CRAIG Physical and Motor Development of Retarded Persons ROBERT H. BRUININKS Subject Index Volume 8

Subject Index

Volume 9 The Processing of Information from Short-Term Visual Store: Developmental and Intellectual Differences LEONARD E. ROSS AND THOMAS B. WARD Information Processing in Mentally Retarded Individuals KEITH E. STANOVICH Mediational Process in the Retarded: Implications for Teaching Reading CLESSEN J. MARTIN Psychophysiology in Mental Retardation J. CLAUSEN

Self-Injurious Behavior ALFRED A. BAUMEISTER AND JOHN PAUL ROLLINGS

Theoretical and Empirical Strategies for the Study of the Labeling of Mentally Retarded Persons SAMUEL L. GUSKIN

Toward a Relative Psychology of Mental Retardation with Special Emphasis on Evolution HERMAN H. SPITZ

The Biological Basis of an Ethic in Mental Retardation ROBERT L. ISAACSON AND CAROL VAN HARTESVELDT

300

Public Residential Services for the Mentally Retarded R. C. SCHEERENBERGER Research on Community Residential Alternatives for the Mentally Retarded LAIRD W. HEAL, CAROL K. SIGELMAN, AND HARVEY N. SWITZKY Mainstreaming Mentally Retarded Children: Review of Research LOUIS CORMAN AND JAY GOTTLIEB Savants: Mentally Retarded Individuals with Special Skills A. LEWIS HILL Subject Index Volume 10 The Visual Scanning and Fixation Behavior of the Retarded LEONARD E. ROSS AND SUSAM M. ROSS Visual Pattern Detection and Recognition Memory in Children with Profound Mental Retardation PATRICIA ANN SHEPHERD AND JOSEPH F. FAGAN III Studies of Mild Mental Retardation and Timed Performance T. NETTELBECK AND N. BREWER Motor Function in Down’s Syndrome FERIHA ANWAR Rumination NIRBHAY N. SINGH Subject Index Volume 11 Cognitive Development of the LearningDisabled Child JOHN W. HAGEN, CRAIG R. BARCLAY, AND BETTINA SCHWETHELM Individual Differences in Short-Term Memory RONALD L. COHEN Inhibition and Individual Differences in Inhibitory Processes in Retarded Children PETER L. C. EVANS


Stereotyped Mannerisms in Mentally Retarded Persons: Animal Models and Theoretical Analyses MARK H. LEWIS AND ALFRED A. BAUMEISTER An Investigation of Automated Methods for Teaching Severely Retarded Individuals LAWRENCE T. STODDARD Social Reinforcement of the Work Behavior of Retarded and Nonretarded Persons LEONIA K. WATERS Social Competence and Interpersonal Relations between Retarded and Nonretarded Children ANGELA R. TAYLOR The Functional Analysis of Imitation WILLIAM R. MCCULLER AND CHARLES L. SALZBERG Index Volume 12 An Overview of the Social Policy of Deinstitutionalization BARRY WILLER AND JAMES INTAGLIATA Community Attitudes toward Community Placement of Mentally Retarded Persons CYNTHIA OKOLO AND SAMUEL GUSKIN Family Attitudes toward Deinstitutionalization AYSHA LATIB, JAMES CONROY, AND CARLA M. HESS Community Placement and Adjustment of Deinstitutionalized Clients: Issues and Findings ELLIS M. CRAIG AND RONALD B. MCCARVER Issues in Adjustment of Mentally Retarded Individuals to Residential Relocation TAMAR HELLER Salient Dimensions of Home Environment Relevant to Child Development KAZUO NIHIRA, IRIS TAN MINK, AND C. EDWARD MEYERS Current Trends and Changes in Institutions for the Mentally Retarded R. K. EYMAN, S. A. BORTHWICK, AND G. TARJAN

301


Methodological Considerations in Research on Residential Alternatives for Developmentally Disabled Persons LAIRD W. HEAL AND GLENN T. FUJIURA A Systems Theory Approach to Deinstitutionalization Policies and Research ANGELA A. NOVAK AND TERRY R. BERKELEY Autonomy and Adaptability in Work Behavior of Retarded Clients JOHN L. GIFFORD, FRANK R. RUSCH, JAMES E. MARTIN, AND DAVID J. WHITE Index

Volume 14 Intrinsic Motivation and Behavior Effectiveness in Retarded Persons H. CARL HAYWOOD AND HARVEY N. SWITZKY The Rehearsal Deficit Hypothesis NORMAN W. BRAY AND LISA A. TURNER Molar Variability and the Mentally Retarded STUART A. SMITH AND PAUL S. SIEGEL Computer-Assisted Instruction for the Mentally Retarded FRANCES A CONNERS, DAVID R. CARUSO, AND DOUGLAS K. DETTERMAN

Sustained Attention in the Mentally Retarded: The Vigilance Paradigm JOEL B. WARM AND DANIEL B. BERCH

Procedures and Parameters of Errorless Discrimination Training with Developmentally Impaired Individuals GIULO E. LANCIONI AND PAUL M. SMEETS

Communication and Cues in the Functional Cognition of the Mentally Retarded JAMES E. TURNURE

Reading Acquisition and Remediation in the Mentally Retarded NIRBHAY N. SINGH AND JUDY SINGH

Metamemory: An Aspect of Metacognition in the Mentally Retarded ELAINE M. JUSTICE

Families with a Mentally Retarded Child BERNARD FARBER AND LOUIS ROWITZ

Volume 13

Inspection Time and Mild Mental Retardation T. NETTELBECK Mild Mental Retardation and Memory Scanning C. J. PHILLIPS AND T. NETTELBECK Cognitive Determinants of Reading in Mentally Retarded Individuals KEITH E. STANOVICH

Social Competence and Employment of Retarded Persons CHARLES L. SALZBERG, MARILYN LIKINS, E. KATHRYN MCCONAUGHY, AND BENJAMIN LINGUGARIS/KRAFT Toward a Taxonomy of Home Environments SHARON LANDESMAN

Comprehension and Mental Retardation LINDA HICKSON BILSKY

Behavioral Treatment of the Sexually Deviant Behavior of Mentally Retarded Individuals R. M. FOXX, R. G. BITTLE, D. R. BECHTEL, AND J. R. LIVESAY

Semantic Processing, Semantic Memory, and Recall LARAINE MASTERS GLIDDEN

Behavior Approaches to Toilet Training for Retarded Persons S. BETTISON

Proactive Inhibition in Retarded Persons: Some Clues to Short-Term Memory Processing JOHN J. WINTERS, JR.

Index

A Triarchic Theory of Mental Retardation ROBERT J. STERNBERG AND LOUIS C. SPEAR Index

Volume 15 Mental Retardation as Thinking Disorder: The Rationalist Alternative to Empiricism HERMAN H. SPITZ

302

Developmental Impact of Nutrition on Pregnancy, Infancy, and Childhood: Public Health Issues in the United States ERNESTO POLLITT The Cognitive Approach to Motivation in Retarded Individuals SHYLAMITH KREITLER AND HANS KREITLER Mental Retardation, Analogical Reasoning, and the Componential Method J. MCCONAGHY Application of Self-Control Strategies to Facilitate Independence in Vocational and Instructional Settings JAMES E. MARTIN, DONALD L. BURGER, SUSAN ELIAS-BURGER, AND DENNIS E. MITHAUG Family Stress Associated with a Developmentally Handicapped Child PATRICIA M. MINNES Physical Fitness of Mentally Retarded Individuals E. KATHRYN MCCONAUGHY AND CHARLES L. SALZBERG Index Volume 16 Methodological Issues in Specifying Neurotoxic Risk Factors for Developmental Delay: Lead and Cadmium as Prototypes STEPHEN R. SCHROEDER The Role of Methylmercury Toxicity in Mental Retardation GARY J. MYERS AND DAVID O. MARSH Attentional Resource Allocation and Mental Retardation EDWARD C. MERRILL Individual Differences in Cognitive and Social Problem-Solving Skills as a Function of Intelligence ELIZABETH J. SHORT AND STEVEN W. EVANS Social Intelligence, Social Competence, and Interpersonal Competence JANE L. MATHIAS


Conceptual Relationships Between Family Research and Mental Retardation ZOLINDA STONEMAN Index

Volume 17 The Structure and Development of Adaptive Behaviors KEITH F. WIDAMAN, SHARON A. BORTHWICK-DUFFY, AND TODD D. LITTLE Perspectives on Early Language from Typical Development and Down Syndrome MICHAEL P. LYNCH AND REBECCA E. EILERS The Development of Verbal Communication in Persons with Moderate to Mild Mental Retardation LEONARD ABBEDUTO Assessment and Evaluation of Exceptional Children in the Soviet Union MICHAEL M. GERBER, VALERY PERELMAN, AND NORMA LOPEZ-REYNA Constraints on the Problem Solving of Persons with Mental Retardation RALPH P. FERRETTI AND AL R. CAVALIER Long-Term Memory and Mental Retardation JAMES E. TURNURE Index

Volume 18 Perceptual Deficits in Mildly Mentally Retarded Adults ROBERT FOX AND STEPHEN OROSS, III Stimulus Organization and Relational Learning SAL A. SORACI, JR. AND MICHAEL T. CARLIN Stimulus Control Analysis and Nonverbal Instructional Methods for People with Intellectual Disabilities WILLIAM J. MCILVANE

303


Sustained Attention in Mentally Retarded Individuals PHILLIP D. TOMPOROWSKI AND LISA D. HAGER How Modifiable Is the Human Life Path? ANN M. CLARKE AND ALAN D. B. CLARKE Unraveling the ‘‘New Morbidity’’: Adolescent Parenting and Developmental Delays JOHN G. BORKOWSKI, THOMAS L. WHITMAN, ANNE WURTZ PASSINO, ELIZABETH A. RELLINGER, KRISTEN SOMMER, DEBORAH KEOUGH, AND KERI WEED Longitudinal Research in Down Syndrome JANET CARR Staff Training and Management for Intellectual Disability Services CHRIS CULLEN Quality of Life of People with Developmental Disabilities TREVOR R. PARMENTER Index Volume 19 Mental Retardation in African Countries: Conceptualization, Services, and Research ROBERT SERPELL, LILIAN MARIGA, AND KARYN HARVEY Aging and Alzheimer Disease in People with Mental Retardation WARREN B. ZIGMAN, NICOLE SCHUPF, APRIL ZIGMAN, AND WAYNE SILVERMAN Characteristics of Older People with Intellectual Disabilities in England JAMES HOGG AND STEVE MOSS

Ways of Analyzing the Spontaneous Speech of Children with Mental Retardation: The Value of Cross-Domain Analyses CATHERINE E. SNOW AND BARBARA ALEXANDER PAN Behavioral Experimentation in Field Settings: Threats to Validity and Interpretation Problems WILLY-TORE MRCH Index Volume 20 Parenting Children with Mental Retardation BRUCE L. BAKER, JAN BLACHER, CLAIRE B. KOPP, AND BONNIE KRAEMER Family Interactions and Family Adaptation FRANK J. FLOYD AND CATHERINE L. COSTIGAN Studying Culturally Diverse Families of Children with Mental Retardation IRIS TAN MINK Older Adults with Mental Retardation and Their Families TAMAR HELLER A Review of Psychiatric and Family Research in Mental Retardation ANN GATH A Cognitive Portrait of Grade School Students with Mild Mental Retardation MARCIA STRONG SCOTT, RUTH PEROU, ANGELIKA HARTL CLAUSSEN, AND LOIS-LYNN STOYKO DEUEL Employment and Mental Retardation NEIL KIRBY Index Volume 21

Epidemiological Thinking in Mental Retardation: Issues in Taxonomy and Population Frequency TOM FRYERS

An Outsider Looks at Mental Retardation: A Moral, a Model, and a Metaprincipal RICHARD P. HONECK

Use of Data Base Linkage Methodology in Epidemiological Studies of Mental Retardation CAROL A. BOUSSY AND KEITH G. SCOTT

Understanding Aggression in People with Intellectual Disabilities: Lessons from Other Populations GLYNIS MURPHY

304


A Review of Self-Injurious Behavior and Pain in Persons with Developmental Disabilities FRANK J. SYMONS AND TRAVIS THOMPSON

The Cost-Efficiency of Supported Employment Programs: A Review of the Literature ROBERT E. CIMERA AND FRANK R. RUSCH

Recent Studies in Psychopharmacology in Mental Retardation MICHAEL G. AMAN

Decision Making and Mental Retardation LINDA HICKSON AND ISHITA KHEMKA

Methodological Issues in the Study of Drug Effects on Cognitive Skills in Mental Retardation DEAN C. WILLIAMS AND KATHRYN J. SAUNDERS The Behavior and Neurochemistry of the Methylazoxymethanol-Induced Microencephalic Rat PIPPA S. LOUPE, STEPHEN R. SCHROEDER, AND RICHARD E.TESSEL Longitudinal Assessment of CognitiveBehavioral Deficits Produced by the Fragile-X Syndrome GENE S. FISCH Index Volume 22 Direct Effects of Genetic Mental Retardation Syndromes: Maladaptive Behavior and Psychopathology ELISABETH M. DYKENS Indirect Effects of Genetic Mental Retardation Disorders: Theoretical and Methodological Issues ROBERT M. HODAPP The Development of Basic Counting, Number, and Arithmetic Knowledge among Children Classified as Mentally Handicapped ARTHUR J. BAROODY The Nature and Long-Term Implications of Early Developmental Delays: A Summary of Evidence from Two Longitudinal Studies RONALD GALLIMORE, BARBARA K. KEOGH, AND LUCINDA P. BERNHEIMER Savant Syndrome TED NETTELBECK AND ROBYN YOUNG

‘‘The Child That Was Meant?’’ or ‘‘Punishment for Sin?’’: Religion, Ethnicity, and Families with Children with Disabilities LARAINE MASTERS GLIDDEN, JEANNETTE ROGERS-DULAN, AND AMY E. HILL Index Volume 23 Diagnosis of Autism before the Age of 3 SALLY J. ROGERS The Role of Secretin in Autistic Spectrum Disorders AROLY HORVATH AND J. TYSON TILDON The Role of Candidate Genes in Unraveling the Genetics of Autism CHRISTOPHER J. STODGELL, JENNIFER L. INGRAM, AND SUSAN L. HYMAN Asperger’s Disorder and Higher Functioning Autism: Same or Different? FRED R. VOLKMAR AND AMI KLIN The Cognitive and Neural Basis of Autism: A Disorder of Complex Information Processing and Dysfunction of Neocortical Systems NANCY J. MINSHEW, CYNTHIA JOHNSON, AND BEATRIZ LUNA Neural Plasticity, Joint Attention, and a Transactional Social-Orienting Model of Autism PETER MUNDY AND A. REBECCA NEAL Theory of Mind and Autism: A Review SIMON BARON-COHEN Understanding the Language and Communicative Impairments in Autism HELEN TAGER-FLUSBERG

305


Early Intervention in Autism: Joint Attention and Symbolic Play CONNIE KASARI, STEPHANNY F. N. FREEMAN, AND TANYA PAPARELLA Attachment and Emotional Responsiveness in Children with Autism CHERYL DISSANAYAKE AND MARIAN SIGMAN Families of Adolescents and Adults with Autism: Uncharted Territory MARSHA MAILICK SELTZER, MARTY WYNGAARDEN KRAUSS, GAEL I. ORSMOND, AND CARRIE VESTAL

Intellectual Disability and Serious Challenging Behavior JIM MANSELL, PETER MCGILL, AND ERIC EMERSON The Mysterious Myth of Attention Deficits and Other Defect Stories: Contemporary Issues in the Developmental Approach to Mental Retardation JACOB A. BURACK, DAVID W. EVANS, CHERYL KLAIMAN, AND GRACE IAROCCI Guiding Visual Attention in Individuals with Mental Retardation RICHARD W. SERNA AND MICHAEL T. CARLIN

Index

Index

Volume 24

Volume 25

Self-Determination and Mental Retardation MICHAEL L. WEHMEYER

Characterizations of the Competence of Parents of Young Children with Disabilities CARL J. DUNST, TRACY HUMPHRIES, AND CAROL M. TRIVETTE

International Quality of Life: Current Conceptual, Measurement, and Implementation Issues KENNETH D. KEITH Measuring Quality of Life and Quality of Services through Personal Outcome Measures: Implications for Public Policy JAMES GARDNER, DEBORAH T. CARRAN, AND SYLVIA NUDLER Credulity and Gullibility in People with Developmental Disorders: A Framework for Future Research STEPHEN GREENSPAN, GAIL LOUGHLIN, AND RHONDA S. BLACK Criminal Victimization of Persons with Mental Retardation: The Influence of Interpersonal Competence on Risk T. NETTELBECK AND C. WILSON The Parent with Mental Retardation STEVE HOLBURN, TIFFANY PERKINS, AND PETER VIETZE

Parent–Child Interactions When Young Children Have Disabilities DONNA SPIKER, GLENNA C. BOYCE, AND LISA K. BOYCE The Early Child Care Study of Children with Special Needs JEAN F. KELLY AND CATHRYN L. BOOTH Diagnosis of Autistic Disorder: Problems and New Directions ROBYN YOUNG AND NEIL BREWER Social Cognition: A Key to Understanding Adaptive Behavior in Individuals with Mild Mental Retardation JAMES S. LEFFERT AND GARY N. SIPERSTEIN Proxy Responding for Subjective Well-Being: A Review ROBERT A. CUMMINS

Psychiatric Disorders in Adults with Mental Retardation STEVE MOSS

People with Intellectual Disabilities from Ethnic Minority Communities in the United States and the United Kingdom CHRIS HATTON

Development and Evaluation of Innovative Residential Services for People with Severe

Perception and Action in Mental Retardation W. A. SPARROW AND ROSS H. DAY

306 Volume 26 A History of Psychological Theory and Research in Mental Retardation since World War II DONALD K. ROUTH AND STEPHEN R. SCHROEDER Psychopathology and Intellectual Disability: The Australian Child to Adult Longitudinal Study BRUCE J. TONGE AND STEWART L. EINFELD Psychopathology in Children and Adolescents with Intellectual Disability: Measurement, Prevalence, Course, and Risk JAN L. WALLANDER, MARIELLE C. DEKKER, AND HANS KOOT Resilience, Family Care, and People with Intellectual Disabilities GORDON GRANT, PAULRAMCHARAN, AND PETER GOWARD Prevalence and Correlates of Psychotropic Medication Use among Adults with Developmental Disabilities: 1970–2000 MARIA G. VALDOVINOS, STEPHEN R. SCHROEDER, AND GEUNYOUNG KIM Integration as Acculturation: Developmental Disability, Deinstitutionalization, and Service Delivery Implications M. KATHERINE BUELL Cognitive Aging and Down Syndrome: An Interpretation J. P. DAS Index Volume 27 Language and Communication in Individuals with Down Syndrome ROBIN S. CHAPMAN Language Abilities of Individuals with Williams Syndrome CAROLYN B. MERVIS, BYRON F. ROBINSON, MELISSA L. ROWE, ANGELA M. BECERRA, AND BONITA P. KLEIN-TASMAN Language and Communication in Fragile X Syndrome MELISSA M. MURPHY AND LEONARD ABBEDUTO


On Becoming Socially Competent Communicators: The Challenge for Children with Fetal Alcohol Exposure TRUMAN E. COGGINS, LESLEY B. OLSWANG, HEATHER CARMICHAEL OLSON, AND GERALYN R. TIMLER Memory, Language Comprehension, and Mental Retardation EDWARD C. MERRILL, REGAN LOOKADOO, AND STACY RILEA Reading Skills and Cognitive Abilities of Individuals with Mental Retardation FRANCES A. CONNERS Language Interventions for Children with Mental Retardation NANCY C. BRADY AND STEVEN F. WARREN Augmentative and Alternative Communication for Persons with Mental Retardation MARYANN ROMSKI, ROSE A. SEVCIK, AND AMY HYATT FONSECA Atypical Language Development in Individuals with Mental Retardation: Theoretical Implications JEAN A. RONDAL Index Volume 28 Promoting Intrinsic Motivation and SelfDetermination in People with Mental Retardation EDWARD L. DECI Applications of a Model of Goal Orientation and Self-Regulated Learning to Individuals with Learning Problems PAUL R. PINTRICH AND JULIANE L. BLAZEVSKI Learner-Centered Principles and Practices: Enhancing Motivation and Achievement for Children with Learning Challenges and Disabilities BARBARA L. MCCOMBS Why Pinocchio Was Victimized: Factors Contributing to Social Failure in People with Mental Retardation STEPHEN GREENSPAN

307


Understanding the Development of Subnormal Performance in Children from a MotivationalInteractionist Perspective JANNE LEPOLA, PEKKA SALONEN, MARJA VAURAS, AND ELISA POSKIPARTA Toward Inclusion Across Disciplines: Understanding Motivation of Exceptional Students HELEN PATRICK, ALLISON M. RYAN, ERIC M. ANDERMAN, AND JOHN KOVACH Loneliness and Developmental Disabilities: Cognitive and Affective Processing Perspectives MALKA MARGALIT The Motivation to Maintain Subjective Well-Being: A Homeostatic Model ROBERT A. CUMMINS AND ANNA L. D. LAU Quality of Life from a Motivational Perspective ROBERT L. SCHALOCK Index

Risk Factors for Alzheimer’s Disease in Down Syndrome LYNN WARD Precursors of Mild Mental Retardation in Children with Adolescent Mothers JOHN G. BORKOWSKI, JULIE J. LOUNDS, CHRISTINE WILLARD NORIA, JENNIFER BURKE LEFEVER, KERI WEED, DEBORAH A. KEOGH, AND THOMAS L. WHITMAN The Ecological Context of Challenging Behavior in Young Children with Developmental Disabilities ANITA A. SCARBOROUGH AND KENNETH K. POON Employment and Intellectual Disability: Achieving Successful Employment Outcomes KAYE SMITH, LYNNE WEBBER, JOSEPH GRAFFAM, AND CARLENE WILSON Technology Use and People with Mental Retardation MICHAEL L. WEHMEYER, SEAN J. SMITH, SUSAN B. PALMER, DANIEL K. DAVIES, AND STEVEN E. STOCK Index

Volume 29 Behavioral Phenotypes: Going Beyond the Two-Group Approach ROBERT M. HODAPP Prenatal Drug Exposure and Mental Retardation ROBERT E. ARENDT, JULIA S. NOLAND, ELIZABETH J. SHORT, AND LYNN T. SINGER Spina Bifida: Genes, Brain, and Development JACK M. FLETCHER, MAUREEN DENNIS, HOPE NORTHRUP, MARCIA A. BARNES, H. JULIA HANNAY, SUSAN H. LANDRY, KIMCOPELAND, SUSAN E. BLASER, LARRY A. KRAMER, MICHAEL E. BRANDT, AND DAVID J. FRANCIS The Role of the Basal Ganglia in the Expression of Stereotyped, Self-Injurious Behaviors in Developmental Disorders HOWARD C. CROMWELL AND BRYAN H. KING

Volume 30 Neurodevelopmental Effects of Alcohol THOMAS M. BURBACHER AND KIMBERLY S. GRANT PCBs and Dioxins HESTIEN J. I. VREUGDENHIL AND NYNKE WEISGLAS-KUPERUS Interactions of Lead Exposure and Stress: Implications for Cognitive Dysfunction DEBORAH A. CORY-SLECHTA Developmental Disabilities Following Prenatal Exposure to Methyl Mercury from Maternal Fish Consumption: A Review of the Evidence GARY J. MYERS, PHILIP W. DAVIDSON, AND CONRAD F. SHAMLAYE Environmental Agents and Autism: Once and Future Associations SUSAN L. HYMAN, TARA L. ARNDT, AND PATRICIA M. RODIER

308

Endocrine Disruptors as a Factor in Mental Retardation BERNARD WEISS The Neurotoxic Properties of Pesticides HERBERT L. NEEDLEMAN Parental Smoking and Children’s Behavioral and Cognitive Functioning MICHAEL WEITZMAN, MEGAN KAVANAUGH, AND TODD A. FLORIN Neurobehavioral Assessment in Studies of Exposures to Neurotoxicants DAVID C. BELLINGER From Animals to Humans: Models and Constructs DEBORAH C. RICE Index

Volume 31 The Importance of Cognitive–Motivational Variables in Understanding the Outcome Performance of Persons with Mental Retardation: A Personal View from the Early Twenty-First Century HARVEY N. SWITZKY Self-Determination, Causal Agency, and Mental Retardation MICHAEL L. WEHMEYER AND DENNIS E. MITHAUG The Role of Motivation in the Decision Making of Adolescents with Mental Retardation ISHITA KHEMKA AND LINDA HICKSON Individual Differences in Interpersonal Relationships for Persons with Mental Retardation YONA LUNSKY Understanding Low Achievement and Depression in Children with Learning Disabilities: A Goal Orientation Approach GEORGIOS D. SIDERIDIS Motivation and Etiology-Specific Cognitive–Linguistic Profiles DEBORAH J. FIDLER


The Role of Motivation and Psychopathology in Understanding the IQ–Adaptive Behavior Discrepancy MARC J. TASSE´ AND SUSAN M. HAVERCAMP Behavior-Analytic Experimental Strategies and Motivational Processes in Persons with Mental Retardation WILLIAM V. DUBE AND WILLIAM J. MCILVANE A Transactional Perspective on Mental Retardation H. CARL HAYWOOD Index Volume 32 Research on Language Development and Mental Retardation: History, Theories, Findings, and Future Directions LEONARD ABBEDUTO, YOLANDA KELLER-BELL, ERICA KESIN RICHMOND, AND MELISSA M. MURPHY Residential Services Research in the Developmental Disabilities Sector STEVE HOLBURN AND JOHN W. JACOBSON The Measurement of Poverty and Socioeconomic Position in Research Involving People with Intellectual Disability ERIC EMERSON, HILARY GRAHAM, AND CHRIS HATTON The Influence of Prenatal Stress and Adverse Birth Outcome on Human Cognitive and Neurological Development LAURA M. GLYNN AND CURT A. SANDMAN Fluid Cognition: A Neglected Aspect of Cognition in Research on Mental Retardation CLANCY BLAIR AND MEGAN PATRICK Dietary Supplementation with Highly Unsaturated Fatty Acids: Implications for Interventions with Persons with Mental Retardation from Research on Infant Cognitive Development, ADHD, and Other Developmental Disabilities NATALIE SINN AND CARLENE WILSON

309


Screening for Autism in Infants, Children, and Adolescents KYLIE M. GRAY, BRUCE J. TONGE, AND AVRIL V. BRERETON People with Mental Retardation and Psychopathology: Stress, Affect Regulation and Attachment: A Review CARLO SCHUENGEL AND CEES G. C. JANSSEN Diagnosis of Depression in People with Developmental Disabilities: Progress and Problems ANN R. POINDEXTER Index Volume 33 Developmental Epidemiology of Mental Retardation/Developmental Disabilities: An Emerging Discipline ROBERT M. HODAPP AND RICHARD C. URBANO

KIM VAN NAARDEN BRAUN, AND MARSHALYN YEARGIN-ALLSOPP Using GIS to Investigate the Role of Recreation and Leisure Activities in the Prevention of Emotional and Behavioral Disorders TINA L. STANTON-CHAPMAN AND DEREK A. CHAPMAN The Developmental Epidemiology of Mental Retardation and Developmental Disabilities DENNIS P. HOGAN, MICHAEL E. MSALL, AND JULIA A. RIVERA DREW Evolution of Symptoms and Syndromes of Psychopathology in Young People with Mental Retardation STEWART L. EINFELD, BRUCE J. TONGE, KYLIE GRAY, AND JOHN TAFFE Index

Volume 34

Record Linkage: A Research Strategy for Developmental Epidemiology RICHARD C. URBANO

Historical Overview of Assessment in Intellectual Disability STEPHEN R. SCHROEDER AND R. MATTHEW REESE

Second-Order Linkage and Family Datasets SHIHFEN TU, CRAIG A. MASON, AND QUANSHENG SONG

Assessing Mental Retardation Using Standardized Intelligence Tests BARBARA TYLENDA, JACQUELINE BECKETT, AND ROWLAND P. BARRETT

Incorporating Geographical Analysis into the Study of Mental Retardation and Developmental Disabilities RUSSELL S. KIRBY

Adaptive Behavior Scales DENNIS R. DIXON

Statistical Issues in Developmental Epidemiology and Developmental Disabilities Research: Confounding Variables, Small Sample Size, and Numerous Outcome Variables JENNIFER URBANO BLACKFORD Economic Perspectives on Service Choice and Optimal Policy: Understanding the Effects of Family Heterogeneity on MR/DD Outcomes STEPHANIE A. SO Public Health Impact: Metropolitan Atlanta Developmental Disabilities Surveillance Program RACHEL NONKIN AVCHEN, TANYA KARAPURKAR BHASIN,

Educational Assessment MARK F. O’REILLY, BONNIE O’REILLY, JEFF SIGAFOOS, GIULIO LANCIONI, VANESSA GREEN, AND WENDY MACHALICEK Autism and Pervasive Developmental Disorders BART M. SEVIN, CHERYL L. KNIGHT, AND SCOTT A. BRAUD Psychopathology: Depression, Anxiety, and Related Disorders PETER STURMEY Psychotropic Medication Effect and Side Effects ERIK A. MAYVILLE Memory Disorders HEATHER ANNE STEWART AND HOLLY GARCIE-MERRITT

310

Assessment of Self-Injurious and Aggressive Behavior JOHANNES ROJAHN, THEODORE A. HOCH, KATIE WHITTAKER, AND MELISSA L. GONZA´ LEZ Social Skills JONATHAN WILKINS AND JOHNNY L. MATSON Self-Care Skills REBECCA L. MANDAL, BRANDI SMIROLDO, AND JOANN HAYNES-POWELL Feeding Disorders DAVID E. KUHN, PETER A. GIROLAMI, AND CHARLES S. GULOTTA Pain Assessment FRANK ANDRASIK AND CARLA RIME Index Volume 35 Theory and Research on Autism: Do We Need a New Approach to Thinking About and Studying This Disorder? THOMAS L. WHITMAN AND NAOMI EKAS Social Cognition in Children with Down Syndrome KATIE R. CEBULA AND JENNIFER G. WISHART The Development of Social Competence Among Persons with Down Syndrome: From Survival to Social Inclusion GRACE IAROCCI, JODI YAGER, ADRIENNE ROMBOUGH, AND JESSICA MCLAUGHLIN The Flynn Effect and the Shadow of the Past: Mental Retardation and the Indefensible and Indispensable Role of IQ JAMES R. FLYNN AND KEITH F. WIDAMAN Remaining Open to Quantitative, Qualitative, and Mixed-Method Designs: An Unscientific Compromise, or Good Research Practice? KEITH R. MCVILLY, ROGER J. STANCLIFFE, TREVOR R. PARMENTER, AND ROSANNE M. BURTON-SMITH


Active Support: Development, Evidence Base, and Future Directions VASO TOTSIKA, SANDY TOOGOOD, AND RICHARD P. HASTINGS Child Abuse Among Children with Disabilities: What We Know and What We Need to Know MARISA H. FISHER, ROBERT M. HODAPP, AND ELISABETH M. DYKENS Siblings of Children with Mental Retardation: The Role of Helping ELIZABETH MIDLARSKY, MARY ELIZABETH HANNAH, EREL SHVIL, AND AMANDA JOHNSON Index

Volume 36 Newborn Screening for Intellectual Disability: Past, Present, and Future DON BAILEY Responsive Parenting: Closing the Learning Gap for Children with Early Developmental Problems SUSAN H. LANDRY, HEATHER B. TAYLOR, CATHY GUTTENTAG, AND KAREN E. SMITH Trisomy 21: Causes and Consequences JEANNIE VISOOTSAK AND STEPHANIE L. SHERMAN Alzheimer’s Disease in Adults with Down Syndrome WARREN B. ZIGMAN, DARLYNNE A. DEVENNY, SHARON J. KRINSKYMCHALE, EDMUND C. JENKINS, TIINA K. URV, JERZY WEGIEL, NICOLE SCHUPF, AND WAYNE SILVERMAN Foolish Action in Adults with Intellectual Disabilities: The Forgotten Problem of Risk-Unawareness STEPHEN GREENSPAN Animal Models of Self-Injurious Behavior: Induction, Prevention, and Recovery STEPHEN R. SCHROEDER, PIPPA S. LOUPE, AND RICHARD E. TESSEL

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Theoretical and Methodological Issues in Sibling Research J. CAROLYN GRAFF, SUSAN NEELYBARNES, AND HEATHER SMITH Understanding Individual Differences in Adaptation in Parents of Children with Intellectual Disabilities: A Risk and Resilience Perspective MALIN B. OLSSON ‘‘What do you Think if . . .’’: Using Vignettes to Study Attitudes Toward Adult Sibling Caregiving and Competence of Parents of Children with Disabilities BRIAN M. JOBE AND LARAINE M. GLIDDEN Index Volume 37 Mothers and Fathers Together: Contrasts in Parenting Across Preschool to Early School Age in Children with Developmental Delays KEITH CRNIC, ANITA PEDERSEN Y ARBONA, BRUCE BAKER, AND JAN BLACHER The Transition to Adulthood for Individuals with Intellectual Disability FRANK J. FLOYD, CATHERINE L. COSTIGAN, AND VIVIAN E. PIAZZA By Choice or By Chance: Longitudinal Perspectives on Resilience and Vulnerability in Adoptive and Birth Parents of Children with Developmental Disabilities LARAINE MASTERS GLIDDEN AND BRIAN M. JOBE

Socioeconomic Position, Poverty, and Family Research ERIC EMERSON AND CHRIS HATTON Using Large-Scale Databases to Examine Families of Children with Intellectual and Developmental Disabilities ROBERT M. HODAPP AND RICHARD C. URBANO A Rich Mosaic: Emerging Research on Asian Families of Persons with Intellectual and Developmental Disabilities SUBHARATI GHOSH AND SANDY MAGAÑA Biomarkers in the Study of Families of Children with Developmental Disabilities MARSHA MAILICK SELTZER, LEONARD ABBEDUTO, JAN S. GREENBERG, DAVID ALMEIDA, JINKUK HONG, AND WHITNEY WITT Siblings of Children with Intellectual Disabilities: Normal, Average, or Not Too Different? ZO STONEMAN Family Support Interventions for Families of Adults with Intellectual and Developmental Disabilities TAMAR HELLER AND ABIGAIL SCHINDLER Interventions Aimed at Improving Child Language by Improving Maternal Responsivity NANCY BRADY, STEVEN F. WARREN, AND AUDRA STERLING Index


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