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National Institute of Neurological Disorders and Stroke (NINDS)

Emotional and Behavioral Health in Persons with Mental Retardation/Developmental Disabilities: Research Challenges and Opportunities
November 29 – December 1, 2001 • Rockville, Maryland


Agenda Participants Workshop Summary

Agenda

Thursday, November 29
 
8:00 a.m. Welcome
Audrey Penn, Acting Director, NINDS
Duane Alexander, Director, NICHD
Richard Nakamura, Deputy Director, NIMH
8:20 a.m. Workshop Format
Giovanna Spinella
  Workshop Goals
James Harris
8:30 a.m. Opening Remarks
Eunice Kennedy Shriver
Executive Vice President
Joseph P. Kennedy Jr. Foundation
8:35 a.m. Overview Sessions
Session 1: Research Opportunities and Challenges
  Introduction
Travis Thompson
8:45 a.m. Behavior and Genetics
Jonathan Flint
9:00 a.m. Neuroimaging
Judith Rumsey
9:15 a.m. Mechanisms of Brain Plasticity and Mental Retardation Syndromes
William Greenough
9:30 a.m. Psychosocial Issues
Sharon Ramey
9:45 a.m. Questions/Issues for Working Group Discussions
10:10 a.m. Session 2: Epidemiology
  Introduction
James Harris
10:20 a.m. Concepts and Issues with Respect to Epidemiological Studies of Mental Retardation
Sir Michael Rutter
10:35 a.m. Rates and Types of Psychopathology: Lessons from Mental Retardation Syndromes
Elisabeth Dykens
10:50 a.m. Considerations for the Epidemiology of Emotional and Behavioral Disorders in Persons with Cognitive Disabilities
Vincent Campbell
11:05 a.m. New Service Delivery Systems: Relationship to Epidemiological Studies
David Braddock
11:20 a.m. Questions/Issues for Working Group Discussion
12:30 p.m. Session 3: Diagnosis/Assessment
  Introduction
Bryan King
12:40 p.m. New Ideas, Assessment Instruments, and
Dual Diagnosis
Steven Reiss
12:55 p.m. Instrument Development-Psychosocial Aspects
Catherine Lord
1:10 p.m. Functional Analysis
Brian Iwata
1:25 p.m. Diagnosis in Specific Syndromes
Carl Feinstein
1:40 p.m. Questions/Issues for Working Group Discussion
1:50 p.m. Session 4: Ethical Considerations (Assent/Consent/Social Policy/Legal)
  Introduction
Norm Fost
2:10 p.m. Assent/Consent/Surrogate Decision Making
Robert Dinerstein
2:25 p.m. Federal Response to the National Bioethics Advisory Commission (NBAC) Report
William Raub
2:40 p.m. The Minimal Risk Rule
Robert M. Nelson
2:55 p.m. Questions/Issues for Working Group Discussion
3:30 p.m. Session 5: Interventions Research
  Introduction
Mark Lewis
3:40 p.m. Psychopharmacology
Michael Aman
3:55 p.m. Psychosocial Interventions
Wayne Fisher
4:10 p.m. Quality of Life Outcomes
Marsha Seltzer
4:25 p.m. Questions/Issues for Working Group Discussion
4:35 p.m. Session 6: Research Design
  Introduction
Mark Appelbaum
4:45 p.m. Psychopharmacology Research Design
C. Keith Conners
5:00 p.m. Pediatric Trials
Robert Sprague
5:15 p.m. Pediatric Clinical Trials: The FDA Experience/Orphan Drug–FDA
William Rodriguez/Tan Nguyen
5:35 p.m. Questions/Issues for Working Group Discussion
5:45 p.m. Adjourn Day 1/Overview Areas 1-6
Work Group leaders will hand to NIH program officer any revisions to work group discussion topics: Epidemiology; Ethical Considerations; Diagnosis/Assessment; Intervention Research; Research Design
 
Friday, November 30
 
8:30 a.m. Session 7: Research Training Needs (Building the Infrastructure)
  Introduction
James McCracken
8:40 a.m. Psychiatric Training
James McCracken
8:55 a.m. Pediatric and Genetics Training
Rodney Howell
9:10 a.m. Psychology, Special Education, Behavioral Training
Donald Routh
9:25 a.m. Developmental Cognitive Neurology
Roger Brumback
9:40 a.m. Questions/Issues for Working Group Discussion
9:50 a.m. Divide into Work Group Sessions
 
  • Epidemiology
  • Diagnosis/Assessment
  • Ethical Considerations
  • Interventions Research
  • Research Design
  • Research Training Needs: Building the Infrastructure
5:00 p.m. Adjourn Day 2/Workgroup Sessions
 
Saturday, December 1
 
8:30 a.m. Workgroup Presentations/ Discussions
8:40 a.m. Epidemiology
9:10 a.m. Diagnosis/Assessment
9:40 a.m. Ethical Considerations
10:10 a.m. Interventions Research
11:00 a.m. Research Design
11:30 a.m. Research Training Needs–Building the Infrastructure
12:00 noon General Discussion
1:00 p.m. Closing Remarks/Adjourn

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Participants

Pasquale J. Accardo, M.D.
Professor of Pediatrics
New York Medical College
Director of Pediatrics
Westchester Institute for Human Development
332 Cedarwood WIND/WMC
Valhalla, NY 10595
Phone: (914) 493-1330
Fax: (914) 493-1973

Duane Alexander, M.D.
Director
National Institute of Child Health and Human Development
10 Center Drive, Building 31, Room 2A03
Bethesda, MD 20892-2425
Phone: (301) 496-3454
Fax: (301) 402-1104

Michael G. Arnall, Ph.D.
Professor
Department of Psychology and Psychiatry
Ohio State University
The Nisonger Center
1581 Dodd Drive
Columbus, OH 43210-1257
Phone: (614) 688-4196
Fax: (614) 688-4908

James C. Anthony, Ph.D.
Professor
Bloomberg School of Public Health
The Johns Hopkins University
624 N. Broadway
Baltimore, MD 21205
Phone: (410) 955-8551
Fax: (410) 933-9088

Mark I. Appelbaum, Ph.D.
Department of Psychology
University of California, San Diego
Psychology-0109
La Jolla, CA 92093-0109
San Diego, CA 92093-0109
Phone: (858) 534-7959
Fax: (858) 534-7190

B. J. Arseneau, M.D., M.P.H.
Chief Psychiatrist
Office of Disability
Division of Medical and Vocational Policy
Social Security Administration
OD DMVP 3-A-8
OPNS Building
6401 Security Boulevard
Baltimore, MD 21235-0001
Phone: (410) 966-1970
Fax: (410) 966-3372

Natalie S. Baker
Pre-Doctoral Research Fellow
National Institute of Mental Health
National Institutes of Health
Child Psychiatry Branch
10 Center Drive, Building 10, Rm. 3N202
Bethesda, MD 20892-1150
Phone: (301) 435-4506
Fax: (301) 402-7160

Beth I. Barol, Ph.D.
Clinical Director
Pennsylvania Office of Mental Retardation
Statewide Training and T.A. Institute
Office of Mental Retardation-Pennsylvania
1420 Firethorne Lane
Wyndmoor, PA 19038
Phone: (215) 233-3520
Fax: (215) 836-5435

Trisha Beuhring
Institute on Community Integration
University of Minnesota
105 Pattel Hall
150 Pillsbury Dr., SE
Minneapolis, MN 53455
Phone: (651) 644-3168

Sharon Borthwick-Duffy, Ph.D.
Professor
Graduate School of Education
University of California, Riverside
Riverside, CA 92521
Phone: (909) 787-4598
Fax: (909) 787-4598

David Braddock, Ph.D.
Executive Director
Associate Vice President for Research
University of Colorado
Coleman Institute for Cognitive Disabilities
University of Colorado, 586 SYS
4001 Discovery Drive, Suite 210
Boulder, CO 80309-0586
Phone: (303) 492-0639
Fax: (303) 735-5643

Roger A. Brumback, M.D.
Professor and Chairman Pathology Department
Creighton University School of Medicine
St. Joseph Hospital
601 North 30th Street
Omaha, NE
Phone: (402) 280-4858
Fax: (402) 280-5247

Janice Buelow, Ph.D., R.N.
College of Nursing
Indiana University
1715 N. Diary Lane
Arlington Heights, IL
Phone: (847) 253-0221
Fax: (312) 939-0391

Carlos E. Caban, Ph.D., M.P.H.
Extramural Program Policy Officer
Office of Extramural Research
Office of the Director
National Institutes of Health
6701 Rockledge Drive, Room 6190
Bethesda, MD 20892-7911
Phone: (301) 435-2690
Fax: (301) 480-8443

Vincent A. Campbell, Ph.D.
Health Scientist
National Center of Birth Defects and
Developmental Disabilities
Centers for Disease Control and Prevention
4770 Buford Highway, F-35
Atlanta, GA 30341-3724
Phone: (770) 488-7684
(TDD) (770) 488-7083
Fax: (770) 488-7075

Katherine N. Clapp, M.S.
President and Co-Founder
FRAXA Research Foundation
45 Pleasant Street
Newburyport, MA 01950
Phone: (978) 462-1866
Fax: (978) 463-9985

Lisa J. Colpe, Ph.D., M.P.H.
Chief, Epidemiology and Risk Factors Program
Division of Mental Disorders,
Behavioral Research and AIDS
Adult Psychopathology and Prevention
Research Branch (DMDBA/APPRB)
National Institute of Mental Health
National Institutes of Health
6001 Executive Boulevard, Room 6182
Bethesda, MD 20892
Phone: (301) 443-3944
Fax: (301) 443-4611

C.K. Conners, Ph.D.
Professor Emeritus of Medical Psychology
Department of Psychiatry
Duke University
718 Rutherford Street
Durham, NC 27705
Phone: (919) 416-2084
Fax: (919) 401-0218/(919) 286-7081

Robert Cudeck, Ph.D.
Professor
Department of Psychology
University of Minnesota
75 East River Road
Minneapolis, MN 55455
Phone: (612) 625-3078
Fax: (612) 626-2079

Douglas S. Diekema, M.D., M.P.H.
Associate Professor
Pediatrics and Medical History and Ethics
Department of Pediatrics
University of Washington
18140 60th Place, NE
Kenmore, WA 98028
Phone: (206) 526-2599
Fax: (206) 729-3070

Robert D. Dinerstein, Jr., J.D.
Professor of Law and Associate Dean
Office of Academic Affairs American University
Washington College of Law
4801 Massachusetts Avenue, NW
Washington, DC 20016
Phone: (202) 274-4141
Fax: (202) 274-4015

Elisabeth M. Dykens, Ph.D.
Professor
Department of Child and Adolescent Psychiatry
University of California at Los Angeles
760 Westwood Plaza, NPI 47-422B
Los Angeles, CA 90024
Phone: (310) 206-4633
Fax: (310) 206-4446

Emmeline Edwards, Ph.D.
Program Director
Systems and Cognitive Neuroscience
National Institute of Neurological Disorders and Stroke
National Institutes of Health
6001 Executive Boulevard, Room 2109
Bethesda, MD 20892
Phone: (301) 496-9964
Fax: (301) 402-2060

Steven M. Eidelman
Executive Director
The Arc of the United States
1010 Wayne Avenue, Suite 650
Silver Spring, MD 20910
Phone: (301) 565-3842
Fax: (301) 565-3843

Stewart L. Einfeld, M.D., D.C.H.
Child and Adolescent Psychiatry Programs
University of New South Wales
2 Short Street
Kogarah, NSW 2217
Australia
Phone: 61-2-9350-2447
Fax: 61-2-9350-2451

Nadia N. Facey
Public Policy Coordinator
Government Affairs
Epilepsy Foundation
4351 Garden City Drive
Landover, MD 20785-7223
Phone: (301) 459-3700
Fax: (301) 577-2684

Carl Feinstein, M.D.
Professor
Department of Psychiatry
Stanford University School of Medicine
401 Quarry Road
Stanford, CA 94305-5719
Phone: (650) 498-5246
Fax: (650) 723-5531

Sandy Finucane, J.D.
Vice President
Legal & Government Affairs
Epilepsy Foundation
4351 Garden City Drive
Landover, MD 20785-7223
Phone: (301) 459-3700
Fax: (301) 577-2684

Robert J. Fletcher, D.S.W., M.S.W.
Executive Director, CEO, and Founder
National Association for the Dually Diagnosed (NADD)
132 Fair Street
Kingston, NY 12401
Phone: (845) 331-4336
Fax: (845) 331-4569

Wayne W. Fisher, Ph.D.
Executive Director
Marcus Behavior Center
Kennedy Krieger Institute/Marcus Institute
1605 Chantilly Drive
Atlanta, GA 30324
Phone: (404) 727-9406
Fax: (404) 727-9550

Jonathan Flint, M.D.
Wellcome Trust Centre for Human Genetics
Roosevelt Drive
Oxford OX3 7BN
United Kingdom
Phone: 44 1865287512
Fax: 44 1865287501

Eric Fombonne, M.D.
Professor
Department of Psychiatry
McGill University
Montreal Children's Hospital
4028 Ste Catherine West
Montreal, Quebec H3Z 1P2
Canada
Phone: (514) 412-440
Fax: (514) 412-4337

Norman Fost, M.D., MPH
Professor of Pediatrics
Director of the Program in Medical Ethics
University of Wisconsin-Madison
Department of Pediatrics, Room H4/452
University of Wisconsin Hospital
600 Highland Avenue
Madison, WI 53792
Phone: (608) 263-8562
Fax: (608)263-6210

William T. Greenough, Ph.D.
Professor
Department of Psychology
Beckman Institute
University of Illinois
405 North Matthews Avenue
Urbana, IL 61801
Phone: (217) 333-4472
Fax: (217) 244-5180

Surya N. Gupta, M.D.
Clinical Fellow
National Institute of Neurological Disorders and Stroke
Neuroscience Center
9000 Rockville Pike, Building 10, Room 3D/03
Bethesda, MD 20892-9527
Phone: (301) 496-1465
Fax: (301) 480-8354

James W. Hanson, M.D.
Acting Chief
Clinical and Genetic Epidemiology Research Branch
Division of Cancer Control and Population Sciences
National Cancer Institute
6130 Executive Boulevard, MSC 7393 EPN Room 5134
Bethesda, MD 20892-7393
Phone: (301) 435-6612
Fax: (301) 435-5477

Janalee Heinemann, M.S.W.
Executive Director, National Office
The Prader- Willi Syndrome Association
5700 Midnight Pass Road
Sarasota, FL 34242
Phone: (800) 926-4797
Fax: (941) 312-0142

Deborah G. Hirtz, M.D.
Program Director
Clinical Trials and Studies
National Institute of Neurological Disorders and Stroke
National Institutes of Health
6001 Executive Boulevard
Bethesda, MD 20892
Phone: (301) 496-5821
Fax: (301) 480-1080

R. Rodney Howell, M.D.
Professor and Chairman
Department of Pediatrics
University of Miami School of Medicine (D820)
P.O. Box D16820
Miami, FL 33101
Phone: (305) 243-3985
Fax: (305) 243-3990

Jamie Davis Hueston, Ph.D.
Principal Psychology Consultant
Indian Health Service
OPH/OCPS/Behavioral Health
12300 Twinbrook Parkway, Suite 605
Rockville, MD 20852
Phone: (301) 443-2038
Fax: (301) 443-7623

Anne DesNoyers Hurley, Ph.D.
Director, Developmental Disabilities Clinic
Department of Psychiatry
Tufts-New England Medical Center
750 Washington Street, #1007
Boston, MA 02111
Phone: (617) 636-8763
Fax: (617) 636-8442

Henrietta Hyatt-Knorr
Acting Director
Office of Rare Diseases
National Institutes of Health
31 Center Drive, 1B19
Bethesda, MD 20892
Phone: 301-435-6045
Fax: 301-480-9655

Brian A. Iwata, Ph.D.
Professor of Psychology and Psychiatry
Department of Psychology
University of Florida
114 Psychology Building
Gainesville, FL 32611
Phone: (352) 392-0601 ext.281
Fax: (352) 392-7985

John W. Jacobson, Ph.D.
Program Operations Specialist
Planning and Service Design
New York State Office of MRDD
44 Holland Avenue
Albany, NY 12229-0001
Phone: (518) 474-4904
Fax: (518) 473-0054

George Jesien, Ph.D.
Executive Director
Association of University Centers of Disabilities
8630 Fenton Street, Suite 410
Silver Spring, MD 20910
Phone: (301) 588-8252
Fax: (301) 588-2842

H. A. Jinnah, M.D.
Associate Professor
Department of Neurology
The Johns Hopkins University
600 N. Wolfe Street, Meyer 56-181
Baltimore, MD 12187
Phone: (410) 614-6551
Fax: (410) 614-8585

Melinda Kelley, Ph.D.
Science Advisor
Office of Science Policy and Planning
National Institute of Neurological Disorders and Stroke
National Institutes of Health
31 Center Drive
Building 31, Room 8A03
Rockville, MD 20892
Phone: (301)435-7572
Fax: (301) 480-9172

Kenneth (Charlie) Lakin, Ph.D.
Director
Research and Training Center of Community Living
University of Minnesota
214 Pattee Hall
150 Pillsbury Drive, SE
Minneapolis, MN 55455
Phone: (612) 624-5005
Fax: (612) 625-6619

Bennett L. Leventhal, M.D.
Professor of Psychiatry and Pediatrics
Department of Psychiatry
The University of Chicago
5841 S. Maryland Avenue, MC 3077
Chicago, IL 60637
Phone: (773) 702-6751
Fax: (773) 702-6649

Andrew Levitas, M.D.
Associate Professor of Psychiatry
Director
Division of Prevention & Treatment
of Developmental Disorders
National Fragile X Foundation
University of Medicine & Dentistry
of New Jersey
40 East Laurel Rd. #200
Stratford, NJ 08084-1504
Phone: (856) 566-6034
Fax: (856) 566-6208

Catherine Lord, Ph.D.
Director
University of Michigan Autism and
Communications Disorders Center
University of Michigan
1111 E. Catherine Street
Ann Arbor, MI 48109-2054
Phone: (734) 936-8600
Fax: (734) 936-0068

Earl L. Loschen, M.D.
Chair
Department of Psychiatry
Southern Illinois University
School of Medicine
PO Box 19642
Springfield, IL 62794-9642
Phone: (217) 545-3847
Fax: (217) 545-2275

William E. MacLean, Jr., Ph.D.
Professor of Psychology
Department of Psychology
University of Wyoming
P.O. Box 3415
Laramie, WY 82071
Phone: (307) 766-2944
Fax: (307) 766-2926

James T. McCracken, M.D.
Professor and Division Director
Department of Child and Adolescent Psychiatry
University of California, Los Angeles
760 Westwood Plaza, Room 48-270
Los Angeles, CA 90024
Phone: (310) 825-0470
Fax: (310) 206-4446

Kathleen H. McGinley, Ph.D.
Director of Health and Housing Policy
The Arc of the United States
1730 K Street, NW, Suite 1212
Washington, DC 20006
Phone: (202) 785-3388
Fax: (202) 467-4179

Eve K. Mosciki, Sc.D., M.P.H.
Senior Advisor
Office of the Surgeon General
5600 Fishers Lane, Room 18-67
Rockvi11e, MD 20857
Phone: (301) 443-1773
Fax: (301) 443-3574

Richard K. Nakamura, Ph.D.
Deputy Director
National Institute of Mental Health
National Institutes of Health
6001 Executive Boulevard, Room 8235, MSC 9669
Bethesda, MD 20892-9669
Phone: (301) 443-3675
Fax: (301) 443-2578

Robert M. Nelson, M.D., Ph.D.
Associate Professor of Anesthesia and Pediatrics
Department of Anesthesia and Critical Care
Medicine, 9th Floor
The Children's Hospital of Philadelphia
34th Street and Civic Center Boulevard
Philadelphia, PA 19104-4399
Phone: (215) 590-9180
Fax: (215) 590-1415

Tan Nguyen, M.D., Ph.D.
Medical Officer
Office of Orphan Product Development
Food and Drug Administration
5600 Fishers Lane, Room 15A-08/HF-35
Rockville, MD 20857
Phone: (301) 827-0983
Fax: (301) 827-0017

Karen Anderson Oliver, Ph.D.
Clinical Epidemiology and Quality of Care Programs
National Institute of Mental Health
6001 Executive Boulevard, Room 7146, MSC 9631
Bethesda, MD 20892-9631
Use Rockville, MD 20852 for FedEx and UPS, etc.
Phone: (301) 443-3267
Fax: (301) 443-4045

David Patterson, Ph.D.
Chair, NDSS Science Advisory Board
President, Eleanor Roosevelt Institute
1899 Gaylord Street
Denver, CO 80206
Phone: (303) 336-5650
Fax: (303) 333-8423

Sussan Paydar, Ph.D.
Program Analyst
Neurogenetics Cluster
National Institute of Neurological Disorders and Stroke
National Institutes of Health
6001 Executive Boulevard, Room 2141
Bethesda, MD 20892
Phone: (301) 496-6012
Fax: (301) 402-1501

Audrey S. Penn, M.D.
Acting Director
National Institute of Neurological Disorders and Stroke
National Institutes of Health
Building 31, Room 8A52
Bethesda, MD 20892
Phone: (301) 496-3167
Fax: (301) 496-0296

James M. Perrin, M.D.
Professor of Pediatrics
Harvard Medical School
Center for Child and Adolescent Health Policy
Massachusetts General Hospital for Children
50 Stamford Street, #901
Boston, MA 02114
Phone: (617) 726-8716
Fax: (617) 726-7886

Siegfried Poeschel, M.D., Ph.D., J.D., M.P.H.
Professor of Pediatrics
Brown University School of Medicine
Rhode Island Hospital
593 Eddy Street
Providence, RI 02903
Phone: (401) 444-8477
Fax: (401) 444-6115

Sharon Ramey, Ph.D.
Director and Professor
Civitan International Research Center
University of Alabama, Birmingham
1719 6th Avenue, South, Suite 137
Birmingham, AL 35294-0021
Phone: (205) 934-8542
Fax: (205) 975-6330

William F. Raub
Deputy Assistant Secretary for Science Policy
DHHS/OSI ASPEI Science Policy
200 Independence Ave., S.W., Room 434E, MSC 445F
Washington DC 20201
Phone: (202) 690-5874
Fax: (202) 205-8835

Steven Reiss, Ph.D.
Professor
Nisonger Center
Ohio State University
868 Cherryfield Avenue
Columbus, OH 43210
Phone: (614) 292-2390
Fax: (614) 292-3727

William J. Rodriguez, M.D.
Science Director for Pediatrics
Pediatric Office CDER
Food and Drug Administration
9201 Corporate Boulevard, Room S410, HFD960
Rockville, MD 20850
Phone: (301) 827-2350
Fax: (301) 827-2520

(Helaine) Lainie Friedman Ross, M.D., Ph.D.
Associate Professor
Department of Pediatricsv University of Chicago
5841 S. Maryland Avenue, MC 6098
Chicago, IL 60637
Phone: (773) 702-6323
Fax: (773) 702-0090

Donald Routh, Ph.D.
Professor
Department of Psychology
University of Miami
PO Box 249229
Coral Gables, FL 33124
Phone: (305) 284-5222
Fax: (305) 284-4795

Judith Rumsey, Ph.D.
Developmental Neuroscience of Schizophrenia,
Mood and Other Brain Disorders
National Institute of Mental Health
National Institutes of Health
6001 Executive Boulevard, Room 7175
Bethesda, MD 20892-9633
Phone: (301) 443-9264

Sir Michael Rutter, M.D.
Professor
SGDP Research Centre
Institute of Psychiatry, Denmark Hill
London, England SE5 8AF
United Kingdom
Phone: 44 0 20 7848 0882
Fax: 44 0 20 7848 0881

Ron Schoenfeld, Ph.D.
Senior Advisor
Office of the Surgeon General
5600 Fishers Lane, Room 18-66
Rockville, MD 20857
Phone: (301) 443-2132

Stephen Schroeder, Ph.D.
Director
Association of University Centers on Disability
University of Kansas
1000 Sunnyside Avenue, 1052 Dole
Lawrence, KS 66045
Phone: (785) 864-4295
Fax: (785) 864-5323

Nichelle Schoultz
Grassroots Coordinator
Government Affairs
Epilepsy Foundation
4351 Garden City Drive
Landover, MD 20785-7223
Phone: (301) 459-3700
Fax: (301) 577-2684

Marsha M. Seltzer, Ph.D.
Acting Director
Waisman Center
University of Wisconsin-Madison
1500 Highland Avenue
Madison, WI 53705
Phone: (608) 263-5940
Fax: (608) 263-3300

Eunice Kennedy Shriver
Executive Vice-President
The Joseph P. Kennedy, Jr. Foundation
1325 G. Street, NW, Suite 500
Washington, DC 20005-3104
Phone: (202) 393-1250

Sargent Shriver
Chairman of the Board
Special Olympics, Inc.
1325 G Street, N.W., Suite 500
Washington, DC 20005-3104
Phone 202-824-0242
Fax 202-628-0067

Robert L. Sprague, Ph.D.
Professor Emeritus
Department of Psychology, Kinesiology
& Special Education
University of Illinois
1306 Old Farm Road
Champaign, IL 61821-5940
Phone: (217) 398-6539
Fax: (217) 356-2080

Matthew W. State, M.D., Ph.D.
Assistant Professor of Child Psychiatry
Director, Section on Child Psychiatric Genetics
Child Study Center
Yale University School of Medicine
230 South Frontage Road
New Haven, CT 06520
Phone: (203) 737-4342
Fax: (203) 785-7611

Sue Swenson
Executive Director
The Joseph P. Kennedy Jr. Foundation
1325 G Street, NW, Suite 500
Washington, DC 20005
Phone: (202) 393-1250
Fax: (202) 824-0351

Ludwik S. Szymanski, M.D.
Director of Psychiatry (Emeritus)
Institute for Community Inclusion
Associate Professor of Clinical Psychiatry
Children's Hospital Boston
Harvard Medical School
53 West Boulevard Road
Newton, MA 02459
Phone: (617) 969-2839
Fax: (617) 559-0845

Elaine Tierney, M.D.
Director of Psychiatry
Department of Psychiatry
Kennedy Krieger Institute
391 Greenspring Avenue
Baltimore, MD 21211
Phone: (443) 923-7657
Fax: (443) 923-7695

Marcia Van Riper, Ph.D.
Genomics Institute and School of Nursing
University of North Carolina at Chapel Hill
Carrington Hall CB#7460
Chapel Hill, NC 27599
Phone: (919) 966-5329
Fax: (919) 843-9969

Vicky Whittemore, Ph.D.
Senior Scientific Advisor and Director
Center Without Walls
National Tuberous Sclerosis Association
P.O. Box 3784
Crofton, MD 20910
Phone: (410) 721-2615
Fax: (410) 721-2815

Lucille A. Zeph
Director
Center for Community Inclusion
University of Maine
5717 Corbett Hall
Orono, ME 04469
Phone: (207) 581-1084
TTY: (207) 581-3328
Fax: (207) 581-1231

Ann E. Wagner, Ph.D.
Chief, Autism/PDD
Intervention Research Program
Division of Services and Intervention Research
National Institute of Mental Health
National Institutes of Health
6001 Executive Boulevard, Room 7149
Bethesda, MD 20892-9633
Phone: (301) 443-4283
Fax: (301) 443-4045

Organizing Committee

Felix de la Cruz, M.D., M.P.H.
Chief, Mental Retardation and Developmental
Disabilities Branch (CRMC)
National Institute of Child Health and Human Development
National Institutes of Health
6100 Executive Boulevard, Room 4B09
Bethesda, MD 20892
Phone: (301) 496-1383
Fax: (301) 496-3791

James C. Harris, M.D.
Director, Developmental Neuropsychiatry
Professor of Psychiatry and Pediatrics
Johns Hopkins University School of Medicine
CMSC 341 Johns Hopkins Hospital
600 N. Wolfe Street
Baltimore, MD 21287
Phone: (410) 955-6181
Fax: (410) 955-8691
Fax: (410) 889-5623

Bryan King, M.D.
Professor of Psychiatry and Pediatrics
Director of Child and Adolescent Psychiatry
Dartmouth Medical School
One Medical Center Drive
Lebanon, NH 03756
Phone: (603) 650-7229
Fax: (603) 650-5842

Mark H. Lewis, Ph.D.
Professor, Department of Psychiatry
University of Florida
P.O. Box 100256
Gainesville, FL 32610
Phone: (352) 392-3471
Fax: (352) 392-2579

Edgardo J. Menvielle, M.D.
National Institute of Mental Health/DSIR
6001 Executive Boulevard, Room 7149
Bethesda, MD 20892
Phone: (301) 443-3815
Fax: (301) 443-4045

Paul L. Nichols, Ph.D.
Program Director
Systems and Cognitive Neuroscience
National Institute of Neurological Disorders and Stroke
Neuroscience Center, Room 2108
6001 Executive Blvd
Bethesda, MD 20892
Phone: (301) 496-9964
Fax: (301) 402-2060

Quandra Scudder
Program Analyst
Neurogenetics Cluster
National Institute of Neurological Disorders and Stroke
Neuroscience Center
6001 Executive Boulevard, Room 2145
Bethesda, MD 20892-9525
Phone: (301) 496-5745
Fax: (301) 402-1501

Giovanna Spinella, M.D.
Program Director, Neurogenetics and Development
National Institute of Neurological Disorders and Stroke
Neuroscience Center
6001 Executive Boulevard, Room 2132
Bethesda, MD 20892-9527
Phone: (301) 496-5745
Fax: (301) 402-1501

Travis Thompson, Ph.D.
Smith Professor of Psychiatry
Director, Institute for Child Development
University of Kansas Medical Center
3901 Rainbow Boulevard
Kansas City, KS 66160
Phone: (913) 588-5588
Fax: (913) 588-5916

Benedetto Vitiello, M.D.
Chief, Child and Adolescent Treatment and
Preventive Intervention Research Branch
Division of Services and Intervention Research
National Institute of Mental Health
6001 Executive Boulevard, Room 7147
Bethesda, MD 20892-9633
Phone: (301) 443-4283
Fax: (301) 443-4045

Tarsha M. McCrae
Program Staff Neurogenetics
National Institute of Neurological Disorders and Stroke
6001 Executive Boulevard, Room 2132
Bethesda, MD 20892-9527
Phone: (301) 496-5745
Fax: (301) 402-1501


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Summary

Individuals with mental retardation and developmental disabilities are estimated to be 3-4 times more likely than those in the general population to experience an emotional, behavioral, or psychiatric disorder. Recent advances in a number of fields and disciplines–including the neurosciences, genetics, psychopharmacology, developmental neuropsychiatry, psychology, and education--show promise for improving the treatment and lives of those with mental retardation and developmental disabilities. Despite advances in each of these areas, mental retardation is frequently a criterion for exclusion from research studies. Enrollment of individuals with mental retardation in research protocols addressing emotional and behavioral disorders has been limited, issues of informed consent persist, and more researchers with an interest and expertise in this population are needed.

To address these issues, a two-and-a-half-day Workshop was convened by the National Institute of Neurological Disorders and Stroke (NINDS), the National Institute of Child Health and Human Development (NICHD), the National Institute of Mental Health (NIMH), the National Institutes of Health (NIH) Office of Rare Diseases, and the Joseph P. Kennedy Jr. Foundation.

The Workshop, titled “Emotional and Behavioral Health in Persons with Mental Retardation/Developmental Disabilities: Research Challenges and Opportunities," drew 97 participants, from academic research institutions, government agencies, service providers, and consumer advocacy organizations. (A copy of the meeting agenda and participant list is attached.)

The Workshop was designed to identify barriers to the inclusion of people with mental retardation and developmental disabilities in federally funded research in the United States. The goal was to define ways to increase inclusion of people with mental retardation in research in order to promote evidenced-based treatment for this population.

The Workshop was held several days prior to a Surgeon General’s Conference on Health Disparities and Mental Retardation, December 5-6, 2001 so that results of this Workshop could be presented. (To view webcasts of the Workshop and the Surgeon Generals Conference, visit the NIH web site at http://videocast.nih.gov and click on Past Events, then Conferences.)

Opening Remarks and Overview Presentations

The meeting began with welcoming messages from leaders of the sponsoring NIH Institutes:

  • Audrey Penn, M.D., Acting Director of NINDS noted that three NIH Institutes were represented and the Office of Rare Diseases, but there easily could have been a dozen, given the breadth of the subject matter. She said that NINDS neuroscientists share with their NIH colleagues an interest in the developmental neurobiology, genetics, and cognitive aspects of syndromes associated with mental retardation, and she expressed eagerness to review all that Workshop participants brought to the table.

  • Duane Alexander, M.D., Director of NICHD, said that the conference provided an opportunity to take stock of what research has been done, what has been learned, what has been learned but has not been applied, and what important issues remain to be addressed. He said the speaker roster read like an “Olympics of champion investigators” in this field, and he looked forward to reviewing and implementing the recommendations that emerged from the Workshop.

  • Richard Nakamura, Ph.D., Deputy Director of NIMH, said that the emotional and behavioral health of those with mental retardation and developmental disabilities has been a neglected subject. He said NIMH recognizes that it has a special duty to help foster research that will advance the health and lives of those with mental retardation and developmental disabilities, and the Institute aims to fully honor that commitment.
Eunice Kennedy Shriver, Executive Vice President of the Kennedy Foundation, issued a call to action to the group to work to ensure that the more than seven million people in the United States with mental retardation and developmental disabilities have the mental health supports and services they need throughout their lives. She noted that:
  • Some 20-30 percent of people with mental retardation and developmental disabilities suffer from a psychiatric disorder, yet services are organized as if people have either mental retardation or mental illness--but not both.

  • People with all levels of disability experience a full range of mental health challenges, yet 39 percent of psychiatrists admit they prefer not to treat people with mental retardation and developmental disabilities.

  • Children with mental retardation and developmental disabilities who are depressed are less likely to get the help they need in schools, but unless their behavior is disruptive, they are likely to be ignored or removed from class for poor performance.
To improve mental health supports to every American who has mental retardation, and their families, we must focus on research and training,” she said. “But over the next few days, do not forget that people need services!” For full text, please see Mrs. Eunice Kennedy Shriver's Remarks on Mental Health and Mental Retardation.

Following the opening remarks, leaders in the field provided overviews on research opportunities in genetics, neuroimaging, brain plasticity, and psychosocial issues. Subsequent overview presentations provided background in six key areas related to the emotional and behavioral health of individuals with mental retardation: (1) epidemiology, (2) diagnosis/assessment, (3) interventions, (4) ethical issues, (5) research design, and (6) research training needs.

Working Groups met later to identify critical issues in each of these areas and craft recommendations, which were presented to the full group of Workshop participants for discussion. Recommendations from the Working Groups are being reviewed by the three sponsoring NIH Institutes (NINDS, NICHD, NIMH) and appropriate action will be taken to move toward their implementation.

Working Group Discussions

Each of the Working Groups met on the second day of the Workshop to consider a number of questions, including:

  • Epidemiology--What epidemiologic evidence is needed to provide accurate measures of the prevalence and incidence of behavioral, emotional, and psychiatric problems; to identify risk and protective factors; and to establish the natural history of these conditions? Which designs, research strategies, and approaches should be considered?

  • Diagnosis and Assessment--What are the appropriate instruments needed to diagnose and formulate treatment for individuals with mental retardation and developmental disabilities? How can current diagnostic and assessment methods be made more applicable to individuals with mental retardation and developmental disabilities?

  • Ethical Considerations--How should “consent” and “assent” apply to research involving individuals with mental retardation and developmental disabilities, particularly research with no immediate therapeutic benefit? Should the standard for surrogate decision-making regarding the participation in research (both when to permit it and the standards for its exercise) be the same for children and adults where there are questions about their ability to make decisions?

  • Interventions Research--What are the most pressing needs of people with mental retardation and developmental disabilities and cooccurring emotional and behavioral disturbances? How might interventions research be most effectively carried out in hospitals, schools, and community settings?

  • Research Design--What are the most pertinent research designs to conduct research involving people with mental retardation and developmental disabilities and cooccurring emotional and behavioral disturbances? What steps should be taken to adapt FDA guidelines for clinical medication trials in individuals with mental retardation and developmental disabilities?

  • Research Training Needs--What are the best approaches to establishing new researchers with an interest in the emotional and behavioral health of people with mental retardation and developmental disabilities? Are there useful models from other areas of biomedical research that can be used?

Working Group Recommendations

Epidemiology

The Working Group on Epidemiology noted that epidemiology is the study of the distribution and dynamics of health and illness in human populations. An important aspect of epidemiology is research on the functioning of health services. Epidemiology is more than simply counting how many individuals are affected and measuring prevalence and demography. Epidemiology can teach us about the nature and scope of mental retardation/developmental disabilities and the associated behavioral, emotional, and psychiatric problems. Modern experimental approaches to epidemiology allow the study of causative processes, factors that influence the course of the disorder, and service needs. Epidemiological methods often are combined with other methods (for example, psychosocial and neurobiological measures). Epidemiological studies can disclose individual developmental trajectories and the influences that shape those trajectories. Some of these influences promote risk; others provide protection and promote resiliency.

Although we have an approximate understanding of the prevalence of emotional and behavioral disturbances in individuals with mental retardation, there are almost no studies on service utilization. We must ask which new data are needed in order to design and conduct research aimed at improving the functioning of persons with mental retardation?

Critical Issues

  • There is a lack of adequate data on risk and protective factors for mental illness in people at different developmental stages, including preschool, school entry, school-to-work transition, and aging.

  • New opportunities are available to study the interaction between genes and the environment. It is expected that variations in the heritability of behavioral or cognitive traits will be observed across different social levels and environmental conditions.

  • Issues in sampling and measuring behavior and related characteristics, including adaptative behavior, are critically important.

  • Ensuring adequate sample sizes is paramount. For example, many individuals with mild mental retardation may be difficult to locate beyond the school years. In addition, recruiting adequate numbers of individuals with more rare neurogenetic syndromes may be difficult.

  • There is a need to examine behavioral features, emotional problems, and psychiatric diagnoses in groups with specific neurodevelopmental syndromes, including Down syndrome, Fragile X, and Prader Willi syndrome.

  • Systems research studies are needed, including investigations focusing on state systems, residential units, group homes, supported living settings, and prisons. Understudied areas include: (1) factors leading to the success or failure of deinstitutionalization and (2) the probable increase in use of medications during the last decade.

Recommendations

  • Conduct longitudinal studies to examine key life-stage transitions regarding risk and protective factors. Comparisons should be made of such factors as time of diagnosis of the disorder, residential status, and family functioning. Although the Working Group focused primarily on concurrent longitudinal designs in its recommendations, the group also recognized the utility of case-control designs for behavioral genetics research and non concurrent longitudinal designs that make use of record linkages.

  • Use informative samples and innovative research designs for behavioral genetic research, including twin pairs studied from birth and informative family and sibling studies.

  • Develop and refine an array of measures of outcomes and hypothesized risk and protective factors.

  • Ensure that those with mild mental retardation or rare neurogenetic disorders are sampled adequately. Exploit opportunities to piggyback on existing surveys, such as the CDC health risk behavior surveys and the National Health Interview Survey.

  • Conduct syndrome-specific cohort studies to: ascertain specific vulnerabilities to common disorders; inform analysis of interactions between genes, brain, and behavior; and improve understanding of functioning.

  • Conduct system research studies that target state systems, schools, residential units, group homes, supported living settings, and prisons.

  • Consider convening another Workshop to address the need for systems research studies.

Diagnosis and Assessment

The Working Group on Diagnosis and Assessment noted that psychiatric disorders have a profound effect on the well being of people with mental retardation and developmental disabilities. The prevalence of emotional and behavioral disorders among this group is greater than in the general population, and the consequences of such disorders are more severe, through their impact on careers (both family and professional care-providers), and because they can prevent those affected with these disorders from being included in family and community life. As a result, the cost to the nation is great.

A major impediment to our ability to treat these disorders effectively in people with mental retardation and developmental disabilities is our inability to recognize mental health problems in these individuals. Assessment and diagnosis of emotional and behavioral disturbance are particularly difficult due to intellectual, adaptive, and verbal impairments that limit reliability or reporting, and the presence of organic or environmental factors that either produce or exacerbate the specific forms of pathologic behavior. Receipt of services is also frequently linked to the diagnosis of a specific condition or the severity of functional impairment related to it.

As a consequence of difficulties with diagnosis and assessment, persons with developmental disorders may not receive effective treatments, the treatment they receive may actually be harmful, and they may be denied important services. Additionally, the lack of diagnostic tools holds back basic research into the etiology of mental retardation, without which we cannot expect to develop effective rational therapies.

Critical Needs

  • Research aimed at developing appropriate diagnostic procedures applicable to individuals with mental retardation and developmental disabilities.

  • Investigations to evaluate the clinical utility of assessment procedures in treatment trials.

  • Studies to identify the biological and environmental determinants of emotional and behavioral disorders in individuals with mental retardation and developmental disabilities.

  • Research to determine whether differences in the expression of emotional disorders are due to cognitive or functional impairment. The complex interrelationship between the expression of emotional disorders and cognitive and functional impairment places additional demands on the adequacy of assessment procedures.

Recommendations

  • Conduct research to assess the validity and reliability of adaptations of standard diagnostic and assessment strategies to accommodate people with mental retardation and developmental disabilities.

  • Develop direct observation instruments to identify overt characteristics of emotional disturbance and their environmental correlates.

  • Undertake studies to evaluate the tolerability of the diagnostic and assessment process for people with mental retardation and developmental disabilities and their families and care providers.

  • Develop techniques to assess the impact of psychosocial stressors in the lives of people with mental retardation and developmental disabilities and to integrate this knowledge with diagnostic protocols, treatment strategies and service systems. It is equally important to determine protective factors and methods to quantify an individual’s resilience.

  • Undertake efforts to characterize the phenotypic diversity of mental retardation and developmental disabilities. Develop appropriate animal models to explore how genetic abnormalities give rise to mental retardation. In addition, complete neuropsychological assessments of mental retardation and developmental disabilities to determine the pattern of cognitive disabilities and competencies and associated behavioral abnormalities present in various forms of mental retardation and developmental disabilities.

  • Conduct prospective studies to describe the developmental trajectory of behavior and skill acquisition in genetic disorders or syndromes associated with mental retardation and developmental disabilities. While it is often assumed that the pattern of abilities and impairments will be maintained throughout development, available evidence suggests this is not so.

  • Carry out research aimed to uncover how the manifestations of particular emotional and behavioral disorders and the response to treatment may vary as a function of cognitive or functional disability or developmental level.

    Ethical Considerations

    The Working Group on Ethical Considerations identified a wide variety of ethical and legal issues surrounding the participation of people with mental retardation and developmental disabilities in research protocols. The members affirmed that well designed nontherapeutic research serves the interests of individuals with mental retardation and developmental disabilities. When there is no effective treatment available, it is in the interest of people with mental retardation and developmental disabilities to participate in well designed placebo-controlled trials. The alternative is not to receive treatment for disabling conditions, or to receive treatments where safety and efficacy have not been studied, often resulting in harm without compensating benefit. Any determinations about an individual’s capacity to decide whether to participate in research should be based on his or her functional ability, not on IQ score or the simple label of mental retardation and developmental disabilities.

    Critical Issues

    • The “Common Rule” governing the participation of human subjects in federally sponsored research does not adequately address the inclusion of individuals with mental retardation and developmental disabilities. It remains unclear whether separate provisions are necessary for individuals with mental retardation and developmental disabilities, mental illness, or impaired decisional capacity.

    • The “minimal risk rule” guiding participation in nontherapeutic research is ambiguous and is being interpreted widely by institutional review boards (IRBs). Considerable variability is apparent in how IRBs determine the risk level in “ordinary” life, what medical tests are viewed as “routine,” and whether such factors should be indexed to people who are healthy or those who are ill.

    • The current understanding of informed consent and assent frequently does not take into consideration the varied capacities of individuals with mental retardation and developmental disabilities to make decisions. Decisional capacity in adults with mental retardation and developmental disabilities often is assumed to be global, across all decision-making tasks, which is contrary to available evidence.

    • Individuals with mental retardation and developmental disabilities may be more or less vulnerable to undue influence and/or coercion depending on such factors as their residential setting or the absence or presence of an appropriate advocate.

    • Fear of federal sanctions is restraining IRBs from approving ethically acceptable research, particularly in vulnerable populations. Similarly IRB members and investigators express growing concerns about litigation. Punitive sanctions are frequently levied by federal oversight bodies for regulatory compliance infractions that are not related to harm to research subjects.

    • Lack of clarity in guidelines may impede genetic testing of individual with mental retardation and developmental disabilities. There is no consensus on whether or when family members should be considered research subjects if mentioned in the course of a genetic family history. The authority of surrogate decision-makers to consent for genetic research on adults with decisional incapacity with no direct therapeutic benefit to the subject remains unclear.

    Recommendations

    • The Institute of Medicine should convene a meeting of all stakeholders to focus on problems in the Common Rule with regard to individuals with mental retardation and developmental disabilities, mental illness, or impaired decisional capacity in federally sponsored research.

    • This group should address the minimal risk rule as it applies to individuals with mental retardation and developmental disabilities who lack capacity to make decisions regarding research participation. The group should also address federal guidelines that affect genetic family histories--specifically, whether a family history that was obtained as part of a research study necessarily makes a family member a “research subject.” This group should also address when nontherapeutic genetic testing for research purposes is permissible for individuals with mental retardation and developmental disabilities.

    • Further analysis is needed concerning issues of assent and dissent in individuals with mental retardation and developmental disabilities. Individuals with mental retardation and developmental disabilities who have the capacity to consent should be given the widest latitude to consent or refuse to participate in research. If an individual with mental retardation and developmental disabilities appears unwilling to participate in research, dissent should generally be honored, even for procedures of minimal risk. Analysis is needed about the relevance of objection in individuals with limited language ability, including infants and severely impaired older children and adults.

    • Appropriate tools for the assessment of decisional capacity in individuals with mental retardation and developmental disabilities need to be developed. Further research is needed on techniques to enhance the decision-making capabilities of individuals with mental retardation and developmental disabilities.

    • Continued discussion is needed on the legal authority of surrogate decision makers to authorize participation in research that does not offer the prospect of direct benefit to the subject. Because decisional capacity is situational in individuals with mental retardation and developmental disabilities, appointment of a surrogate for all decision-making may be inappropriate. Absent state law to the contrary, the authority of a surrogate to authorize research that does not have the prospect of direct benefit to the person with mental retardation and developmental disabilities should be clarified.

    • Additional discussion is needed on the role of the residential setting and associated factors in research protections. Issues of home ownership/control and contracting for services within the residential setting are a necessary part of the discussion.

    • Federal oversight bodies, such as the Office of Human Research Protection (OHRP) and the National Human Research Protections Advisory Committee (NHRPAC), should review the appropriateness of punitive sanctions for infractions that have little apparent relevance to protection of research subjects.

    Interventions Research

    The Working Group on Interventions Research said that advances in the fields of genetics, neuroimaging, brain plasticity, behavioral sciences, and education show promise in generating effective interventions to improve the emotional and behavioral health of individuals with mental retardation and developmental disabilities. The group noted that there are a number of behavioral and psychosocial treatments that have been well researched in the general population, but have not been tested for their efficacy in individuals with mental retardation and developmental disabilities. Similarly, newer and safer medications are available for a range of emotional and behavioral disorders, yet few rigorous tests of their efficacy in individuals with mental retardation and developmental disabilities have been performed. These and other challenges present critical opportunities for well-designed intervention studies in people with mental retardation and developmental disabilities.

    Critical Opportunities

    • In genetics, characterization of specific behavioral phenotypes associated with varied genetic etiologies of mental retardation and developmental disabilities show promise in allowing researchers to craft and target interventions for specific disorders or behavioral disturbances. Research identifying genes that code for new target proteins and the development of drugs that target those proteins could lead to better focused pharmacologic treatment strategies.

    • In neuroimaging, scientists have begun to document the effects of environment and experience on the brains of individuals with mental retardation and developmental disabilities using a variety of imaging techniques.

    • Research on the plasticity of the brain, much of it accomplished in animal models, has illustrated the profound effects of early experience on brain development and development of aberrant behavior. Research has shown that the consequences of central nervous system damage, even as a result of genetic mutations, can be ameliorated by complex environments. Evidence is also accumulating that neural regeneration, in at least some areas of the brain, is possible across the life span.

    • In psychosocial research, functional analysis of behavior has been shown essential for prescribing effective behavioral treatments for people with mental retardation and developmental disabilities who display severe behavior disorders. Communication training has been shown to reduce the occurrence of emotional and behavioral problems. Intensive early educational interventions produce long-term gains in social and intellectual functioning in some at-risk populations.

    Recommendations

    • In genetics, assess the differential effects of genetic etiology on treatment outcomes. Develop psychotropic drugs selective for newly identified target proteins and test in animal models and controlled clinical trials. Test the effects of experiential/behavioral manipulations on gene expression.

    • Use neuroimaging as a technique for assessing treatment outcome (along with behavioral and clinical outcomes). Use neuroimaging to establish likely mechanisms of treatment effects.

    • In neuroplasticity research, test theory-based interventions for specific populations, using an array of new technologies to document central nervous system changes as well as behavioral outcomes. Evaluate importance of age, timing, and intensity of intervention on neuroplasticity. Utilize appropriate animal models to test neuroplasticity associated with developmental insults to the central nervous system.

    • In psychosocial research, evaluate the impact of using functional analysis to test the efficacy of different treatment techniques and in a wider variety of emotional and behavioral disorders. Test the efficacy of intensive early intervention and communication training to prevent or reduce emotional or behavioral disorders in children with mental retardation and developmental disabilities.

    • Assess the effects of combining pharmacological treatments with behavioral, psychosocial, and educational interventions and/or natural supports.

    • Develop innovative research designs to address the potential confounds of ongoing treatments and cooccurring conditions.

    • Urge revision of Food and Drug Administration drug approval standards to include alternative research designs.

    • Engage consumers and family members in design, implementation, and evaluation of interventions.

    • Create a federal task force to develop and implement an interdisciplinary clinical research network. Fund meetings to create coalitions of researchers, advocates, and service providers.

    • Place funding priority on testing promising interventions. Review portfolio of NIH-funded studies that address prevention or treatment of emotional and behavioral disorders. Determine the feasibility of supplemental funding to add participants with mental retardation and developmental disabilities.

    Research Design

    The Working Group on Research Design noted that the primary purpose of research design is the production of unbiased and efficient data that can be used to serve the needs of all partners in the research enterprise. The group affirmed that while a multiplicity of research designs and analyses can be valid for addressing specific problems and questions in individuals with mental retardation and developmental disabilities, the randomized, controlled, blinded protocol remains the “gold standard” against which other research designs should be judged. In current practice, research on individuals with mental retardation and developmental disabilities frequently relies on single-subject case reports, which do not uniformly produce optimal, valid answers. Through more informed decision-making about research designs and their fit to research questions, the state of knowledge about the emotional and behavioral health of individuals with mental retardation and developmental disabilities could be improved.

    Recommendations

    • It should be the normal expectation that individuals with mental retardation and developmental disabilities will be included in federally funded research. The investigator must assume the responsibility for justifying any exclusionary rules. Exclusions should be based on the functional and safety features of the experiments or interventions being studied, not sweeping criteria, such as “IQ less than 70,” that frequently exclude many individuals with mental retardation and developmental disabilities from research.

    • Research designs that emphasize our understanding of “for which person” and “under what circumstances” should be emphasized in research on mental retardation and developmental disabilities. These research designs are preferred to models that only yield estimates of group means, such as ANOVA and regression models.

    • Multi-site, multi-collaborator designs should be the norm, not the exception. These designs need to be utilized creatively and not limited to single protocols that are simply replicated at all sites.

    • Research designs used to assess the outcomes of interventions in individuals with mental retardation and developmental disabilities should include measures that assess functional and clinical improvement. In general, a single variable, or a small number of variables, should be pre-specified as the outcome variable of interest.

    • Longitudinal follow-up is critical for a full understanding of the consequences of an intervention. Longitudinal follow-up should be pre-planned and examine both the primary outcome variable and other potential mediating variables.

    • Creating an archive of complete and well-documented data sets is essential for the development of cumulative science. Federally supported researchers should expect to make their data available to the research community after an appropriate delay for publication of the study’s primary outcomes.

    • For clinical studies designed to elaborate new therapeutic applicability of a drug, early intervention with the FDA is essential.

    Research Training Needs

    The Working Group on Research Training Needs emphasized that the training of investigators with an interest in the emotional and behavioral health of individuals with mental retardation and developmental disabilities should draw on knowledge in a wide variety of disciplines. Efforts should be collaborative, emphasizing training, research, and clinical work--with a particular focus on translational research that bridges basic science and more applied investigations. Initiatives to train more researchers in this area face a number of impediments, including stigma (for example, the notion that individuals with mental retardation and developmental disabilities are “difficult to treat”) research in this area is frequently viewed as less prestigious or “real” compared with other fields; and disciplinary “silos” maintain barriers to the cross-disciplinary collaboration necessary in this research and essential to attracting new investigators to the field.

    Critical Issues

    • There is no clear home for grants supporting research on the emotional and behavioral health of individuals with mental retardation and developmental disabilities. Individual Research Career Development (K) awards may come too late in medical careers and provide insufficient research and salary support in the face of significant debt. Research into the emotional and behavioral health of people with mental retardation and developmental disabilities is not well regarded or understood by funding review groups.

    • Research training opportunities can come too late in clinical training. Opportunities can be provided at the undergraduate level, during graduate/medical training, and during the residency/post-doctoral fellowship period. Junior faculty need forums to present their work, get feedback, and build networks. Trainees at all levels lack sufficient exposure to individuals with mental retardation and developmental disabilities and co-occurring emotional and behavioral problems.

    • Junior faculty face pressure to publish discrete inquiries in which they retain lead authorship. Tenure committees frequently do not assign significant value to participation in large, interdisciplinary investigations characteristic of work in this area.

    • There is insufficient funding for seasoned investigators to mentor those in training or young investigators. Not all mentors are “good” mentors; it is difficult to get good trainees to the right mentor.

    Recommendations

    • Encourage a variety of training pathways and mechanisms for different individuals and disciplines. Systematically evaluate all current research training mechanisms, seeking feedback from junior trainees who have successfully entered research careers and those who have chosen other career paths.

    • Consider which other disciplines should be at the table. Special education; speech, language, and communication; social work; nursing; and occupational therapy are among the fields that could contribute to this discussion.

    • Look to program models in other areas to develop new training mechanisms. Work group members highlighted the NINDS Neurological Sciences Academic Development Award and the NICHD Pediatric Scientist Development Training Program as examples.

    • Augment existing support and seek new avenues to channel support. Increase stipend and research dollar funding for training MD clinical investigators (the NIH T32 program). Add other training funds to University Centers for Excellence in Developmental Disabilities (UCDDs), formerly known as University Affiliated Programs (UAPs) on Developmental Disabilities and other centers where research on mental retardation and developmental disabilities is performed. Craft a specific request for applications (RFA) to encourage research on the emotional and behavioral health of people with mental retardation and developmental disabilities. Develop new support for research training during medical residency and the post-residency fellowship years.

    • Create an NIH inter-institute coordinating group to promote research and training on the emotional and behavioral health of people with mental retardation and developmental disabilities. Designate a program officer at each relevant Institute to shepherd research in this area. Create special review groups for research training proposals and develop intramural programs for research on the emotional and behavioral health of individuals with mental retardation and developmental disabilities.

    • Create training centers of excellence in mental retardation and developmental disabilities research to promote the value of training efforts.

    • Support and augment existing research training networks that could expand training of researchers in mental retardation and developmental disabilities. These include the UCDDs, the Mental Retardation Research Centers funded by NICHD, as well as the Leadership Education in Neurodevelopmental and Related Disabilities program (LEND) funded by the Maternal Child Health Bureau.

    • Create topical career development clubs for young investigators. Use these as a vehicle to encourage early career mentoring, network building, and as a source of information about available funding opportunities.

    • Create new categories of special individual research career development awards (“special K’s”) for investigators at various stages of their careers. Craft a special-K RFA for researchers interested in the emotional and behavioral health of people with mental retardation and developmental disabilities.

    • Seek private foundation partnerships.

    • Develop a plan to monitor progress from the outside. The Institute of Medicine represents one venue with experience in charting change and setting priorities.
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    Last Reviewed: February 11, 2004
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