Executive Function in Probands With Autism With Average IQ and Their Unaffected First-Degree Relatives - 23/08/14
Abstract |
Objective |
This study aimed to characterize executive function (EF) in pedigrees of children with autism spectrum disorder (ASD) and average IQ. The authors examined the hypothesis that deficits in EF relate to lower levels of adaptive functioning, and they assessed evidence for a cognitive extended phenotype in unaffected relatives in a large, well-characterized sample.
Method |
Proband EF was assessed by parent-report questionnaires (Behavior Rating Inventory of Executive Functioning [BRIEF], n = 109) and child neuropsychological tests (Delis-Kaplan Executive Functioning System [D-KEFS], n = 35). EF also was examined in parents (D-KEFS, n = 335) and unaffected siblings (BRIEF, n = 114; D-KEFS, n = 57). Adaptive functioning was assessed by the Vineland Adaptive Behavior Scales–II (n = 155). All data were obtained from the Autism Consortium Clinical Genetics Database.
Results |
Individuals with ASD showed important EF weaknesses. Multiple regression analyses showed that parent-reported EF deficits were related to profound decreases in adaptive functioning even after controlling for age, IQ, and severity of ASD symptoms. Parent-reported EF also was related to adaptive skills in preschoolers. First-degree unaffected relatives did not demonstrate difficulties with EF compared with normative data.
Conclusion |
In this study, EF impairments do not appear to relate to broad familial risk factors for ASD but may be associated with factors relevant to the expression of ASD in probands. Results support the benefits of EF assessment as a way to identify potential therapeutic targets that could lead to improved adaptive behavior in children with ASD and average IQ.
Le texte complet de cet article est disponible en PDF.Key Words : autism spectrum disorder, executive functioning, adaptive functioning, IQ, pedigree
Plan
| Clinical Guidance is available at the end of this article. |
|
| This article was reviewed under and accepted by deputy editor Stephen V. Faraone, PhD. |
|
| This research was supported by Dr. Morrow’s Career Award in Medical Science from the Burroughs Wellcome Fund and the National Institutes of Health (NIH) / National Institute of General Medical Sciences (NIGMS) grant P20GM103645 01A1: Project Award under Neuroscience COBRE Project. The collection of data and biomaterials comes from the Phenotypic and Genetic Factors in Autism Spectrum Disorders Study. Since 2008, this project has been supported by the Autism Consortium and by NIMH grants (1R01MH085143, principal investigator, Louis M. Kunkel, PhD; 1R01MH083565, principal investigator, Christopher Walsh, MD, PhD). |
|
| The study was conducted through a collaborative network of 5 hospitals: Boston Children’s Hospital (BCH), the Lurie Family Autism Center at Massachusetts General Hospital (MGH), The Floating Hospital for Children at Tufts Medical Center, Boston Medical Center (BMC), and University of Massachusetts Medical Center. The principal investigators are Christopher Walsh, MD, PhD, and Louis M. Kunkel, PhD, at BCH and Susan Santangelo, ScD, at MGH. Co-investigators at the participating sites include Ingrid A. Holm, MD, MPH, Leonard Rappaport, MD, MS, and Ellen Hanson, PhD (BCH); Elizabeth Caronna, MD, and Marilyn Augustyn, MD (BMC); Ann Neumeyer, MD, and Patricia Davis, MD (MGH); Karen Miller, MD, and Laurie Demmer, MD (Tufts); and Jean Frazier, MD (University of Massachusetts). The authors are grateful to the families who participated in this study and made the authors’ contribution to this repository possible. |
|
| Disclosure: Dr. Johnson Harrison has received support from NIMH. Dr. Joseph has received support from NIMH, the National Institute of Child Health and Human Development/National Institute of Deafness and Other Communication Disorders, and the National Institute of Neurological Diseases and Stroke. Dr. Morrow has received awards and/or funding from NIH/NIMH, NIH/National Center for Research Resources under Perinatal Medicine COBRE, Rhode Island Hospital, Brown University, the Simons Foundation Autism Research Initiative, and the Society of Biological Psychiatry. He holds a patent for Methods for Treatment of Microcephaly-Associated Autism Disorders (US Patent Application Number 61/739,351; International Application Number PCT/US2013/076609). Drs. McLean and Zimak report no biomedical financial interests or potential conflicts of interest. |
Vol 53 - N° 9
P. 1001-1009 - septembre 2014 Retour au numéro
Article précédent
- Subthreshold Psychotic Symptoms in 22q11.2 Deletion Syndrome
- Sunny X. Tang, James J. Yi, Tyler M. Moore, Monica E. Calkins, Christian G. Kohler, Daneen A. Whinna, Margaret C. Souders, Elaine H. Zackai, Donna M. McDonald-McGinn, Beverly S. Emanuel, Warren B. Bilker, Ruben C. Gur, Raquel E. Gur
- Attention-Deficit/Hyperactivity Disorder (ADHD) and Motor Timing in Adolescents and Their Parents: Familial Characteristics of Reaction Time Variability Vary With Age
- Andrieke J.A.M. Thissen, Marjolein Luman, Catharina Hartman, Pieter Hoekstra, Marloes van Lieshout, Barbara Franke, Jaap Oosterlaan, Nanda N.J. Rommelse, Jan K. Buitelaar
Bienvenue sur EM-consulte, la référence des professionnels de santé.
L’accès au texte intégral de cet article nécessite un abonnement.
Déjà abonné à cette revue ?