Longitudinal Profiles of Adaptive Behavior in Fragile X Syndrome

• Type of Material: Article
• Authors: Cheryl Klaiman, Emory University; Eve-Marie Quintin, Georgia Institute of Technology; Booil Jo, Stanford University; Amy A. Lightbody, Stanford University; Heather Cody Hazlett, University of North Carolina; Joseph Piven, University of North Carolina; Scott S. Hall, Stanford University; Allan L. Reiss, Stanford University
• Language: English
• Date: 2014-08-01
• Publisher: American Academy of Pediatrics
• Publication Version: Final Published Version
• Copyright Statement: © 2014 by the American Academy of Pediatrics
• Final Published Version (URL): http://dx.doi.org/10.1542/peds.2013-3990
• Title of Journal or Parent Work: Pediatrics
• ISSN: 0031-4005
• Volume: 134
• Issue: 2
• Start Page: 315
• End Page: 324
• Grant/Funding Information: This research was supported by National Institutes of Health grants MH064708 (Dr Reiss), MH085899 (Dr Reiss), MH050046 (Dr Reiss), MH64708 (Drs Piven and Reiss), and MH61696 (Dr Piven) and a postdoctoral grant to Dr Quintin through the Fonds Quebecois Pour la Recherche sur la Société et la Culture.; Support for this project was obtained from National Institutes of Health grants MH050047, MH064708, and MH019908, and a gift from the Canel Family Fund. Funded by the National Institutes of Health (NIH).
• Keywords: fragile X syndrome; Vineland; adolescents; adaptive behavior; children
• Research Categories: Health Sciences, Mental Health; Health Sciences, General

Abstract

OBJECTIVE: To examine longitudinally the adaptive behavior patterns in fragile X syndrome. METHOD: Caregivers of 275 children and adolescents with fragile X syndrome and 225 typically developing children and adolescents (2-18 years) were interviewed with the Vineland Adaptive Behavior Scales every 2 to 4 years as part of a prospective longitudinal study. RESULTS: Standard scores of adaptive behavior in people with fragile X syndrome are marked by a significant decline over time in all domains for males and in communication for females. Socialization skills are a relative strength as compared with the other domains for males with fragile X syndrome. Females with fragile X syndrome did not show a discernible pattern of developmental strengths and weaknesses. CONCLUSIONS: This is the first large-scale longitudinal study to show that the acquisition of adaptive behavior slows as individuals with fragile X syndrome age. It is imperative to ensure that assessments of adaptive behavior skills are part of intervention programs focusing on childhood and adolescence in this condition. Copyright © 2014 by the American Academy of Pediatrics.