Biological and environmental contributions to adaptive behavior in fragile X syndrome

• Published in: American journal of medical genetics. Part A 2003-02, Vol.117A (1), p.21-29
• Main Authors: Glaser, Bronwyn, Hessl, David, Dyer-Friedman, Jennifer, Johnston, Cindy, Wisbeck, Jacob, Taylor, Annette, Reiss, Allan
• Format: Article
• Language: English
• Subjects: Adaptation, Psychological; adaptive behavior; Adolescent; Child; developmental disability; Family Health; Female; fragile X syndrome; Fragile X Syndrome - genetics; Fragile X Syndrome - psychology; home environment; Humans; Male; Multivariate Analysis; phenotype; Social Class
• ISSN: 1552-4825; 1552-4833
• DOI: 10.1002/ajmg.a.10549

Fragile X syndrome is the most common cause of inherited developmental disability. The purpose of the present study is to understand how both biological and environmental influences affect the development of adaptive behavior in children with fragile X. In‐home assessments were conducted on 120 children (80 boys and 40 girls) with the fragile X full mutation and their unaffected siblings (58 boys and 62 girls). Hierarchical multiple regression analyses were utilized. Independent variables included biological or demographic variables specific to the child (age, gender, full‐scale IQ, and FMRP percentage), as well as factors specific to his/her environment (household income, home environment, maternal psychopathology, and effectiveness of educational/therapeutic services). Results of these analyses showed that for both boys with fragile X and the control sibling group, adaptive behavior was predicted by IQ, age, gender, and home environment. For girls with fragile X, adaptive behavior was most strongly associated with IQ. Adaptive behavior was not significantly associated with FMRP in boys or girls with fragile X. By examining the relative influences of biological and environmental factors on adaptive behavior in children with fragile X, we begin to lay the foundation for the development of more specific treatment studies in children with this disorder. © 2002 Wiley‐Liss, Inc.