1. Autism spectrum disorder in fragile X syndrome: A longitudinal evaluation
- Author
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R. Nick Hernandez, Rachel L. Feinberg, Richard E. Thompson, Natalie M. Passanante, Walter E. Kaufmann, and Rebecca Vaurio
- Subjects
Male ,congenital, hereditary, and neonatal diseases and abnormalities ,Population ,Child Behavior ,Stanford-Binet Test ,Severity of Illness Index ,behavioral disciplines and activities ,Article ,Cohort Studies ,Reference Values ,Adaptation, Psychological ,Interview, Psychological ,mental disorders ,Genetics ,medicine ,Pervasive developmental disorder ,Humans ,Interpersonal Relations ,Longitudinal Studies ,Autistic Disorder ,Child ,Social Behavior ,education ,Genetics (clinical) ,Language Disorders ,education.field_of_study ,Language Tests ,Intelligence quotient ,Autism Diagnostic Interview ,medicine.disease ,Fragile X syndrome ,Developmental disorder ,Child Development Disorders, Pervasive ,Autism spectrum disorder ,Child, Preschool ,Fragile X Syndrome ,Regression Analysis ,Autism ,Psychology ,Clinical psychology - Abstract
The present study extends our previous work on characterizing the autistic behavior profile of boys with fragile X syndrome (FXS) who meet Diagnostic and Statistical Manual for Mental Disorders, 4th Edition criteria for autism spectrum disorder (ASD) into a longitudinal evaluation of ASD in FXS (FXS + ASD). Specifically, we aimed to determine the stability of the diagnosis and profile of ASD in FXS over time. Through regression models, we also evaluated which autistic and social behaviors and skills were correlates of diagnosis and autistic behavior severity (i.e., Autism Diagnostic Interview-Revised total scores). Finally, we assessed the evolution of cognitive parameters in FXS + ASD. A population of 56 boys (30-88 months at baseline) with FXS was evaluated using measures of autistic, social, and cognitive behaviors and skills at three yearly evaluations. We found that the diagnosis of ASD in FXS was relatively stable over time. Further emphasizing this stability, we found a set of behaviors and skills, particularly those related to peer relationships and adaptive socialization, that differentiated FXS + ASD from the rest of the FXS cohort (FXS + None) and contributed to autistic severity at all time points. Nevertheless, the general improvement in autistic behavior observed in FXS + ASD coupled with the concurrent worsening in FXS + None resulted in less differentiation between the groups over time. Surprisingly, FXS + ASD IQ scores were stable while FXS + None non-verbal IQ scores declined. Our findings indicate that ASD is a distinctive subphenotype in FXS characterized by deficits in complex social interaction, with similarities to ASD in the general population.
- Published
- 2009
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