Statements in which the resource exists as a subject.
PredicateObject
rdf:type
lifeskim:mentions
pubmed:issue
6
pubmed:dateCreated
2009-6-2
pubmed:abstractText
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.
pubmed:grant
pubmed:commentsCorrections
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pubmed:language
eng
pubmed:journal
pubmed:citationSubset
IM
pubmed:status
MEDLINE
pubmed:month
Jun
pubmed:issn
1552-4833
pubmed:author
pubmed:copyrightInfo
(c) 2009 Wiley-Liss, Inc.
pubmed:issnType
Electronic
pubmed:volume
149A
pubmed:owner
NLM
pubmed:authorsComplete
Y
pubmed:pagination
1125-37
pubmed:dateRevised
2009-11-18
pubmed:meshHeading
pubmed-meshheading:19441123-Adaptation, Psychological, pubmed-meshheading:19441123-Autistic Disorder, pubmed-meshheading:19441123-Child, pubmed-meshheading:19441123-Child, Preschool, pubmed-meshheading:19441123-Child Behavior, pubmed-meshheading:19441123-Child Development Disorders, Pervasive, pubmed-meshheading:19441123-Cohort Studies, pubmed-meshheading:19441123-Fragile X Syndrome, pubmed-meshheading:19441123-Humans, pubmed-meshheading:19441123-Interpersonal Relations, pubmed-meshheading:19441123-Interview, Psychological, pubmed-meshheading:19441123-Language Disorders, pubmed-meshheading:19441123-Language Tests, pubmed-meshheading:19441123-Longitudinal Studies, pubmed-meshheading:19441123-Male, pubmed-meshheading:19441123-Reference Values, pubmed-meshheading:19441123-Regression Analysis, pubmed-meshheading:19441123-Severity of Illness Index, pubmed-meshheading:19441123-Social Behavior, pubmed-meshheading:19441123-Stanford-Binet Test
pubmed:year
2009
pubmed:articleTitle
Autism spectrum disorder in fragile X syndrome: a longitudinal evaluation.
pubmed:affiliation
Kennedy Krieger Institute, Baltimore, MD 21211, USA.
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