Original Paper

Journal of Autism and Developmental Disorders

, Volume 42, Issue 7, pp 1377-1392

Psychometric Study of the Aberrant Behavior Checklist in Fragile X Syndrome and Implications for Targeted Treatment

  • Stephanie M. SansoneAffiliated withMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California Davis Medical Center
  • , Keith F. WidamanAffiliated withDepartment of Psychology, University of California, Davis
  • , Scott S. HallAffiliated withDepartment of Psychiatry and Behavioral Sciences, Center for Interdisciplinary Brain Sciences Research, Stanford University
  • , Allan L. ReissAffiliated withDepartment of Psychiatry and Behavioral Sciences, Center for Interdisciplinary Brain Sciences Research, Stanford University
  • , Amy LightbodyAffiliated withDepartment of Psychiatry and Behavioral Sciences, Center for Interdisciplinary Brain Sciences Research, Stanford University
  • , Walter E. KaufmannAffiliated withCenter for Genetic Disorders of Cognition and Behavior, Kennedy Krieger Institute, Johns Hopkins University School of Medicine
  • , Elizabeth Berry-KravisAffiliated withDepartments of Pediatrics, Neurological Sciences, Biochemistry, Rush University Medical Center
  • , Ave LachiewiczAffiliated withDepartments of Pediatrics and Psychiatry and Behavioral Sciences, Duke University Medical CenterPrivate Practice
  • , Elaine C. BrownAffiliated withPrivate Practice
    • , David HesslAffiliated withMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California Davis Medical CenterDepartment of Psychiatry and Behavioral Sciences, University of California, Davis, School of Medicine Email author 

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Abstract

Animal studies elucidating the neurobiology of fragile X syndrome (FXS) have led to multiple controlled trials in humans, with the Aberrant Behavior Checklist-Community (ABC-C) commonly adopted as a primary outcome measure. A multi-site collaboration examined the psychometric properties of the ABC-C in 630 individuals (ages 3–25) with FXS using exploratory and confirmatory factor analysis. Results support a six-factor structure, with one factor unchanged (Inappropriate Speech), four modified (Irritability, Hyperactivity, Lethargy/Withdrawal, and Stereotypy), and a new Social Avoidance factor. A comparison with ABC-C data from individuals with general intellectual disability and a list of commonly endorsed items are also reported. Reformulated ABC-C scores based on this FXS-specific factor structure may provide added outcome measure specificity and sensitivity in FXS clinical trials.

Keywords

FMR1 gene Fragile X syndrome Autism Factor analysis Rating scale Social avoidance