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.
Publication types
-
Comparative Study
-
Research Support, N.I.H., Extramural
-
Validation Study
MeSH terms
-
Adolescent
-
Checklist*
-
Child
-
Child Behavior Disorders / diagnosis*
-
Child Behavior Disorders / genetics
-
Child Behavior Disorders / psychology
-
Child Behavior Disorders / therapy*
-
Child, Preschool
-
DNA Mutational Analysis
-
Factor Analysis, Statistical
-
Female
-
Fragile X Mental Retardation Protein / genetics
-
Fragile X Syndrome / diagnosis*
-
Fragile X Syndrome / genetics
-
Fragile X Syndrome / psychology
-
Fragile X Syndrome / therapy*
-
Humans
-
Intellectual Disability / diagnosis
-
Intellectual Disability / psychology
-
Intellectual Disability / therapy
-
Male
-
Outcome Assessment, Health Care / statistics & numerical data
-
Personality Assessment / statistics & numerical data*
-
Psychometrics / statistics & numerical data
-
Psychotropic Drugs / therapeutic use
-
Reproducibility of Results
-
Social Adjustment
-
Young Adult
Substances
-
FMR1 protein, human
-
Psychotropic Drugs
-
Fragile X Mental Retardation Protein