title: Psychometric study of the Aberrant Behavior Checklist in Fragile X Syndrome and implications for targeted treatment.
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.
authors.affiliation: Medical Investigation of Neurodevelopmental Disorders Institute, University of California Davis Medical Center, 2825 50th Street, Sacramento, CA 95817, USA.
authors.forename: Stephanie M
authors.initials: SM
authors.lastname: Sansone
authors.forename: Keith F
authors.initials: KF
authors.lastname: Widaman
authors.forename: Scott S
authors.initials: SS
authors.lastname: Hall
authors.forename: Allan L
authors.initials: AL
authors.lastname: Reiss
authors.forename: Amy
authors.initials: A
authors.lastname: Lightbody
authors.forename: Walter E
authors.initials: WE
authors.lastname: Kaufmann
authors.forename: Elizabeth
authors.initials: E
authors.lastname: Berry-Kravis
authors.forename: Ave
authors.initials: A
authors.lastname: Lachiewicz
authors.forename: Elaine C
authors.initials: EC
authors.lastname: Brown
authors.forename: David
authors.initials: D
authors.lastname: Hessl
mesh_terms: D000293:Adolescent; D057189:Checklist; D002648:Child; D002653:Child Behavior Disorders / Q000175:diagnosis* / Q000235:genetics / Q000523:psychology / Q000628:therapy*; D002675:Child, Preschool; D004252:DNA Mutational Analysis; D005163:Factor Analysis, Statistical; D005260:Female; D051860:Fragile X Messenger Ribonucleoprotein 1 / Q000235:genetics; D005600:Fragile X Syndrome / Q000175:diagnosis* / Q000235:genetics / Q000523:psychology / Q000628:therapy*; D006801:Humans; D008607:Intellectual Disability / Q000175:diagnosis / Q000523:psychology / Q000628:therapy; D008297:Male; D017063:Outcome Assessment, Health Care / Q000706:statistics & numerical data; D010552:Personality Assessment / Q000706:statistics & numerical data*; D011594:Psychometrics / Q000706:statistics & numerical data; D011619:Psychotropic Drugs / Q000627:therapeutic use; D015203:Reproducibility of Results; D012917:Social Adjustment; D055815:Young Adult
chemical_list: D051860:Fragile X Messenger Ribonucleoprotein 1; D011619:Psychotropic Drugs; C497683:FMR1 protein, human