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.
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Acknowledgments
We are especially grateful for the participants and their families for their contribution to the understanding of fragile X syndrome. We would also like to thank Lisa Cordeiro, Elizabeth Ballinger, Alyssa Chavez, Ava Rezvani, Lia Boyle, Victor Talisa, Nana Asante, Crystal Hervey, and Anna DeSonia for their help with site coordination, data entry and management. This work was supported by NIH K23MH77554 (Hessl), the National Fragile X Foundation (Hessl), FRAXA (Hessl), NIH K08MH081998 (Hall), R01MH050047 (Reiss), and NIH HD24061 (Kaufmann).
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Sansone, S.M., Widaman, K.F., Hall, S.S. et al. Psychometric Study of the Aberrant Behavior Checklist in Fragile X Syndrome and Implications for Targeted Treatment. J Autism Dev Disord 42, 1377–1392 (2012). https://doi.org/10.1007/s10803-011-1370-2
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DOI: https://doi.org/10.1007/s10803-011-1370-2