1. Social communication in fragile X syndrome: pilot examination of the Brief Observation of Social Communication Change (BOSCC)
- Author
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Shaffer, Rebecca, Thurman, Angela John, Ronco, Lucienne, Cadavid, Diego, Raines, Shane, and Kim, So Hyun
- Subjects
Biomedical and Clinical Sciences ,Neurosciences ,Clinical Trials and Supportive Activities ,Rare Diseases ,Pediatric ,Brain Disorders ,Intellectual and Developmental Disabilities (IDD) ,Mental Health ,Fragile X Syndrome ,Autism ,Behavioral and Social Science ,Clinical Research ,Mental health ,Autism Spectrum Disorder ,Child ,Child ,Preschool ,Communication ,Humans ,Male ,Reproducibility of Results ,Social Communication Disorder ,Fragile X syndrome ,BOSCC ,Social communication ,Repetitive behaviors ,Outcome measure ,Psychology - Abstract
BackgroundSocial communication is a key area of difficulty in fragile X syndrome (FXS) and there are not yet adequate outcome measurement tools. Appropriate outcome measures for FXS have been identified as a key area of research interest in order to evaluate future therapeutic trials. The Brief Observation of Social Communication Change-Minimally Verbal (BOSCC-MV), an outcome measure with strong psychometrics developed for autism spectrum disorder, has promise as an outcome measure to assess social communication change with FXS participants.MethodsWe examined the BOSCC-MV via central coders in this multi-site-trial to assess its appropriateness for FXS. Eighteen minimally verbal males ages 3-12 years were enrolled and assessed on two consecutive days and 7 participants completed a third visit 6 months later. We examined test-retest reliability, inter-rater reliability, and both convergent and divergent validity with standard clinical measures including the Autism Diagnostic and Observation Schedule-2, Vineland 3, Social Responsiveness Scale, and the Aberrant Behavior Checklist.ResultsThe BOSCC-MV in FXS demonstrated strong inter-rater and test-retest reliability, comparable to previous trials in idiopathic ASD. Strong convergent validity was found with Autism Diagnostic Observation Schedule-2 and Vineland-3. Divergent validity was demonstrated between BOSCC-MV and unrelated measures.ConclusionsThe BOSCC-MV shows promise as a FXS social communication outcome measure, warranting further large-scale evaluation.
- Published
- 2022