Social communication in fragile X syndrome: pilot examination of the Brief Observation of Social Communication Change (BOSCC).

Shaffer, Rebecca; Thurman, Angela John; Ronco, Lucienne; Cadavid, Diego; Raines, Shane; Kim, So Hyun

Background: Social 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. Methods: We 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. Results: The 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. Conclusions: The BOSCC-MV shows promise as a FXS social communication outcome measure, warranting further large-scale evaluation.

VINELAND (N.J.); FRAGILE X syndrome; COMMUNICATIVE disorders; SOCIAL change; INTELLECTUAL disabilities; AUTISM spectrum disorders; STATISTICAL reliability

Journal of Neurodevelopmental Disorders, 2022, Vol 14, Issue 1, p1

1866-1947

Academic Journal

10.1186/s11689-021-09411-z