In fragile X syndrome (FXS), social communication is a primary area of difficulty, and there are currently no good outcome evaluation techniques. To evaluate future therapy trials, appropriate outcome metrics for FXS have been recognized as a critical topic of scientific focus. The Brief Observation of Social Communication Change-Minimally Verbal (BOSCC-MV), a robust psychometric outcome measure created for autism spectrum disorder, holds promise as an outcome measure to assess social communication change in FXS patients. For a multi-site trial, researchers used central coders to test the BOSCC-suitability MV’s for FXS. Eighteen barely verbal men are aged 3–12 were enrolled and tested for 2 days, with 7 participants returning 6 months later for a 3rd examination. With standard clinical measures such as the Autism Diagnostic and Observation Schedule-2, Vineland 3, Social Responsiveness Scale, and the Aberrant Behavior Checklist, the researchers looked at test-retest reliability, inter-rater reliability, and convergent and divergent validity. The inter-rater and test-retest reliability of the BOSCC-MV in FXS were comparable to prior trials in idiopathic ASD. Autism Diagnostic Observation Schedule-2 and Vineland-3 both had high convergent validity. BOSCC-MV and unrelated measures were shown to have divergent validity. The BOSCC-MV showed promise as an FXS social communication outcome measure, indicating that it should be evaluated on a larger scale.
Link:link.springer.com/article/10.1186/s11689-021-09411-z
