We carried out a psychometric assessment of the Social Communication Questionnaire (SCQ) and the Social Responsiveness Scale (SRS-2) in fragile X syndrome (FXS), relative to clinician DSM5-based diagnosis of autism spectrum disorder (ASD) in FXS. This was followed by instrument revisions that included: removal of non-discriminating and/or low face validity items for FXS; use of receiver operating characteristic (ROC) curves to determine optimal cut points for the original and revised measures; an exploratory factor analysis to outline subscales better representing ASD in FXS; and creation of a “triple criteria” diagnosis to better delineate ASD subgroups in FXS. These methods improved the sensitivity and/or specificity of the SCQ and SRS-2, but diagnostic accuracy of ASD remains problematic in FXS.
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This publication was supported by cooperative agreements #U01DD000231, #U19DD000753 and # U01DD001189, Funded by the Centers for Disease Control and Prevention. Its contents are solely the responsibility of the authors and do not necessarily represent the official views of the Centers for Disease Control and Prevention or the Department of Health and Human Services. We thank the families and individuals with fragile X syndrome that contributed their information to the FORWARD Registry and Database.
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Kidd, S.A., Berry-Kravis, E., Choo, T.H. et al. Improving the Diagnosis of Autism Spectrum Disorder in Fragile X Syndrome by Adapting the Social Communication Questionnaire and the Social Responsiveness Scale-2. J Autism Dev Disord 50, 3276–3295 (2020). https://doi.org/10.1007/s10803-019-04148-0
- Autism spectrum disorder
- Fragile X syndrome
- Social Communication Questionnaire
- Social Responsiveness Scale-2
- Receiver operating characteristic curves
- Exploratory factor analysis