AUTHOR=Tosin Michelle H. S. , Stebbins Glenn T. , Goetz Christopher G. , Hagerman Randi J. , Hessl David , Zolecki Melissa A. , Todd Peter K. , Leehey Maureen A. , Hall Deborah A. 

TITLE=Fragile X-associated tremor ataxia syndrome rating scale: Revision and content validity using a mixed method approach

JOURNAL=Frontiers in Neurology

VOLUME=Volume 13 - 2022

YEAR=2022

URL=https://www.frontiersin.org/journals/neurology/articles/10.3389/fneur.2022.977380

DOI=10.3389/fneur.2022.977380

ISSN=1664-2295

ABSTRACT=Background: The original Fragile X-associated Tremor Ataxia Syndrome Rating Scale (FXTAS-RS) contained 61 items, some requiring modifications to better meet recommendations for patient-focused rating scale development. Purpose: Provide initial validation of a revised version of the FXTAS-RS for motor signs. Method: We conducted a two-phase mixed-method approach. In Phase 1, revision, we implemented a Delphi technique identifying pertinent domains/subdomains and developing items through expert consensus. In Phase 2, content validation, we conducted cognitive pretesting assessing comprehensibility, comprehensiveness, and relevance of items to FXTAS motor signs. Results: After five rounds of Delphi panel and two rounds of cognitive pretesting, the revised version of the FXTAS-RS was established with 18 items covering five domains and 13 subdomains of motor signs. Cognitive pretesting revealed adequate content validity for the assessment of FXTAS motor signs. Conclusion: The revised FXTAS-RS has been successfully validated for content and it is now ready for large-scale field validation.