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=13

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=<sec><title>Background</title><p>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.</p></sec><sec><title>Purpose</title><p>Provide initial validation of a revised version of the FXTAS-RS for motor signs.</p></sec><sec><title>Method</title><p>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.</p></sec><sec><title>Results</title><p>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.</p></sec><sec><title>Conclusion</title><p>The revised FXTAS-RS has been successfully validated for content and it is now ready for large-scale field validation.</p></sec>