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Frontiers in neurology · 2022

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

СтатьяНазвание и аннотация: Перевод в очереди
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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

Аннотация

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.

ИП Урсолов Владлен Сергеевич

ИНН 500118482352 · ОГРНИП 314500117700049

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