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


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.

SUBMITTER: Tosin MHS 

PROVIDER: S-EPMC9515309 | biostudies-literature | 2022

REPOSITORIES: biostudies-literature

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Publications

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

Tosin Michelle H S MHS   Stebbins Glenn T GT   Goetz Christopher G CG   Hagerman Randi J RJ   Hessl David D   Zolecki Melissa A MA   Todd Peter K PK   Leehey Maureen A MA   Hall Deborah A DA  

Frontiers in neurology 20220914


<h4>Background</h4>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.<h4>Purpose</h4>Provide initial validation of a revised version of the FXTAS-RS for motor signs.<h4>Method</h4>We conducted a two-phase mixed-method approach. In Phase 1, revision, we implemented a Delphi technique identifying pertinent domains/subdomains and developing item  ...[more]

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