Evidence map›Paper›PMID 36188408›Full record

ArticleFrontiers in neurology2022

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

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

Abstract read
In one paragraph

Article in Frontiers in neurology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Essential tremor-like phenotype in Fragile X carrier women.Clinical parkinsonism & related disorders · 2026
    Article
  2. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

9 authors.

Michelle H S TosinDepartment of Neurological Sciences, Rush University Medical Center, Chicago, IL, United States.
Glenn T StebbinsDepartment of Neurological Sciences, Rush University Medical Center, Chicago, IL, United States.
Christopher G GoetzDepartment of Neurological Sciences, Rush University Medical Center, Chicago, IL, United States.
Randi J HagermanDepartment of Pediatrics and the MIND Institute, University of California, Davis School of Medicine, Sacramento, CA, United States.
David HesslDepartment of Psychiatry and Behavioral Sciences and the MIND Institute, University of California Davis School of Medicine, Sacramento, CA, United States.
Melissa A ZoleckiNational Fragile X Foundation, McLean, VA, United States.
Peter K ToddUniversity of Michigan, Ann Harbor, MI, United States.
Maureen A LeeheyUniversity of Colorado School of Medicine, Aurora, CO, United States.
Deborah A HallDepartment of Neurological Sciences, Rush University Medical Center, Chicago, IL, United States.

Funding

Trajectories and Markers of Neurodegeneration in Fragile X Premutation CarriersR01NS110100 · NINDS · UNIVERSITY OF CALIFORNIA AT DAVIS · PI DAVID R HESSL, Susan M Rivera · 2018 to 2026
$5.4M
CGG repeat associated translation in Fragile X-associated Tremor/Ataxia Syndrome-Diversity SupplementR01NS086810 · NINDS · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI TODD, PETER K · 2014 to 2024
$4.1M
NINDS NIH HHS R01 NS086810NINDS NIH HHS R01 NS110100
6 · The paper itself

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.

Indexed as

fragile X syndromefragile X tremor ataxia syndromeFXTASoutcome measuresseverity of illness index

Identifiers

PMID36188408
PMCPMC9515309

What OpenQuestion holds

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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.