ArticleEuropean journal of neurology2025
The Facioscapulohumeral Muscular Dystrophy Rasch-Built Overall Disability Scale (FSHD-RODS): Longitudinal Assessment of a Disease-Specific Patient Reported Outcome.
Article in European journal of neurology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers, 1 of them a synthesis that pooled it.
What it found
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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.
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.
Who cites it
3 citing papers in PubMed, 1 synthesis or guideline pooled it.
- Measuring health-related quality of life in facioscapulohumeral muscular dystrophy: a COSMIN systematic review and conceptual framework.Health and quality of life outcomes · 2026Pooled it
- Response to: Disentangling Facioscapulohumeral Muscular Dystrophy Disability From Age and Comorbidities: A Call for Refined Stratification.European journal of neurology · 2025Article
- The Facioscapulohumeral Muscular Dystrophy Rasch-Built Overall Disability Scale (FSHD-RODS): Longitudinal Assessment of a Disease-Specific Patient Reported Outcome.European journal of neurology · 2025Article
Corrections and comments
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Authors and funding
6 authors.
Funding
Abstract
objectiveTo assess changes in the facioscapulohumeral muscular dystrophy Rasch-Built Overall Disability Scale (FSHD-RODS) over 6.5 years in FSHD patients.
methodsFSHD patients of 18 years or older were assessed at baseline (T1) and followed up at 5 years (T2) and 6.5 years (T3). The patient-reported FSHD-RODS and Sickness Impact Profile 68 (SIP68) questionnaires were evaluated, alongside the FSHD clinical severity score, FSHD clinical score and Motor Function Measure (MFM). Individual minimal clinically important differences (MCID-SE) of the FSHD-RODS were calculated to assess the clinical importance of the change over time. A subgroup analysis included patients eligible for current clinical trials.
resultsSixty-two patients were included, representing the full disease spectrum. The FSHD-RODS and SIP68 questionnaire outcomes remained stable, whereas overall disease severity slightly increased (FSHD clinical severity score 6 to 6.5, p < 0.001) and MFM slightly decreased (94% to 91%, p < 0.001). FSHD-RODS correlated strongly with SIP68, MFM and disease severity (ρ ≥ 0.69). The clinical trial subgroup (n = 33) showed similar results. Ten patients (16%), all with moderate-to-severe baseline disease (FSHD clinical severity score ≥ 6), showed clinically important deterioration over 6.5 years.
conclusionsFSHD progression is generally slow, with FSHD-RODS only detecting meaningful decline in moderately to severely affected patients. The ability to compensate for muscle weakness may limit the sensitivity of patient-reported outcomes like FSHD-RODS in clinical trials. These findings highlight the need for alternative measures to capture disease progression and treatment effects in FSHD research.
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