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DOI: 10.1055/s-0045-1812408
A systematic review of measurement properties of physical function outcomes in people with haemophilia
Autoren
Background The aim of this systematic review was to investigate the measurement properties of assessments, determining physical fitness and function in adult people with hemophilia (PwH), as well as to evaluate the methodological quality of the studies, and the level of evidence for the measurement properties.
Method Articles in English or German from Medline, EMBASE, CINAHL, Cochrane, PsycINFO, Scopus, PEDRO, or Cosmin were included if they evaluated performance-based or clinician-reported outcome measures (OMIs) for physical function in adult PwH. Studies were excluded if they only used OMIs for assessment, or focused on patient-reported outcomes or laboratory values. Data extraction and quality evaluation followed COSMIN guidelines, assessing risk of bias and measurement properties for each OMI. Results were summarized, and evidence quality was defined using a modified GRADE approach.
Results Fifteen articles describing 12 OMIs (Colorado Adult Joint Assessment Scale, Functional Independence Score in Haemophilia, Haemophilia Joint Health Score, Four Square Step Test, M3 diagnos, Microsoft Kinect V2 sensor, Three-Dimensional Gait Analysis, Timed Up and Go, and Short Physical Performance Battery) were included. Out of the nine possible measurement properties defined by COSMIN, six were examined: construct validity (convergent and discriminative), reliability, measurement error, internal consistency, criterion validity, and responsiveness. None of the included OMIs has sufficiently described measurement properties. Overall, the quality of evidence of all evaluated measurement properties was “very low” in 77%, “low” in 8% and “moderate” in 15%. Furthermore, the rating of the criteria for good measurement properties could not be determined in 54%.
Discussion Only the Hemophilia Joint Health Score 2.1 showed “sufficient” convergent validity with “moderate” quality of evidence and can be recommended. Due to the unsatisfactory methodological quality of the included studies and low quality of evidence, it still remains a challenge for researchers and clinicians to select appropriate OMIs for adult PwH. Further clinimetric studies are urgently needed and it is highly recommended to follow the COSMIN reporting guidelines for their conduction.
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Artikel online veröffentlicht:
23. Oktober 2025
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