Measuring physical function capacity in persons with haemophilia: A systematic review of performance-based methods

Journal article

Holdsworth, C,, Bladen, M., Harbridge, H., Drechsler, W., Mahaffey, R., Perez-Alenda, S., Sayers, F., Strike, K., Timmer, M. and Stephensen, D. 2005. Measuring physical function capacity in persons with haemophilia: A systematic review of performance-based methods. Haemophilia. https://doi.org/10.1111/hae.70081

Publication dates
Authors	Holdsworth, C,, Bladen, M., Harbridge, H., Drechsler, W., Mahaffey, R., Perez-Alenda, S., Sayers, F., Strike, K., Timmer, M. and Stephensen, D.
Abstract	Introduction: Currently, physical health assessments in persons with haemophilia focus on bleed-related events and after-effects. The aim of the systematic review was to review and apply standardised criteria to evaluate reliability, responsiveness and construct validity of performance-based instruments evaluating physical capability in persons with haemophilia. Methods: Medline, CINAHL, Embase, EMCARE, and Cochrane (inception-March 2024) were searched using COSMIN filters for 7 performance-based tests in haemophilia, supplemented by manual searches. Reliability, responsiveness and construct validity of; six-minute walk test (6MWT), timed up and go test (TUG), timed up and down stairs (TUDS), 30-second sit to stand (30-STS), single leg stance (SLS), tandem stance (TS) and single hop for distance (SH) was evaluated. Results: The search yielded 88 abstracts; 25 studies remained after full-text screening, covering 5 of 7 performance-based instruments: 6MWT, TUG, TUDS, SLS, and 30-STS. No performance-based test was evaluated for all properties across all ages. Only TUG in adults and older adults and 6MWT in children and adolescents has been tested for all properties. No test received a high grading. Low and very low grades were given mostly for indeterminate results, small or single studies and lack of similar construct of comparator. The 6MWT in all age groups was the only performance-based test graded moderate, and this was for responsiveness. Conclusion: With increasing use of performance-based methods of physical function capacity, evaluating measurement properties is a priority. Until evidence is generated, we can only advocate the 6MWT to monitor responsiveness in adult persons with haemophilia affected with marked arthropathy.
Keywords	Core outcome set; Haemophilia; Joint health; Musculoskeletal; Physical capacity; Physical function; Systematic review
Year	2005
Journal	Haemophilia
Publisher	Wiley
ISSN	1351-8216
	1365-2516
Digital Object Identifier (DOI)	https://doi.org/10.1111/hae.70081
Official URL	https://onlinelibrary.wiley.com/doi/10.1111/hae.70081
Funder	European Association of Haemophilia and Associated Disorders (EAHAD)
Online	20 Jul 2025
Publication process dates
Accepted	18 Jun 2025
Deposited	23 Jul 2025
Publisher's version	Haemophilia - 2025 - Holdsworth - Measuring Physical Function Capacity in Persons With Haemophilia A Systematic Review of.pdf License CC BY 4.0 File Access Level Open
Supplemental file	Supplementary File 1.pdf Supplementary File 2_Selection Guidance Flow Chart.pdf License CC BY 4.0 File Access Level Open
Output status	Published

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	Supplementary File 2_Selection Guidance Flow Chart.pdf
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