Validation of PROMIS Profile-29 in adults with hemophilia in the Netherlands

Erna C. van Balen, Lotte Haverman, Shermarke Hassan, Elisabeth M. Taal, Cees Smit, Mariëtte H. Driessens, Erik A.M. Beckers, Michiel Coppens, Jeroen Eikenboom, Hélène L. Hooimeijer, Frank W.G. Leebeek, Lize F.D. van Vulpen, Saskia E.M. Schols, Caroline B. Terwee, Frits R. Rosendaal, Johanna G. van der Bom, Samantha C. Gouw*

*Corresponding author for this work

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Background: The Patient-Reported Outcomes Measurement Information System (PROMIS) Profile-29 questionnaire is widely used worldwide, but it has not yet been validated in the Netherlands, nor in persons with hemophilia. Objective: To validate the Dutch-Flemish version of the PROMIS-29 Profile v2.01 in adults with hemophilia. Methods: Dutch males with hemophilia (all severities) completed questionnaires that contained sociodemographic and clinical characteristics, the PROMIS-29, RAND-36, and the Hemophilia Activities List (HAL). Structural validity of each subscale was assessed with confirmatory factor analysis (CFA). Internal consistency was calculated for each subscale with sufficient model fit in CFA. Construct validity was assessed by testing hypotheses about (1) correlations of each PROMIS-29 subscale with corresponding scales of RAND-36 and domains of HAL, and (2) mean differences in T-scores between subgroups with different hemophilia severities, self-reported joint impairment, and HIV infection status. We considered ≥75% of data in accordance with the hypotheses evidence for construct validity. Results: In total, 770 persons with hemophilia participated in this cross-sectional study. CFA revealed sufficient structural validity for five subscales: Physical Function, Depression, Sleep Disturbance, Ability to Participate in Social Roles and Activities, and Pain Interference. Internal consistency was high and Cronbach's alpha ranged from 0.79 for Sleep Disturbance to 0.96 for Pain Interference. Differences between clinical subgroups were in the expected direction. Construct validity was confirmed for Physical Function, Anxiety, Depression, Fatigue, Sleep Disturbance, and Pain Intensity. Conclusion: This study revealed sufficient evidence for structural validity, internal consistency, and construct validity for most PROMIS Profile-29 subscales among people with hemophilia in the Netherlands.

Original languageEnglish
Pages (from-to)2687-2701
Number of pages15
JournalJournal of Thrombosis and Haemostasis
Issue number11
Early online date10 Jul 2021
Publication statusPublished - Nov 2021

Bibliographical note

Funding Information:
This study was funded by the Dutch Ministry of Health, Welfare and Sports as part of the Hemophilia in the Netherlands study.

Funding Information:
E.C. van Balen, L. Haverman, S. Hassan, E.M. Taal, C. Smit, M.H. Driessens, E.A.M. Beckers, H.L. Hooimeijer, S.E.M. Schols, C.B. Terwee, and F.R. Rosendaal have no conflicts of interest to disclose. M. Coppens has received financial support for research from Bayer, CSL Behring, Daiichi Sankyo, Portola/Alexion, Roche, Sanquin Blood Supply, and UniQure and consultancy or lecturing fees from Bayer, CSL Behring, Medcon International, MEDtalks, NovoNordisk, Pfizer, and Sobi. J. Eikenboom received research support from CSL Behring and has been a teacher on educational activities of Roche. F.W.G. Leebeek received unrestricted research grants from CSL Behring, Takeda, UniQure and Sobi, is a consultant for UniQure, Novo Nordisk, Biomarin, and Takeda, of which the fees go to the institution, and has received a travel grant from Sobi. L.F.D. van Vulpen received a research grant form CSL Behring, is a consultant for Sobi and Tremeau; all fees go to the institution. He is also a DSMB member for a study by Roche. J.G. van der Bom has been a teacher on the educational activities of Bayer. S.C. Gouw has received unrestricted research grants from Sobi.

Publisher Copyright:
© 2021 The Authors. Journal of Thrombosis and Haemostasis published by Wiley Periodicals LLC on behalf of International Society on Thrombosis and Haemostasis.


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