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dc.contributor.authorBurke, Tom
dc.contributor.authorRodriguez-Santana, Idaira
dc.contributor.authorChowdary, Pratima
dc.contributor.authorCurtis, Randall
dc.contributor.authorKhair, Kate
dc.contributor.authorLaffan, Michael
dc.contributor.authorMcLaughlin, Paul
dc.contributor.authorNoone, Declan
dc.contributor.authorO'Mahony, Brian
dc.contributor.authorPasi, John
dc.contributor.authorSkinner, Mark W.
dc.contributor.authorO'Hara, Jamie
dc.date.accessioned2022-12-27T20:00:17Z
dc.date.available2022-12-27T20:00:17Z
dc.date.issued2022-12-27
dc.identifierhttps://chesterrep.openrepository.com/bitstream/handle/10034/627400/hae.14731.pdf?sequence=3
dc.identifier.citationBurke, T., Rodriguez‐Santana, I., Chowdary, P., Curtis, R., Khair, K., Laffan, M., McLaughlin, P., Noone, D., O'Mahony, B., Pasi, J., Skinner, M., & O'Hara, J. (2022). Humanistic burden of problem joints for children and adults with haemophilia. Haemophilia, 29(2), 608-618. https://doi.org/10.1111/hae.14731
dc.identifier.issn1351-8216
dc.identifier.doi10.1111/hae.14731
dc.identifier.urihttp://hdl.handle.net/10034/627400
dc.description.abstractIntroduction: The “problem joint” (PJ) concept was developed to address patient‐centric needs for a more holistic assessment of joint morbidity for people with haemophilia (PwH). Aim: To quantify the humanistic burden of PJs in PwH to further support validation of the PJ outcome measure. Methods: Multivariable regression models evaluated the relationship between PJs and health‐related quality of life (HRQoL, EQ‐5D‐5L) and overall work productivity loss (WPL) using data from the ‘Cost of HaEmophilia: a Socioeconomic Survey’ population studies (adults: CHESS II, CHESS US+; children/adolescents: CHESS‐Paeds). Covariates included were haemophilia severity, age, comorbidities and education. Results: The CHESS II sample included 292 and 134 PwH for HRQoL and WPL analyses, mean age 38.6 years (39% ≥1 PJ, 61% none). CHESS US+ included 345 and 239 PwH for HRQoL and WPL, mean age 35 years (43% ≥1 PJ, 57% none). CHESS‐Paeds included 198 PwH aged 4–17 (HRQoL only), mean age 11.5 years (19% ≥1 PJ, 81% none). In CHESS II and CHESS US+, presence of PJs was associated with worse HRQoL (Both p < .001). Few CHESS‐Paeds participants had PJs, with no significant correlation with HRQoL. In CHESS II, upper body PJs were significantly correlated to WPL (p < .05). In CHESS US+, having ≥1 PJ or upper and lower body PJs were significantly correlated to WPL (vs. none; both p < .05). Conclusion: This study has shown a meaningful burden of PJs on PwH, which should be considered in clinical and health policy assessments of joint health.
dc.languageen
dc.publisherWiley
dc.relation.urlhttps://onlinelibrary.wiley.com/doi/10.1111/hae.14731
dc.rightsLicence for VoR version of this article: http://creativecommons.org/licenses/by-nc-nd/4.0/
dc.rights.urihttps://creativecommons.org/licenses/by-nc-nd/4.0/
dc.sourceissn: 1351-8216
dc.sourceissn: 1365-2516
dc.subjecthaemarthrosis
dc.subjecthaemophilia
dc.subjecthumanistic burden
dc.subjectproblem joint
dc.subjectproductivity impairment
dc.subjectquality of life
dc.titleHumanistic burden of problem joints for children and adults with haemophilia
dc.typeArticle
dc.contributor.departmentHCD Economics; The Innovation Centre; Royal Free London NHS Foundation Trust; Hematology Utilization Group Study (HUGS); Imperial College London; European Haemophilia Consortium; Irish Haemophilia Society; Barts and the London School of Medicine and Dentistry; McMaster University; Institute for Policy Advancement Ltd; University of Chester
dc.identifier.journalHaemophilia
dc.date.updated2022-12-27T20:00:17Z
dc.description.fundingFunder: Sanofi, BioMarin, Takeda, Bayer, Roche, Swedish Orphan Biovitrum AB (Sobi), Novo Nordisk and SHIRE
dc.date.accepted2022-12-14


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