{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,5]],"date-time":"2026-01-05T07:30:12Z","timestamp":1767598212495,"version":"3.40.5"},"reference-count":14,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2022,12,27]],"date-time":"2022-12-27T00:00:00Z","timestamp":1672099200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2023,3]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>The \u201cproblem joint\u201d (PJ) concept was developed to address patient\u2010centric needs for a more holistic assessment of joint morbidity for people with haemophilia (PwH).<\/jats:p><\/jats:sec><jats:sec><jats:title>Aim<\/jats:title><jats:p>To quantify the humanistic burden of PJs in PwH to further support validation of the PJ outcome measure.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Multivariable regression models evaluated the relationship between PJs and health\u2010related quality of life (HRQoL, EQ\u20105D\u20105L) and overall work productivity loss (WPL) using data from the \u2018Cost of HaEmophilia: a Socioeconomic Survey\u2019 population studies (adults: CHESS II, CHESS US+; children\/adolescents: CHESS\u2010Paeds). Covariates included were haemophilia severity, age, comorbidities and education.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The CHESS II sample included 292 and 134 PwH for HRQoL and WPL analyses, mean age 38.6 years (39% \u22651 PJ, 61% none). CHESS US+ included 345 and 239 PwH for HRQoL and WPL, mean age 35 years (43% \u22651 PJ, 57% none). CHESS\u2010Paeds included 198 PwH aged 4\u201317 (HRQoL only), mean age 11.5 years (19% \u22651 PJ, 81% none). In CHESS II and CHESS US+, presence of PJs was associated with worse HRQoL (Both <jats:italic>p<\/jats:italic>\u00a0&lt;\u00a0.001). Few CHESS\u2010Paeds participants had PJs, with no significant correlation with HRQoL. In CHESS II, upper body PJs were significantly correlated to WPL (<jats:italic>p<\/jats:italic>\u00a0&lt;\u00a0.05). In CHESS US+, having \u22651 PJ or upper and lower body PJs were significantly correlated to WPL (vs. none; both <jats:italic>p<\/jats:italic>\u00a0&lt;\u00a0.05).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>This study has shown a meaningful burden of PJs on PwH, which should be considered in clinical and health policy assessments of joint health.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.14731","type":"journal-article","created":{"date-parts":[[2022,12,27]],"date-time":"2022-12-27T18:30:08Z","timestamp":1672165808000},"page":"608-618","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Humanistic burden of problem joints for children and adults with haemophilia"],"prefix":"10.1111","volume":"29","author":[{"given":"Tom","family":"Burke","sequence":"first","affiliation":[{"name":"HCD Economics  Daresbury UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2022-3239","authenticated-orcid":false,"given":"Idaira","family":"Rodriguez\u2010Santana","sequence":"additional","affiliation":[{"name":"HCD Economics  Daresbury UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6690-8586","authenticated-orcid":false,"given":"Pratima","family":"Chowdary","sequence":"additional","affiliation":[{"name":"Katherine Dormandy Haemophilia Centre and Thrombosis Unit Royal Free London NHS Foundation Trust  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6859-6432","authenticated-orcid":false,"given":"Randall","family":"Curtis","sequence":"additional","affiliation":[{"name":"Hematology Utilization Group Study (HUGS)  Walnut Creek USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2001-5958","authenticated-orcid":false,"given":"Kate","family":"Khair","sequence":"additional","affiliation":[{"name":"HCD Economics  Daresbury UK"},{"name":"Haemnet  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8268-3268","authenticated-orcid":false,"given":"Michael","family":"Laffan","sequence":"additional","affiliation":[{"name":"Centre for Haematology Imperial College London  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5962-7647","authenticated-orcid":false,"given":"Paul","family":"Mclaughlin","sequence":"additional","affiliation":[{"name":"Katherine Dormandy Haemophilia Centre and Thrombosis Unit Royal Free London NHS Foundation Trust  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Declan","family":"Noone","sequence":"additional","affiliation":[{"name":"HCD Economics  Daresbury UK"},{"name":"European Haemophilia Consortium  Brussels Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9780-6972","authenticated-orcid":false,"given":"Brian","family":"O'Mahony","sequence":"additional","affiliation":[{"name":"Irish Haemophilia Society  Dublin Ireland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3394-2099","authenticated-orcid":false,"given":"John","family":"Pasi","sequence":"additional","affiliation":[{"name":"Royal London Haemophilia Centre Barts and the London School of Medicine and Dentistry  London UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0934-0680","authenticated-orcid":false,"given":"Mark","family":"Skinner","sequence":"additional","affiliation":[{"name":"McMaster University  Hamilton Canada"},{"name":"Institute for Policy Advancement Ltd  Washington DC"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jamie","family":"O'Hara","sequence":"additional","affiliation":[{"name":"HCD Economics  Daresbury UK"},{"name":"University of Chester  Chester UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2022,12,27]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11420-009-9140-9"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14046"},{"key":"e_1_2_11_4_1","first-page":"35","article-title":"\u201cProblem Joint\u201d a more patient relevant definition for joint morbidity in 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