{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,14]],"date-time":"2026-08-14T15:12:53Z","timestamp":1786720373983,"version":"build-2736575974"},"reference-count":24,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2023,3,10]],"date-time":"2023-03-10T00:00:00Z","timestamp":1678406400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100004339","name":"Sanofi","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004339","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004326","name":"Bayer","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004326","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007013","name":"F. Hoffmann-La Roche","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007013","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004191","name":"Novo Nordisk","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004191","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007343","name":"Shire","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007343","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2023,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>Adequate prophylactic treatment and physical activity improve joint health and clinical outcomes for people with haemophilia A (HA). However, non\u2010clinical joint\u2010related burden of moderate (MHA) and severe (SHA) HA has not been well characterised.<\/jats:p><\/jats:sec><jats:sec><jats:title>Aim<\/jats:title><jats:p>To quantify the joint health\u2010related humanistic and economic burden of MHA and SHA in Europe.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A retrospective analysis of the cross\u2010sectional CHESS population studies using a patient\u2010centric measure of joint health (problem joints, PJs: chronic joint pain and\/or limited range of movement due to compromised joint integrity with or without persistent bleeding) was conducted. Descriptive statistics summarised health\u2010related quality of life (HRQoL), work productivity\/activity impairment and costs by number of PJs (0, 1 or \u22652) and HA severity.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>A total of 1171 patients were included from CHESS\u2010II (<jats:italic>n<\/jats:italic> = 468) and CHESS\u2010PAEDs (<jats:italic>n<\/jats:italic> = 703). In both studies, 41 and 59% of patients had MHA and SHA, respectively. Prevalence of \u22652 PJs was similar with MHA and SHA (CHESS\u2010II: 23 and 26%; CHESS\u2010PAEDs: 4 and 3%, respectively). HRQoL was worse with an increasing number of PJs (CHESS\u2010II: .81 vs. .66 with 0 and \u22652 PJs, respectively, for MHA; .79 vs. .51 for SHA; CHESS\u2010PAEDs: .64 vs. .26 and .72 vs. .14). Total costs increased with increasing PJs regardless of severity in CHESS\u2010II (\u20ac2923 vs. \u20ac22,536 with 0 and \u22652 PJs, respectively, for MHA; \u20ac11,022 vs. \u20ac27,098 for SHA) and CHESS\u2010PAEDs (\u20ac6222 vs. \u20ac11,043 for MHA; \u20ac4457 vs. \u20ac14,039 for SHA).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Presence of PJs was associated with a substantial humanistic and economic burden on patients with MHA or SHA across the lifespan.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.14766","type":"journal-article","created":{"date-parts":[[2023,3,10]],"date-time":"2023-03-10T16:14:40Z","timestamp":1678464880000},"page":"753-760","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["The humanistic and economic burden of problem joints for children and adults with moderate or severe haemophilia A: Analysis of the CHESS population studies"],"prefix":"10.1111","volume":"29","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6690-8586","authenticated-orcid":false,"given":"Pratima","family":"Chowdary","sequence":"first","affiliation":[{"name":"Katharine Dormandy Haemophilia and Thrombosis Centre Royal Free Hospital  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francis","family":"Nissen","sequence":"additional","affiliation":[{"name":"F. Hoffmann\u2010La Roche Ltd  Basel Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tom","family":"Burke","sequence":"additional","affiliation":[{"name":"HCD Economics  Daresbury UK"},{"name":"University of Chester  Chester UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martynas","family":"Aizenas","sequence":"additional","affiliation":[{"name":"F. Hoffmann\u2010La Roche Ltd  Basel Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T\u00fcnde","family":"Czirok","sequence":"additional","affiliation":[{"name":"F. Hoffmann\u2010La Roche Ltd  Basel Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Harpal","family":"Dhillon","sequence":"additional","affiliation":[{"name":"Formerly HCD Economics  Daresbury UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jamie","family":"O'Hara","sequence":"additional","affiliation":[{"name":"HCD Economics  Daresbury UK"},{"name":"University of Chester  Chester UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,3,10]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13214"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S212723"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1182\/bloodadvances.2019001311"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12955-018-0908-9"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13561-018-0185-7"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13111"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13616"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.12672"},{"key":"e_1_2_10_10_1","unstructured":"O'HaraJ KhairK McLaughlinP et\u00a0al.\u201cProblem Joint\u201d A More Patient Relevant Definition for Joint Morbidity in Haemophilia.European Association for Haemophilia and Allied Disorders (EAHAD);2019. 6\u20138th Feb 2019 Prague Czech Republic."},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14731"},{"key":"e_1_2_10_12_1","unstructured":"NooneD AdisaksophaCC CurtisR et\u00a0al.The effect of haemophilia on activities of daily living (ADL): PROBE cohort. In:European Conference on Rare Diseases and Orphan Product Vienna Austria 10\u201012 May 2018.2018."},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.24220"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S87659"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.24218"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-017-0660-y"},{"key":"e_1_2_10_17_1","unstructured":"CastroFA SilkeyM O'HaraJ et\u00a0al.Joint bleeds in paediatric patients with haemophilia: a CHESS study analysis.Presented at: International Society on Thrombosis and Haemostasis; July 6\u201010 2019;Melbourne Australia."},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.3109\/07853890109002087"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2012.02.008"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2021.10.013"},{"key":"e_1_2_10_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2021.10.013"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14046"},{"key":"e_1_2_10_23_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14278"},{"key":"e_1_2_10_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/s0277\u20109536(98)00411\u20100"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.apmr.2006.12.032"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.14766","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.14766","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.14766","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,5,28]],"date-time":"2024-05-28T14:07:49Z","timestamp":1716905269000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.14766"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,3,10]]},"references-count":24,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2023,5]]}},"alternative-id":["10.1111\/hae.14766"],"URL":"https:\/\/doi.org\/10.1111\/hae.14766","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,3,10]]},"assertion":[{"value":"2022-07-18","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-02-13","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-03-10","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}