{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T22:41:24Z","timestamp":1773009684325,"version":"3.50.1"},"reference-count":24,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2025,7,17]],"date-time":"2025-07-17T00:00:00Z","timestamp":1752710400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2025,7,17]],"date-time":"2025-07-17T00:00:00Z","timestamp":1752710400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100008373","name":"Takeda Pharmaceutical Company","doi-asserted-by":"publisher","award":["IISR\u20102018\u2010104288"],"award-info":[{"award-number":["IISR\u20102018\u2010104288"]}],"id":[{"id":"10.13039\/100008373","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100008373","name":"Takeda Pharmaceutical Company","doi-asserted-by":"publisher","award":["IIR\u2010POL\u2010002288"],"award-info":[{"award-number":["IIR\u2010POL\u2010002288"]}],"id":[{"id":"10.13039\/100008373","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2025,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>The development of factor VIII inhibitors is a significant complication of hemophilia A, increasing the risk of bleeding and resulting in high morbidity. Recurrent joint bleeds lead to severe arthropathy and a reduced quality of life compared to patients without inhibitors. Although recent therapies have improved outcomes, many patients with longstanding inhibitors already experience irreversible joint damage. Research on rehabilitation in hemophilia, particularly for patients with inhibitors, remains limited.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>This 4\u2010year study evaluated the impact of individualized physiotherapy on musculoskeletal health and quality of life (QoL) in severe hemophilia A patients with inhibitors.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Twenty\u2010five adults (aged 19\u201369) with severe hemophilia A and inhibitors received personalized home physiotherapy supervised by a specialist, alongside biannual 5\u2010day rehabilitation camps. Prophylactic activated prothrombin complex concentrate (aPCC) was administered. Joint status was assessed biennially via Hemophilia Joint Health Score (HJHS) and MRI of target joints; QoL was measured using EQ\u20105D questionnaires.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>No bleeding episodes occurred during prophylaxis. Thirteen patients completed follow\u2010up. MRI scores stabilized (mean change: +1.3 points; 95% CI: 0.3\u20132.3), while HJHS improved significantly from 41.1 (95% CI: 32.5\u201349.7) to 24.9 (95% CI: 16.9\u201332.7). EQ\u20105D scores rose from 54.0 (95% CI: 48.2\u201359.8) to 86.7 (95% CI: 81.9\u201391.5), reflecting enhanced mobility, reduced pain and greater independence.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Individualized rehabilitation with aPCC prophylaxis significantly improved joint function (notably pain and mobility) and QoL, despite limited MRI changes due to pre\u2010existing arthropathy. These results advocate for long\u2010term, tailored physiotherapy in inhibitor patients.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.70094","type":"journal-article","created":{"date-parts":[[2025,7,17]],"date-time":"2025-07-17T17:39:39Z","timestamp":1752773979000},"page":"1025-1035","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Impact of the Individualized 4\u2010Year Physiotherapy on the Musculoskeletal System and Quality of Life of Patients With Severe Hemophilia A With Inhibitors"],"prefix":"10.1111","volume":"31","author":[{"given":"Maria","family":"Podolak\u2010Dawidziak","sequence":"first","affiliation":[{"name":"Wroclaw Medical University, Department and Clinic of Hematology Cellular Therapies and Internal Medicine  Wroc\u0142aw Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Janusz","family":"Zawilski","sequence":"additional","affiliation":[{"name":"Medical Center Interlab  Pozna\u0144 Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ewa","family":"Stefa\u0144ska\u2010Windyga","sequence":"additional","affiliation":[{"name":"Department of Hemostasis Disorders and Internal Diseases Institute of Hematology and Transfusion Medicine in Warsaw  Warsaw Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Magdalena","family":"G\u00f3rska\u2010Kosicka","sequence":"additional","affiliation":[{"name":"Department of Hemostasis Disorders and Internal Diseases Institute of Hematology and Transfusion Medicine in Warsaw  Warsaw Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anna","family":"Buczma","sequence":"additional","affiliation":[{"name":"Department of Hemostasis Disorders and Internal Diseases Institute of Hematology and Transfusion Medicine in Warsaw  Warsaw Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ewa","family":"Chmielewska","sequence":"additional","affiliation":[{"name":"Department of Hematology and Transplantology Pomeranian Medical University in Szczecin  Szczecin Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jolanta","family":"Oleksiuk","sequence":"additional","affiliation":[{"name":"Department of Hematology Internal Diseases and Angiology with a Subdivision of Hematopoietic Cell Transplantation University Clinical Hospital in Bialystok  Bialystok Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6435-7613","authenticated-orcid":false,"given":"Joanna","family":"Zdziarska","sequence":"additional","affiliation":[{"name":"Clinical Department of Hematology and Internal Diseases University Hospital in Cracow  Krak\u00f3w Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andrzej","family":"Mital","sequence":"additional","affiliation":[{"name":"Department of Hematology and Transplantology University Clinical Center in Gdansk  Gda\u0144sk Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mariola","family":"Bober","sequence":"additional","affiliation":[{"name":"Department of Hematology Independent Public Healthcare Department of the Ministry of Interior and Administration in Poznan  Poznan Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Justyna","family":"Kozi\u0144ska","sequence":"additional","affiliation":[{"name":"Department of Hematology and Bone Marrow Transplantation University Clinical Hospital No. 1 Lublin  Lublin Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Micha\u0142","family":"Jamrozik","sequence":"additional","affiliation":[{"name":"Department of Hematology and Bone Marrow Transplantation Independent Public Clinical Hospital of the Medical University of Silesia in Katowice  Katowice Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3161-3398","authenticated-orcid":false,"given":"Monika","family":"Biernat","sequence":"additional","affiliation":[{"name":"Wroclaw Medical University, Department and Clinic of Hematology Cellular Therapies and Internal Medicine  Wroc\u0142aw Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Agnieszka","family":"Brzozowska","sequence":"additional","affiliation":[{"name":"Department of Hematology Multispecialty Provincial Hospital in Gorzow Wielkopolski, Oncology and Radiotherapy, University of Zielona G\u00f3ra  Zielona G\u00f3ra Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,7,17]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2007.01518.x"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14638"},{"issue":"7","key":"e_1_2_11_4_1","article-title":"Adherence to Prophylactic Treatment in Adult Patients With Severe Hemophilia in Poland","volume":"132","author":"Zdziarska J.","year":"2022","journal-title":"Polish Archives of Internal Medicine"},{"issue":"1","key":"e_1_2_11_5_1","first-page":"657","article-title":"Hemophilia and Inhibitors: Current Treatment Options and Potential New Therapeutic Approaches","volume":"2016","author":"Meeks S. L.","year":"2016","journal-title":"Hematology. American Society of Hematology"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14404"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1177\/2040620718784834"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2006.01312.x"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/acr.20353"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12197"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/rth2.12690"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.15010"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.seminhematol.2015.10.005"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.15676"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pain.2011.04.030"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1155\/2013\/305249"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12320"},{"issue":"1","key":"e_1_2_11_18_1","first-page":"66","article-title":"Promoting Physical Activity in People With Haemophilia: The MEMO (Movement for Persons With haEMOphilia) Expert Consensus Project","volume":"20","author":"Biasoli C.","year":"2022","journal-title":"Blood Transfusion"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.3390\/jcm13113036"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.02111.x"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14775"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.2147\/JBM.S43734"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14900"},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14282"},{"key":"e_1_2_11_25_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.15116"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70094","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.70094","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70094","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T20:55:59Z","timestamp":1773003359000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.70094"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,7,17]]},"references-count":24,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2025,9]]}},"alternative-id":["10.1111\/hae.70094"],"URL":"https:\/\/doi.org\/10.1111\/hae.70094","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,7,17]]},"assertion":[{"value":"2024-11-25","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-07-02","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-07-17","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}