{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T18:35:59Z","timestamp":1791657359338,"version":"4.3.4"},"reference-count":32,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2023,3,28]],"date-time":"2023-03-28T00:00:00Z","timestamp":1679961600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100004191","name":"Novo Nordisk","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004191","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>The World Haemophilia Federation advises regular musculoskeletal assessment covering all International Classification of Functioning and Health (ICF) domains, including limitations in activities and participation in persons with haemophilia (PWH). This enables clinicians to detect changes early and enable adjustments in personalized healthcare when needed. However, data on the course of physical functioning and occurrence of decline is lacking. The aim of this study is to describe changes in perceived limitations in activities of PWH and to identify factors associated with a change.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Data were collected from medical health records of regular check\u2010up visits of adults with moderate and severe haemophilia in two time periods. Perceived limitations in activities was measured with the Haemophilia Activities List (HAL). Association between variables (e.g., age, body mass index, bleeding rate and synovitis) and change in perceived limitations was assessed using a generalized linear model.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>A total of 104 PWH were included. At T0, the median HAL sum score was 79.5 (IQR 62.1\u201393.6) and at T1 the median HAL sum score was 74.2 (IQR 57.5\u201388.3). A functional decline was found in 35.6% of PWH, 55.8% remained stable and 8.7% improved. Among other variables, a BMI\u00a0&gt;\u00a030\u00a0kg\/m<jats:sup>2<\/jats:sup> appeared to be an important factor that negatively influenced the change in perceived functioning in adult PWH. With the included factors we could only explain a small part of this decline (<jats:italic>R<\/jats:italic><jats:sup>2<\/jats:sup><jats:italic><jats:sub>adj<\/jats:sub><\/jats:italic>: .12).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The majority of PWH remained stable in their perceived functional ability over mid\u2010long term (median 3.5 years). However, about a third showed a clinical relevant decline in their functional ability.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.14783","type":"journal-article","created":{"date-parts":[[2023,3,28]],"date-time":"2023-03-28T08:55:40Z","timestamp":1679993740000},"page":"790-798","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Functional decline in persons with haemophilia and factors associated with deterioration"],"prefix":"10.1111","volume":"29","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4291-3735","authenticated-orcid":false,"given":"Johan","family":"Blokzijl","sequence":"first","affiliation":[{"name":"Centre for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek University Medical Centre Utrecht  Utrecht Netherlands"},{"name":"Physical Therapy Research, Department of Rehabilitation Physiotherapy Science and Sport, Brain Center Rudolf Magnus University Medical Center Utrecht  Utrecht Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martijn F.","family":"Pisters","sequence":"additional","affiliation":[{"name":"Physical Therapy Research, Department of Rehabilitation Physiotherapy Science and Sport, Brain Center Rudolf Magnus University Medical Center Utrecht  Utrecht Netherlands"},{"name":"Research Group Empowering Healthy Behaviour, Department of Health Innovations and Technology Fontys University of Applied Sciences  Eindhoven Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cindy","family":"Veenhof","sequence":"additional","affiliation":[{"name":"Physical Therapy Research, Department of Rehabilitation Physiotherapy Science and Sport, Brain Center Rudolf Magnus University Medical Center Utrecht  Utrecht Netherlands"},{"name":"Research Group Innovation of Human Movement Care University of Applied Sciences Utrecht  Utrecht Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roger E. G.","family":"Schutgens","sequence":"additional","affiliation":[{"name":"Centre for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek University Medical Centre Utrecht  Utrecht Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1910-999X","authenticated-orcid":false,"given":"Merel A.","family":"Timmer","sequence":"additional","affiliation":[{"name":"Centre for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek University Medical Centre Utrecht  Utrecht Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,3,28]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.15256\/joc.2011.1.2"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.phrs.2016.11.032"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102516.2005.01065.x"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12955\u2010018\u20100908\u20109"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365\u20102516.9.s1.13.x"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14099"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14046"},{"key":"e_1_2_11_9_1","unstructured":"World Health Organization.International Classification of Functioning Disability and Health;2001."},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1080\/10245332.2018.1505225"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13327"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13835"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.joca.2012.02.002"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.joca.2013.12.007"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13075\u2010015\u20100670\u2010x"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.semarthrit.2020.10.015"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102516.2004.01016.x"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14226"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1186\/1471\u20102458\u201012\u2010715"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1177\/2040620719896959"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.18637\/jss.v045.i03"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1201\/9780429492259"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1002\/sim.4067"},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097\u20100258(19990330)18:6"},{"key":"e_1_2_11_25_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14278"},{"key":"e_1_2_11_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.apmr.2006.12.032"},{"key":"e_1_2_11_27_1","doi-asserted-by":"publisher","DOI":"10.1002\/rth2.12741"},{"key":"e_1_2_11_28_1","doi-asserted-by":"publisher","DOI":"10.1097\/RHU.0000000000000123"},{"key":"e_1_2_11_29_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpainsymman.2008.05.009"},{"key":"e_1_2_11_30_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102753.2004.00505.x"},{"key":"e_1_2_11_31_1","doi-asserted-by":"publisher","DOI":"10.1017\/S2045796022000051"},{"key":"e_1_2_11_32_1","doi-asserted-by":"publisher","DOI":"10.1177\/1012690220934335"},{"key":"e_1_2_11_33_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.lanepe.2021.100265"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.14783","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,5,28]],"date-time":"2024-05-28T14:08:38Z","timestamp":1716905318000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.14783"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,3,28]]},"references-count":32,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2023,5]]}},"alternative-id":["10.1111\/hae.14783"],"URL":"https:\/\/doi.org\/10.1111\/hae.14783","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,3,28]]},"assertion":[{"value":"2022-12-06","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-03-13","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-03-28","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}