{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T02:39:28Z","timestamp":1787020768601,"version":"build-2736575974"},"reference-count":41,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2023,3,10]],"date-time":"2023-03-10T00:00:00Z","timestamp":1678406400000},"content-version":"vor","delay-in-days":68,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2023,3,10]],"date-time":"2023-03-10T00:00:00Z","timestamp":1678406400000},"content-version":"vor","delay-in-days":68,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100000659","name":"Research Trainees Coordinating Centre","doi-asserted-by":"publisher","award":["ICA-CDRF-2015-01-0120"],"award-info":[{"award-number":["ICA-CDRF-2015-01-0120"]}],"id":[{"id":"10.13039\/501100000659","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Journal of Foot and Ankle Research"],"published-print":{"date-parts":[[2023,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Haemophilia is an X\u2010linked recessive genetic disorder characterised by bleeding within soft tissue and joints. The ankle is disproportionally affected by haemarthropathy when compared to the elbows and knees; reported as the most affected joints in patients with haemophilia. Despite advances in treatment, patients still report ongoing pain and disability, however, the impact has not been evaluated, nor has the effect on health\u2010related quality of life (HRQoL) or foot and ankle patient\u2010reported outcome measures (PROMs). The primary aim of this study was to establish the impact of ankle haemarthropathy in patients with severe and moderate haemophilia A and B. Secondly to identify the clinical outcomes associated with a decline in HRQoL and foot and ankle PROMs.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A cross\u2010sectional multi\u2010centre questionnaire study was conducted across 18 haemophilia centres in England, Scotland and Wales with a recruitment target of 245 participants. The HAEMO\u2010QoL\u2010A and Manchester\u2010Oxford Foot Questionnaire (MOXFQ) (foot and ankle) with total and domain scores measured impact on HRQOL and foot and ankle outcomes. Demographics, clinical characteristics, ankle haemophilia joint health scores, multi\u2010joint haemarthropathy and Numerical Pain Rating Scales (NPRS) of \u201cankle pain over the past six months\u201d were collected as a measure of chronic ankle pain.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      A total of 243 of 250 participants provided complete data\n                      <jats:bold>.<\/jats:bold>\n                      HAEMO\u2010QoL\u2010A and MOXFQ (foot and ankle) total and index scores indicated worse HRQoL with total scores ranging from a mean of 35.3 to 35.8 (100 best\u2010health) and 50.5 to 45.8 (0 best\u2010health) respectively. NPRS (mean (SD)) ranged from 5.0 (2.6) to 5.5 (2.5), with median (IQR) ankle haemophilia joint health score of 4.5 (1 to 12.5) to 6.0 (3.0 to 10.0) indicating moderate to severe levels of ankle haemarthropathy. Ankle NPRS over six months and inhibitor status were associated with decline in outcome.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>HRQoL and foot and ankle PROMs were poor in participants with moderate to severe levels of ankle haemarthropathy. Pain was a major driver for decline in HRQoL and foot and ankle PROMs and use of NPRS has the potential to predict worsening HRQoL and PROMs at the ankle and other affected joints.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s13047-023-00611-5","type":"journal-article","created":{"date-parts":[[2023,3,10]],"date-time":"2023-03-10T09:04:24Z","timestamp":1678439064000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Decline in health\u2010related quality of life and foot and ankle patient reported outcomes measures in patients with haemophilia and ankle haemarthropathy"],"prefix":"10.1002","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1885-5472","authenticated-orcid":false,"given":"Richard A.","family":"Wilkins","sequence":"first","affiliation":[{"name":"Leeds Haemophilia Comprehensive Care Centre Leeds Teaching Hospitals NHS Trust Leeds UK"},{"name":"Leeds Institute of Rheumatic and Musculoskeletal Medicine University of Leeds Leeds UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heidi J.","family":"Siddle","sequence":"additional","affiliation":[{"name":"Leeds Institute of Rheumatic and Musculoskeletal Medicine University of Leeds Leeds UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Graham J.","family":"Chapman","sequence":"additional","affiliation":[{"name":"School of Sport and Health Sciences University of Central Lancashire Preston UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth","family":"Horn","sequence":"additional","affiliation":[{"name":"Leeds Haemophilia Comprehensive Care Centre Leeds Teaching Hospitals NHS Trust Leeds UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rebecca","family":"Walwyn","sequence":"additional","affiliation":[{"name":"Clinical Trials Research Unit Leeds Institute of Clinical Trials Research University of Leeds Leeds UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anthony C.","family":"Redmond","sequence":"additional","affiliation":[{"name":"Leeds Institute of Rheumatic and Musculoskeletal Medicine University of Leeds Leeds UK"},{"name":"NIHR Leeds Biomedical Research Centre Leeds Teaching Hospitals NHS Trust Leeds UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,3,10]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140\u20106736(03)13405\u20108"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102141.1958.tb03830.x"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM200106073442307"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14114"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12133"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.16704"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14201"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.13027"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1097\/MBC.0000000000000571"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102516.2008.01876.x"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood\u20102010\u201005\u2010284653"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102141.2008.07386.x"},{"issue":"5","key":"e_1_2_11_14_1","first-page":"401","article-title":"Musculo\u2010skeletal manifestations of haemophilia","volume":"30","author":"Rodriguez\u2010Merchan EC","year":"2016","journal-title":"Haemophilia"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.3390\/jcm6070063"},{"key":"e_1_2_11_16_1","first-page":"S18","volume-title":"Seminars in hematology","author":"Hoots WK"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.jfas.2012.06.005"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clinbiomech.2012.01.009"},{"key":"e_1_2_11_19_1","doi-asserted-by":"crossref","unstructured":"StephensenD DrechslerWI ScottOM. 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November 2022","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"1 March 2023","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"10 March 2023","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Ethical approval was obtained from the NHS Health Research Authority, UK (IRAS: 206141), and Research Ethics Committee (REC ID: 16\/LO\/2251).","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent for publication was obtained.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent to publication"}},{"value":"RAW has received conference registration fees and support for travel from Roche and speaker\u2019s fees from SOBI. RAW has received an HEE\/NIHR clinical doctoral research fellowship which funded this work. EH has received speaker fees from Roche and sponsorship for travel from Sobi. ACR is an NIHR Senior Investigator and has received funding from NIHR who also funded this research. HJS is an NIHR Senior Clinical lecturer and has received funding from NIHR who also funded this research.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"12"}}