{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T23:09:43Z","timestamp":1791328183661,"version":"4.3.1"},"reference-count":25,"publisher":"BMJ","issue":"8","funder":[{"DOI":"10.13039\/501100000272","name":"National Institute for Health Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000272","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Open"],"accepted":{"date-parts":[[2019,6,12]]},"published-print":{"date-parts":[[2019,8]]},"abstract":"<jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>Haemophilia is a rare, inherited disorder in which blood does not clot normally, resulting in bleeding into joints and muscles. Long-term consequence is disabling joint pain, stiffness, muscle weakness, atrophy and reduced mobility. The purpose of this proposed feasibility of a randomised controlled trial (RCT) is to test the feasibility of an age-appropriate physiotherapy intervention designed to improve muscle strength, posture and the way boys use their joints during walking and everyday activities.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods and analysis<\/jats:title>\n                    <jats:p>A small-scale two-centre RCT of a 12-week muscle strengthening exercise intervention versus usual care for young children with haemophilia will be conducted. Primary outcomes will be safety and adherence to the exercise intervention. Secondary outcomes will include recruitment, retention and adverse event rates, clinical data, muscle strength, joint biomechanics and foot loading patterns during walking, 6 min timed walk, timed-up-and-down-stairs, EQ-5D-Y, participants\u2019 perceptions of the study, training requirements and relevant costs. Recruitment, follow-up, safety and adherence rates will be described as percentages. Participant diary and interview data will be analysed using a framework analysis. Demographic and disease variable distributions will be analysed for descriptive purposes and covariant analysis. Estimates of differences between treatment arms (adjusted for baseline) and 75% and 95% CIs will be calculated.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Ethics and dissemination<\/jats:title>\n                    <jats:p>The study has ethical approval from the London\u2014Fulham Research Ethics Committee (17\/LO\/2043) as well as Health Research Authority approval. As well as informing the design of the definitive trial, results of this study will be presented at local, national and international physiotherapy and haemophilia meetings as well as manuscripts submitted to peer-reviewed journals. We will also share the main findings of the study to all participants and the Haemophilia Society.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1136\/bmjopen-2019-029474","type":"journal-article","created":{"date-parts":[[2019,8,2]],"date-time":"2019-08-02T07:29:06Z","timestamp":1564730946000},"page":"e029474","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":5,"title":["Protocol for a feasibility randomised controlled trial of a musculoskeletal exercise intervention versus usual care for children with haemophilia"],"prefix":"10.1136","volume":"9","author":[{"given":"Ferhana","family":"Hashem","sequence":"first","affiliation":[{"name":"Centre for Health Service Studies, University of Kent, Canterbury, Kent, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Melanie","family":"Bladen","sequence":"additional","affiliation":[{"name":"Haemophilia Centre, Great Ormond Street Hospital For Children NHS Foundation Trust, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liz","family":"Carroll","sequence":"additional","affiliation":[{"name":"Haemophilia Society, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charlene","family":"Dodd","sequence":"additional","affiliation":[{"name":"Haemophilia Centre, East Kent Hospitals University NHS Foundation Trust, Canterbury, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wendy I","family":"Drechsler","sequence":"additional","affiliation":[{"name":"School of Population Health and Environmental Sciences, King\u2019s College London, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Lowery","sequence":"additional","affiliation":[{"name":"Centre for Health Service Studies, University of Kent, Canterbury, Kent, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vishal","family":"Patel","sequence":"additional","affiliation":[{"name":"Haemophilia Centre, Barts Health NHS Trust, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T","family":"Pellatt-Higgins","sequence":"additional","affiliation":[{"name":"Centre for Health Service Studies, University of Kent, Canterbury, Kent, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eirini","family":"Saloniki","sequence":"additional","affiliation":[{"name":"Centre for Health Service Studies, University of Kent, Canterbury, Kent, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6175-3343","authenticated-orcid":false,"given":"David","family":"Stephensen","sequence":"additional","affiliation":[{"name":"Haemophilia Centre, East Kent Hospitals University NHS Foundation Trust, Canterbury, UK"},{"name":"Haemophilia Centre, Barts Health NHS Trust, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2019,8,1]]},"reference":[{"key":"2025121221215834000_9.8.e029474.1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2008.01876.x"},{"key":"2025121221215834000_9.8.e029474.2","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2559.1999.00608.x"},{"key":"2025121221215834000_9.8.e029474.3","doi-asserted-by":"crossref","first-page":"571","DOI":"10.1111\/j.1365-2516.2010.02472.x","article-title":"Pathophysiology, diagnosis and prevention of arthropathy in patients with haemophilia","volume":"17","author":"Dunn","year":"2011","journal-title":"Haemophilia"},{"issue":"Suppl. 1","key":"2025121221215834000_9.8.e029474.4","doi-asserted-by":"crossref","first-page":"26","DOI":"10.1111\/j.1365-2516.2005.01151.x","article-title":"Rehabilitation of patients with haemophilia after orthopaedic surgery: a case study","volume":"11 Suppl 1","author":"Stephensen","year":"2005","journal-title":"Haemophilia"},{"key":"2025121221215834000_9.8.e029474.5","doi-asserted-by":"crossref","unstructured":"Iorio A , Marchesini E , Marcucci M , et al . 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