{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,12]],"date-time":"2026-09-12T10:08:59Z","timestamp":1789207739693,"version":"build-2803163510"},"reference-count":46,"publisher":"National Institute for Health and Care Research","license":[{"start":{"date-parts":[[2026,7,16]],"date-time":"2026-07-16T00:00:00Z","timestamp":1784160000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/www.nationalarchives.gov.uk\/doc\/open-government-licence\/version\/3\/"}],"funder":[{"name":"National Institute for Health and Care Research Advanced Fellowship","award":["NIHR303636"],"award-info":[{"award-number":["NIHR303636"]}]},{"DOI":"10.13039\/100015652","name":"Barts Charity","doi-asserted-by":"crossref","award":["G-002757"],"award-info":[{"award-number":["G-002757"]}],"id":[{"id":"10.13039\/100015652","id-type":"DOI","asserted-by":"crossref"}]},{"name":"National Institute for Health and Care Research Barts Biomedical Research Centre","award":["NIHR203330"],"award-info":[{"award-number":["NIHR203330"]}]}],"content-domain":{"domain":["openresearch.nihr.ac.uk"],"crossmark-restriction":false},"short-container-title":["NIHR Open Res"],"abstract":"<ns3:p>Introduction Dystonia is a movement disorder characterised by abnormal movements and\/or postures, initiated or worsened by voluntary action. Non-degenerative childhood dystonia is common, represents a substantial source of lifelong disability, and is often associated with other motor and non-motor phenotypes that can contribute to functional impairments. However, there is no consensus on how to measure motor and non-motor outcomes associated with dystonia, nor is there consensus on what outcomes to measure. This prevents adequate evaluation and development of evidence-based interventions and decision-making in clinical practice. Here, we present the protocol for establishing global expert consensus on the Dystonia Impact Core Outcome Set (DI-COS) to describe what dystonia-associated outcomes to measure and how to measure them. Methods and analysis The DI-COS study includes a mixed-methods programme with three phases. Phase 1 includes; (i) scoping review, (ii) qualitative interview study with children and families, and (iii) international survey of health, social care, and education-based professionals. Phase 2 is a three round international e-Delphi study informed by Phase 1, and expert consensus from: (i) a professional panel and (ii) experts with lived experience (parents and children). Content and thematic analysis, and descriptive statistics will be used to analyse qualitative and quantitative data, respectively. Results will be triangulated to inform development of the Delphi survey in Phase 2. Phase 3 will identify the outcome measures available and recommend assessment outcomes agreed as part of Phase 2 Consensus. Ethics and dissemination Ethical approval has been obtained from Camden and King\u2019s Cross Research Ethics Committee (25\/LO\/0150) and Queen Mary, University of London Research Ethics Committee. All dissemination activities (including papers submitted to peer-reviewed journals, abstracts submitted to national\/international conferences, visual outputs and engagement events), will be co-produced, co-designed and co-presented\/authored with members of the study\u2019s advisory groups and shared via project\u2019s networks.<\/ns3:p>","DOI":"10.3310\/nihropenres.14354.1","type":"journal-article","created":{"date-parts":[[2026,7,16]],"date-time":"2026-07-16T10:04:05Z","timestamp":1784196245000},"page":"92","update-policy":"https:\/\/doi.org\/10.3310\/nihropenres.crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Improving Measurement and Priorities for Assessment in Clinical Trials of childhood-onset Dystonia International Core Outcome Set (Dystonia IMPACT-COS): A protocol"],"prefix":"10.3310","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1488-6415","authenticated-orcid":false,"given":"Hortensia","family":"Gimeno","sequence":"first","affiliation":[{"name":"Childhood Participation Lab, Centre for Bone and Joint Health London England E1 2AT Queen Mary University of London Faculty of Medicine and Dentistry"},{"name":"The Royal London Hospital and Tower Hamlets Community Children\u2019s Therapy Services London England E1 2AT Barts Health NHS Trust"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1243-3500","authenticated-orcid":false,"given":"Hannah","family":"Scott","sequence":"additional","affiliation":[{"name":"Childhood Participation Lab, Centre for Bone and Joint Health London England E1 2AT Queen Mary University of London Faculty of Medicine and Dentistry"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Khamani","family":"Edwards","sequence":"additional","affiliation":[{"name":"Childhood Participation Lab, Centre for Bone and Joint Health London England E1 2AT Queen Mary University of London Faculty of Medicine and Dentistry"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eleanor P","family":"Redding","sequence":"additional","affiliation":[{"name":"Childhood Participation Lab, Centre for Bone and Joint Health London England E1 2AT Queen Mary University of London Faculty of Medicine and Dentistry"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Enna Thea","family":"Kul-Want","sequence":"additional","affiliation":[{"name":"Childhood Participation Lab, Centre for Bone and Joint Health London England E1 2AT Queen Mary University of London Faculty of Medicine and 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Mary University of London Faculty of Medicine and Dentistry"},{"name":"Children\u2019s Integrated Therapies Team Dartford England Oxleas NHS Foundation Trust"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Barbara","family":"Gon\u00e7alves","sequence":"additional","affiliation":[{"name":"Childhood Participation Lab, Centre for Bone and Joint Health London England E1 2AT Queen Mary University of London Faculty of Medicine and Dentistry"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bhooma R","family":"Aravamuthan","sequence":"additional","affiliation":[{"name":"St. Louis Missouri Washington University in St Louis School of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paula S C","family":"Chagas","sequence":"additional","affiliation":[{"name":"Faculty of Physical Therapy Juiz de Fora State of Minas Gerais Universidade Federal de Juiz de Fora Faculdade de Medicina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5783-571X","authenticated-orcid":false,"given":"Marina","family":"de Koning-Tijssen","sequence":"additional","affiliation":[{"name":"Department of Neurology, University Medical Center Groningen (UMCG) Groningen Groningen University of Groningen"},{"name":"Expertise Centre Movement Disorders Groningen Groningen University Medical Centre Groningen"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hendriekje","family":"Eggink","sequence":"additional","affiliation":[{"name":"Department of Neurology, University Medical Center Groningen (UMCG) Groningen Groningen University of Groningen"},{"name":"Expertise Centre Movement Disorders Groningen Groningen University Medical Centre 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The views expressed in this publication are those of the author(s) and not necessarily those of the National Institute for Health and Care Research, National Health Service, or the UK Department of Health and Social Care. Part of the work reported here is funded by seed grant from Barts Charity (G-002757). This work acknowledges the support of the National Institute for Health and Care Research Barts Biomedical Research Centre (NIHR203330); a delivery partnership of Barts Health NHS Trust, Queen Mary University of London, St George\u2019s University Hospitals NHS Foundation Trust and St George\u2019s University of London. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.","order":2,"name":"grant-information","label":"Grant Information"},{"value":"This open access work is licensed under the Open Government Licence v3.0.","order":0,"name":"copyright-info","label":"Copyright"}]}}