{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T20:27:58Z","timestamp":1792787278108,"version":"4.5.0"},"reference-count":33,"publisher":"BMJ","issue":"10","license":[{"start":{"date-parts":[[2026,10,15]],"date-time":"2026-10-15T00:00:00Z","timestamp":1792022400000},"content-version":"unspecified","delay-in-days":14,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"AMC Support Inc"},{"name":"Fonds de recherche du Qu\u00e9bec-Sant\u00e9","award":["2024-2027"],"award-info":[{"award-number":["2024-2027"]}],"id":[{"id":"https:\/\/ror.org\/00w3qhf76","id-type":"ROR","asserted-by":"crossref"}]},{"name":"Fonds de recherche du Qu\u00e9bec-Sant\u00e9","award":["329833"],"award-info":[{"award-number":["329833"]}],"id":[{"id":"https:\/\/ror.org\/00w3qhf76","id-type":"ROR","asserted-by":"crossref"}]}],"content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Open"],"accepted":{"date-parts":[[2026,9,25]]},"published-print":{"date-parts":[[2026,10]]},"DOI":"10.1136\/bmjopen-2026-126300","type":"journal-article","created":{"date-parts":[[2026,10,15]],"date-time":"2026-10-15T12:40:38Z","timestamp":1792068038000},"page":"e126300","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Protocol for the development of a paediatric core outcome set for arthrogryposis multiplex congenita: an international multi-interest-holder consensus study"],"prefix":"10.1136","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0009-0003-4770-8268","authenticated-orcid":false,"given":"Naomi","family":"Zukerman","sequence":"first","affiliation":[{"name":"Faculty of Medicine and Health Sciences, McGill University, Montreal, Quebec, Canada"},{"name":"Clinical Research Department, Shriners Hospitals for Children Canada, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vasiliki Betty","family":"Darsaklis","sequence":"additional","affiliation":[{"name":"Clinical Research Department, Shriners Hospitals for Children Canada, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Clarice","family":"Araujo","sequence":"additional","affiliation":[{"name":"Faculty of Medicine and Health Sciences, McGill University, Montreal, Quebec, Canada"},{"name":"Clinical Research Department, Shriners Hospitals for Children Canada, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"Cachecho","sequence":"additional","affiliation":[{"name":"Clinical Research Department, Shriners Hospitals for Children Canada, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Camille","family":"Costa","sequence":"additional","affiliation":[{"name":"Faculty of Medicine and Health Sciences, McGill University, Montreal, Quebec, Canada"},{"name":"Clinical Research Department, Shriners Hospitals for Children Canada, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristen","family":"Donlevie","sequence":"additional","affiliation":[{"name":"Sargent College of Health and Rehabilitation Sciences, Boston University, Boston, Massachusetts, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alicja","family":"F\u0105fara","sequence":"additional","affiliation":[{"name":"Institute of Physiotherapy, Jagiellonian University Medical College, Krak\u00f3w, Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Usha","family":"Kasar","sequence":"additional","affiliation":[{"name":"Occupational Therapy Training School & Centre, Seth GSMC & KEM Hospital, Mumbai, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Courtney","family":"Krakie","sequence":"additional","affiliation":[{"name":"Department of Social Work, Shriners Hospital for Children, Sacramento, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francis","family":"Lacombe","sequence":"additional","affiliation":[{"name":"Technical Aids Services, Centre de R\u00e9adaptation Lucie-Bruneau, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Verity","family":"Pacey","sequence":"additional","affiliation":[{"name":"Department of Health Sciences, Faculty of Medicine, Health and Human Sciences, Macquarie University, Sydney, New South Wales, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8151-1341","authenticated-orcid":false,"given":"Jaclyn Megan","family":"Sions","sequence":"additional","affiliation":[{"name":"Department of Physical Therapy, University of Delaware, Newark, Delaware, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8567-7173","authenticated-orcid":false,"given":"No\u00e9mi","family":"Dahan-Oliel","sequence":"additional","affiliation":[{"name":"Faculty of Medicine and Health Sciences, McGill University, Montreal, Quebec, Canada"},{"name":"Clinical Research Department, Shriners Hospitals for Children Canada, Montreal, Quebec, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2026,10,15]]},"reference":[{"key":"2026101505401985000_16.10.e126300.1","article-title":"The landscape for rare diseases in 2024","volume":"12","year":"2024","journal-title":"Lancet Glob Health"},{"key":"2026101505401985000_16.10.e126300.2","unstructured":"Resolution adopted by the General Assembly on 29 November 2021\u2014social development. Contract No.: A\/RES\/76\/454. 2021."},{"key":"2026101505401985000_16.10.e126300.3","doi-asserted-by":"crossref","DOI":"10.1186\/s40900-022-00336-y","article-title":"Expert guidance for the rehabilitation of children with arthrogryposis: protocol using an integrated knowledge translation approach","volume":"8","author":"Dahan-Oliel","year":"2022","journal-title":"Res Involv Engagem"},{"key":"2026101505401985000_16.10.e126300.4","doi-asserted-by":"crossref","DOI":"10.1136\/bmjopen-2021-060591","article-title":"Epidemiology, aetiology, interventions and genomics in children with arthrogryposis multiplex congenita: protocol for a multisite registry","volume":"12","author":"Dahan-Oliel","year":"2022","journal-title":"BMJ Open"},{"key":"2026101505401985000_16.10.e126300.5","doi-asserted-by":"crossref","first-page":"1057","DOI":"10.1002\/bdra.20738","article-title":"Prevalence of multiple congenital contractures including arthrogryposis multiplex congenita in Alberta, Canada, and a strategy for classification and coding","volume":"88","author":"Lowry","year":"2010","journal-title":"Birth Defects Research"},{"key":"2026101505401985000_16.10.e126300.6","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31721"},{"key":"2026101505401985000_16.10.e126300.7","article-title":"Diagnosing arthrogryposis multiplex congenita: a review","volume":"2012","author":"Kalampokas","year":"2012","journal-title":"ISRN Obstet Gynecol"},{"key":"2026101505401985000_16.10.e126300.8","doi-asserted-by":"crossref","first-page":"84","DOI":"10.1016\/j.jclinepi.2023.07.002","article-title":"Assessment highlights need for improvement in standards of development of core outcome sets for rare genetic diseases","volume":"161","author":"Ciuc\u0103","year":"2023","journal-title":"J Clin Epidemiol"},{"key":"2026101505401985000_16.10.e126300.9","doi-asserted-by":"crossref","DOI":"10.1186\/s40900-021-00304-y","article-title":"Patient and family engagement in the development of core outcome sets for two rare chronic diseases in children","volume":"7","author":"Vanderhout","year":"2021","journal-title":"Res Involv Engagem"},{"key":"2026101505401985000_16.10.e126300.10","doi-asserted-by":"publisher","DOI":"10.1136\/archdischild-2016-312117"},{"key":"2026101505401985000_16.10.e126300.11","doi-asserted-by":"crossref","DOI":"10.1186\/s13063-021-05791-8","article-title":"Establishing a core outcome set for mucopolysaccharidoses (MPS) in children: study protocol for a rapid literature review, candidate outcomes survey, and Delphi surveys","volume":"22","author":"Howie","year":"2021","journal-title":"Trials"},{"key":"2026101505401985000_16.10.e126300.12","doi-asserted-by":"crossref","DOI":"10.1186\/s13023-021-01682-y","article-title":"A standard set of outcome measures for the comprehensive assessment of osteogenesis imperfecta","volume":"16","author":"Nijhuis","year":"2021","journal-title":"Orphanet J Rare Dis"},{"key":"2026101505401985000_16.10.e126300.13","doi-asserted-by":"crossref","first-page":"1131","DOI":"10.1001\/jamapediatrics.2022.3181","article-title":"Core outcome sets in child health","volume":"176","author":"Lei","year":"2022","journal-title":"JAMA Pediatr"},{"key":"2026101505401985000_16.10.e126300.14","doi-asserted-by":"crossref","DOI":"10.1186\/s13023-023-02667-9","article-title":"Barriers and facilitators to the implementation of guidelines in rare diseases: a systematic review","volume":"18","author":"Gittus","year":"2023","journal-title":"Orphanet J Rare Dis"},{"key":"2026101505401985000_16.10.e126300.15","doi-asserted-by":"crossref","first-page":"825","DOI":"10.1111\/hex.13054","article-title":"A co-designed framework to support and sustain patient and family engagement in health-care decision making","volume":"23","author":"McCarron","year":"2020","journal-title":"Health Expect"},{"key":"2026101505401985000_16.10.e126300.16","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pmed.1002447","article-title":"Core Outcome Set-STAndards for Development: the COS-STAD recommendations","volume":"14","author":"Kirkham","year":"2017","journal-title":"PLoS Med"},{"key":"2026101505401985000_16.10.e126300.17","doi-asserted-by":"crossref","DOI":"10.1186\/s13063-017-1978-4","article-title":"The COMET Handbook: version 1.0","volume":"18","author":"Williamson","year":"2017","journal-title":"Trials"},{"key":"2026101505401985000_16.10.e126300.18","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pmed.1002148","article-title":"Core Outcome Set-STAndards for Reporting: The COS-STAR Statement","volume":"13","author":"Kirkham","year":"2016","journal-title":"PLoS Med"},{"key":"2026101505401985000_16.10.e126300.19","doi-asserted-by":"crossref","first-page":"349","DOI":"10.1093\/intqhc\/mzm042","article-title":"Consolidated criteria for reporting qualitative research (COREQ): a 32-item checklist for interviews and focus groups. (1353-4505 (Print))","volume":"19","author":"Tong","year":"2007","journal-title":"Nt J Qual Health Care"},{"key":"2026101505401985000_16.10.e126300.20","doi-asserted-by":"crossref","DOI":"10.1186\/s40900-023-00479-6","article-title":"Stakeholder engagement in the development of an upper extremity outcome measure for children with rare musculoskeletal conditions","volume":"9","author":"Elfassy","year":"2023","journal-title":"Res Involv Engagem"},{"key":"2026101505401985000_16.10.e126300.21","doi-asserted-by":"crossref","first-page":"96","DOI":"10.1080\/09638288.2022.2161644","article-title":"Current rehabilitation practice for the evaluation and treatment of children with arthrogryposis: an international survey","volume":"46","author":"Cachecho","year":"2024","journal-title":"Disabil Rehabil"},{"key":"2026101505401985000_16.10.e126300.22","doi-asserted-by":"crossref","first-page":"1340","DOI":"10.1111\/dmcn.15898","article-title":"Common data elements for arthrogryposis multiplex congenita: an international framework","volume":"66","author":"Nematollahi","year":"2024","journal-title":"Develop Med Child Neuro"},{"key":"2026101505401985000_16.10.e126300.23","doi-asserted-by":"crossref","first-page":"168","DOI":"10.1186\/s13023-025-03671-x","article-title":"Consensus-based recommendations for the rehabilitation of children with arthrogryposis multiplex congenita: an integrated knowledge translation approach","volume":"20","author":"Dahan\u2011Oliel","year":"2025","journal-title":"Orphanet J Rare Dis"},{"key":"2026101505401985000_16.10.e126300.24","doi-asserted-by":"publisher","DOI":"10.3109\/09638288.2016.1145258"},{"key":"2026101505401985000_16.10.e126300.25","doi-asserted-by":"crossref","DOI":"10.1186\/s40900-023-00442-5","article-title":"Research evidence and implementation gaps in the engagement of people with lived experience in mental health and substance use research: a scoping review","volume":"9","author":"Hawke","year":"2023","journal-title":"Res Involv Engagem"},{"key":"2026101505401985000_16.10.e126300.26","unstructured":"Morgan D . Focus groups as qualitative research. Thousand Oaks, California: SAGE Publications, Inc, 1997. Available: https:\/\/methods.sagepub.com\/book\/mono\/focus-groups-as-qualitative-research\/toc https:\/\/methods.sagepub.com\/book\/mono\/preview\/focus-groups-as-qualitative-research.pdf"},{"key":"2026101505401985000_16.10.e126300.27","doi-asserted-by":"crossref","DOI":"10.1016\/j.jclinepi.2026.112262","article-title":"Sample size and consensus methods used in Delphi studies are heterogeneous: meta-research study","volume":"195","author":"\u0160krinjar","year":"2026","journal-title":"J Clin Epidemiol"},{"key":"2026101505401985000_16.10.e126300.28","doi-asserted-by":"crossref","unstructured":"Kuo SH , Tian C , McKay J , et al . n.d. Delphi study to elicit expert consensus around decision-making in the treatment of Friedreich ataxia. Front Neurol 16:1669059. doi:10.3389\/fneur.2025.1669059","DOI":"10.3389\/fneur.2025.1669059"},{"key":"2026101505401985000_16.10.e126300.29","doi-asserted-by":"publisher","DOI":"10.5662\/wjm.v11.i4.116"},{"key":"2026101505401985000_16.10.e126300.30","doi-asserted-by":"crossref","unstructured":"Webbe J , Allin B , Knight M , et al . How to reach agreement: the impact of different analytical approaches to Delphi process results in core outcomes set development. Trials 2023;24. doi:10.1186\/s13063-023-07285-1","DOI":"10.1186\/s13063-023-07285-1"},{"key":"2026101505401985000_16.10.e126300.31","doi-asserted-by":"crossref","DOI":"10.1186\/s13063-019-3265-z","article-title":"Multimorbidity risk assessment in adolescents and adults with cerebral palsy: a protocol for establishing a core outcome set for clinical research and practice","volume":"20","author":"McPhee","year":"2019","journal-title":"Trials"},{"key":"2026101505401985000_16.10.e126300.32","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/kez214"},{"key":"2026101505401985000_16.10.e126300.33","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31705"}],"container-title":["BMJ Open"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bmjopen-2026-126300","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,15]],"date-time":"2026-10-15T12:40:46Z","timestamp":1792068046000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmjopen.bmj.com\/lookup\/doi\/10.1136\/bmjopen-2026-126300"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,10]]},"references-count":33,"journal-issue":{"issue":"10","published-online":{"date-parts":[[2026,10,15]]},"published-print":{"date-parts":[[2026,10]]}},"alternative-id":["10.1136\/bmjopen-2026-126300"],"URL":"https:\/\/doi.org\/10.1136\/bmjopen-2026-126300","relation":{},"ISSN":["2044-6055","2044-6055"],"issn-type":[{"value":"2044-6055","type":"print"},{"value":"2044-6055","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,10]]}}}