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Protocol for the development of a paediatric core outcome set for arthrogryposis multiplex congenita: an international multi-interest-holder consensus study

Year: 2026

Type: article

Abstract: Introduction Ongoing research for arthrogryposis multiplex congenita (AMC) advocates for holistic and family-centred interventions. However, clinicians working with children with AMC are often met with gaps in the literature and a lack of assessment guidelines, impacting services. Current limitations in clinical practice demonstrate the need for consensus on which outcome measures to use in paediatric rehabilitation for AMC. This multiphasic study aims to develop a core outcome set (COS) for a group of rare musculoskeletal diseases (AMC), fostering standardised and inclusive care practices. Methods and analysis This study follows a mixed-method multiphasic approach guided by the COMET Initiative’s standards for core outcome set (COS) development and COMET Handbook V.1.0. An international advisory panel was established, representing clinicians and individuals with lived experience across Canada, the United States, Poland, Australia and India. Preliminary COS concepts were derived from multidisciplinary research activities conducted between 2016 and 2024. A scoping review and advisory panel consultation supported outcome measure selection and refinement for the AMC population. Five candidate outcome measures were mapped item-by-item to the International Classification of Functioning, Disability and Health (ICF) to assess functional domain coverage and alignment with AMC rehabilitation needs. The resulting draft COS will now guide the consensus-building phase, including modified Delphi rounds, international focus groups and individual interviews, followed by external methodological appraisal. Ethics and dissemination Ethics approval has been provided by McGill University’s Research Ethics Board (effective 21 July 2026; submission 26-05-029-01). The COS will be shared at AMC support organisation meetings, conferences and webinars, with additional knowledge mobilisation activities selected by the international advisory panel. The dissemination of findings will also be guided by the feedback provided during focus groups and interviews, to best address the needs of varied populations and ensure the accessibility of information across settings. OSF registration 10.17605/OSF.IO/QG89H. COMET registration https://www.comet-initiative.org/Studies/Details/3378.

Source: BMJ Open

DOI: 10.1136/bmjopen-2026-126300