{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T08:06:18Z","timestamp":1789632378220,"version":"build-2803163510"},"reference-count":35,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T00:00:00Z","timestamp":1789603200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T00:00:00Z","timestamp":1789603200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100000406","name":"Motor Neurone Disease Association","doi-asserted-by":"publisher","award":["Mazzucco\/Oct24\/2410-794"],"award-info":[{"award-number":["Mazzucco\/Oct24\/2410-794"]}],"id":[{"id":"10.13039\/501100000406","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Res Involv Engagem"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Up to 70% of people living with Motor Neuron Disease (plwMND) report distressing episodes of choking (a feeling of suffocation or airways blockage) due to involvement of bulbar muscles. There is an evidence gap on management of choking in plwMND, which is mirrored by the absence of guidance and training for healthcare professionals. Choking in plwMND is associated with other signs of bulbar involvement, such as communication difficulties due to dysarthria, which can be a barrier to effective participation in research. In this study protocol, we describe an innovative approach to meaningful involvement of plwMND, to co-produce a research project on management of choking tailored to their needs and aligning with their priorities.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Phase 1 (Patient and public involvement co-design meeting): We first engaged in a meeting with plwMND, carers and healthcare professionals, to identify their priorities in relation to improving management of choking in MND. To ensure inclusive participation, we used the Theory of Change framework; people with communication difficulties were supported by a Speech and Language Therapist through their chosen augmentative and alternative communication aids. A key priority identified was the need to produce evidence-based guidance for management of choking tailored to the needs of plwMND. Phase 2 (Healthcare research study protocol on management of choking): based on the outcome of Phase 1, a multidisciplinary team (including individuals with lived experience) designed a mixed-methods, multi-centre research project based on four work packages (WPs). WP1: systematic review of the existing scientific literature on management of choking; WP2: interviews and focus groups with people with lived experience of choking in MND; WP3: co-design of a clinical algorithm detailing interventions to manage choking, based on WP1 and 2; WP4: algorithm development and dissemination with continuous involvement and input from people with lived experience through a Patient and Public Involvement co-applicant and patients\u2019 representatives.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>In plwMND and choking, barriers to inclusion in PPI and research range from reduced accessibility due to physical disabilities, to discriminatory sampling due to communication issues. Through inclusive engagement at the outset, plwMND highlighted a mismatch between their priorities (gaining knowledge about choking and feeling empowered to manage it at home) and available evidence (lack of guidance and training for healthcare professionals). This research project aligns with what matters to most affected patients, their carers and healthcare professionals, aiming to fill an evidence gap through new guidance to manage choking tailored to their needs.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s40900-026-00974-6","type":"journal-article","created":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T07:50:55Z","timestamp":1789631455000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Management of choking in Motor Neuron Disease: patient and public involvement in a co-produced mixed-methods study protocol"],"prefix":"10.1186","volume":"12","author":[{"given":"Dorinda","family":"Moffatt","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lisa","family":"Hinton","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lucinda","family":"Spurway","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Angela","family":"Martin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Neil","family":"Drinkwater","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donna","family":"Noonan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claire","family":"Rose","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara","family":"Mazzucco","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,9,17]]},"reference":[{"key":"974_CR1","doi-asserted-by":"publisher","first-page":"1083","DOI":"10.1016\/S1474-4422(18)30404-6","volume":"17","author":"GBD 2016 Motor Neuron Disease Collaborators","year":"2018","unstructured":"GBD 2016 Motor Neuron Disease Collaborators. Global, regional, and national burden of motor neuron diseases 1990\u20132016: a systematic analysis for the Global Burden of Disease Study 2016. Lancet Neurol. 2018;17:1083\u201397.","journal-title":"Lancet Neurol"},{"key":"974_CR2","doi-asserted-by":"publisher","first-page":"303","DOI":"10.1136\/jnnp.2009.188151","volume":"9","author":"K Talbot","year":"2009","unstructured":"Talbot K. Motor neuron disease. Pract Neurol. 2009;9:303\u20139.","journal-title":"Pract Neurol"},{"key":"974_CR3","doi-asserted-by":"publisher","first-page":"601","DOI":"10.1136\/jnnp.68.5.601","volume":"68","author":"S Hadjikoutis","year":"2000","unstructured":"Hadjikoutis S, Eccles R, Wiles CM. Coughing and choking in motor neuron disease. J Neurol Neurosurg Psychiatry. 2000;68:601\u20134.","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"974_CR4","doi-asserted-by":"publisher","first-page":"400","DOI":"10.1002\/mus.27466","volume":"65","author":"RD Gotesman","year":"2022","unstructured":"Gotesman RD, Lalonde E, McKim DAat al. Laryngospasm in amyotrophic lateral sclerosis. Muscle Nerve. 2022;65:400\u20134.","journal-title":"Muscle Nerve"},{"key":"974_CR5","doi-asserted-by":"crossref","first-page":"1329","DOI":"10.1001\/archneurol.2009.250","volume":"66","author":"MM Van der Graaff","year":"2009","unstructured":"Van der Graaff MM, Grolman W, Westermann EJ, et al. Vocal Cord Dysfunction in Amyotrophic Lateral Sclerosis. Arch Neurol. 2009;66:1329\u201333.","journal-title":"Arch Neurol"},{"key":"974_CR6","doi-asserted-by":"publisher","first-page":"252","DOI":"10.3390\/brainsci12020252","volume":"12","author":"SK Bublitz","year":"2022","unstructured":"Bublitz SK, Mie E, Wasner M, et al. Thick Mucus in ALS: AMixed-Method Study on Associated Factors and Its Impact on Quality of Life of Patients and Caregivers. Brain Sci. 2022;12:252.","journal-title":"Brain Sci"},{"key":"974_CR7","doi-asserted-by":"publisher","DOI":"10.1080\/09638288.2025.2555973","author":"C Massey","year":"2025","unstructured":"Massey C, Hobson E, McDermott C, Griffiths AW. Living with cough and secretion issues: the experiences of people with amyotrophic lateral sclerosis and their caregivers. Disabil Rehabil. 2025. https:\/\/doi.org\/10.1080\/09638288.2025.2555973.","journal-title":"Disabil Rehabil"},{"key":"974_CR8","unstructured":"Motor Neurone Disease Association. A professionals\u2019 guide to end of life care in motor neurone disease. A professionals\u2019 guide to end of life care in MND. Accessed 10 March 2026."},{"key":"974_CR9","doi-asserted-by":"publisher","first-page":"2381","DOI":"10.1016\/S0140-6736(24)00533-6","volume":"403","author":"RL Gould","year":"2024","unstructured":"Gould RL, McDermott CJ, Thompson BJ, et al. Acceptance and Commitment Therapy plus usual care for improving quality of life in people with motor neuron disease (COMMEND): a multicentre, parallel, randomised controlled trial in the UK. Lancet. 2024;403:2381\u201394.","journal-title":"Lancet"},{"key":"974_CR10","unstructured":"National Institute for Health and Care Excellence. Motor neurone disease: assessment and management. https:\/\/www.nice.org.uk\/guidance\/NG42\/chapter\/Recommendations#respiratory-function-and-respiratory-symptoms. Accessed on July 21st 2026."},{"key":"974_CR11","unstructured":"Motor Neurone Disease Association. Swallowing difficulties. Accessed 10 March 2026."},{"key":"974_CR12","unstructured":"Resuscitation council UK. available at Adult choking algorithm 2025.pdf. Accessed 10 March 2026."},{"key":"974_CR13","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1186\/s13063-023-07142-1","volume":"24","author":"S Shariq","year":"2023","unstructured":"Shariq S, Cardoso Pinto AM, Budhathoki SS, Miller M, Cro S. Barriers and facilitators to the recruitment of disabled people to clinical trials: a scoping review. Trials. 2023;24:171.","journal-title":"Trials"},{"key":"974_CR14","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1186\/s40900-021-00260-7","volume":"7","author":"K Broomfield","year":"2021","unstructured":"Broomfield K, Craig C, Smith S, Jones G, Judge S, Sage K. Creativity in public involvement: supporting authentic collaboration and inclusive research with seldom heard voices. Res Involv Engagem. 2021;7:17.","journal-title":"Res Involv Engagem"},{"key":"974_CR15","doi-asserted-by":"publisher","first-page":"e0258575","DOI":"10.1371\/journal.pone.0258575","volume":"16","author":"C Jagoe","year":"2021","unstructured":"Jagoe C, McDonald C, Rivas M, Groce N. Direct participation of people with communication disabilities in research on poverty and disabilities in low and middle income countries: A critical review. PLoS ONE. 2021;16:e0258575.","journal-title":"PLoS ONE"},{"key":"974_CR16","doi-asserted-by":"publisher","first-page":"1","DOI":"10.3109\/17549507.2012.745164","volume":"15","author":"K Wylie","year":"2013","unstructured":"Wylie K, McAllister L, Davidson B, Marshall J. Changing practice: Implications of the World Report on Disability for responding to communication disability in under-served populations. Int J Speech-Lang Pathol. 2013;15:1\u201313.","journal-title":"Int J Speech-Lang Pathol"},{"key":"974_CR17","first-page":"1260","volume":"31","author":"M Jayeshttps","year":"2021","unstructured":"Jayeshttps M, Moulam L, Meredith S, et al. Making Public Involvement in Research More Inclusive of People With Complex Speech and Motor Disorders: The I-ASC Project Qual. Health Res. 2021;31:1260\u201374.","journal-title":"Health Res"},{"key":"974_CR18","volume-title":"World Report on Disability","author":"World Health Organization and the World Bank","year":"2011","unstructured":"World Health Organization and the World Bank. World Report on Disability. Geneva: World Health Organization; 2011. Available at: World Report on Disability. Accessed on 23 March 2026."},{"key":"974_CR19","unstructured":"Andersen A. A community builder\u2019s approach to theory of change: a practical guide to theory development. New York: The Aspen Institute; 2004. Available at The Community Builder\u2019s Approach to Theory of Change: A Practical Guide to Theory Development - Aspen Institute. Accessed 10 March 2026."},{"key":"974_CR20","doi-asserted-by":"publisher","first-page":"103850","DOI":"10.1016\/j.envsci.2024.103850","volume":"160","author":"BM Belcher","year":"2024","unstructured":"Belcher BM, Bonaiuti E, Thiele G. Applying Theory of Change in research program planning: Lessons from CGIAR. Environ Sci Policy. 2024;160:103850.","journal-title":"Environ Sci Policy"},{"key":"974_CR21","doi-asserted-by":"publisher","first-page":"63","DOI":"10.1186\/s13012-016-0422-6","volume":"11","author":"E Breuer","year":"2016","unstructured":"Breuer E, Lee L, De Silva M, Lund C. Using theory of change to design and evaluate public health interventions: a systematic review. Implement Sci. 2016;11:63.","journal-title":"Implement Sci"},{"key":"974_CR22","doi-asserted-by":"publisher","first-page":"e291","DOI":"10.1016\/S2214-109X(25)00438-3","volume":"14","author":"J Mannell","year":"2026","unstructured":"Mannell J, Jama Shai N, Chimbindi N, Gibbs A. Measuring co-design in global health research: methodological challenges and decolonial innovations. Lancet Glob Health. 2026;14:e291\u201395.","journal-title":"Lancet Glob Health"},{"key":"974_CR23","doi-asserted-by":"publisher","first-page":"13","DOI":"10.1186\/s40900-017-0062-2","volume":"3","author":"S Staniszewska","year":"2017","unstructured":"Staniszewska S, Brett J, Simera I, et al. GRIPP2 reporting checklists: tools to improve reporting of patient and public involvement in research. Res Involv Engagem. 2017;3:13.","journal-title":"Res Involv Engagem"},{"key":"974_CR24","unstructured":"Great Western Hospital. Motor Neurone Disease patients share their experiences to drive improvements in care. https:\/\/www.bbc.co.uk\/news\/articles\/cg70e87d0v4o. Accessed July 21st 2026."},{"key":"974_CR25","doi-asserted-by":"publisher","unstructured":"Robert G, Locock L, Williams O, et al. Co-producing and co-designing. Cambridge University Press, 2022. Available online at https:\/\/doi.org\/10.1017\/9781009237024. Accessed July 21st 2026.","DOI":"10.1017\/9781009237024"},{"key":"974_CR26","doi-asserted-by":"publisher","first-page":"n71","DOI":"10.1136\/bmj.n71","volume":"372","author":"MJ Page","year":"2021","unstructured":"Page MJ, McKenzie JE, Bossuyt PM, et al. The PRISMA 2020 statement: An updated guideline for reporting systematic reviews. BMJ. 2021;372:n71.","journal-title":"BMJ"},{"key":"974_CR27","unstructured":"Higgins JPT, Thomas J, Chandler J, Cumpston M, Li T, Page MJ, Welch VA, editors. Cochrane Handbook for Systematic Reviews of Interventions version 6.4 (updated August 2023). Available from Cochrane Handbook for Systematic Reviews of Interventions | Cochrane. (last accessed on 10\/03\/26)."},{"key":"974_CR28","doi-asserted-by":"publisher","first-page":"405","DOI":"10.1111\/j.1365-2929.2006.02467.x","volume":"40","author":"S Ziebland","year":"2006","unstructured":"Ziebland S, McPherson A. Making sense of qualitative data analysis: an introduction with illustrations from DIPEx (personal experiences of health and illness). Med Educ. 2006;40:405\u201314.","journal-title":"Med Educ"},{"key":"974_CR29","doi-asserted-by":"crossref","unstructured":"Locock L, Robert G, Boaz A, Shuldham C, Fielden J, Ziebland S. Testing accelerated experience based co-design: using a national archive of patient experience narrative interviews to promote rapid patient-centred service improvement. In: Patient-Centred Health Care: Achieving Co-ordination, Communication and Innovation 2013 (pp. 173\u201385). London: Palgrave Macmillan UK.","DOI":"10.1057\/9781137308931_14"},{"key":"974_CR31","doi-asserted-by":"publisher","first-page":"258","DOI":"10.1136\/bmjqs-2023-016196","volume":"33","author":"M Woodward","year":"2024","unstructured":"Woodward M, Dixon-Woods M, Randall W, Walker C, Hughes C, Blackwell S, Dewick L, Bahl R, Draycott T, Winter C, Ansari A. How to co-design a prototype of a clinical practice tool: a framework with practical guidance and a case study. BMJ Qual Saf. 2024;33:258\u201370.","journal-title":"BMJ Qual Saf"},{"key":"974_CR32","doi-asserted-by":"publisher","first-page":"511","DOI":"10.1080\/21678421.2019.1643373","volume":"20","author":"LS Musson","year":"2019","unstructured":"Musson LS, McDermott CJ, Hobson EV. Exploring patient and public involvement in motor neuron disease research. Amyotroph Lateral Scler Frontotemporal Degener. 2019;20:511\u201320.","journal-title":"Amyotroph Lateral Scler Frontotemporal Degener"},{"key":"974_CR33","doi-asserted-by":"publisher","first-page":"626","DOI":"10.1136\/bmjqs-2015-004839","volume":"25","author":"J Ocloo","year":"2016","unstructured":"Ocloo J, Matthews R. From tokenism to empowerment: progressing patient and public involvement in healthcare improvement. BMJ Qual Saf. 2016;25:626\u201332.","journal-title":"BMJ Qual Saf"},{"key":"974_CR34","doi-asserted-by":"publisher","first-page":"283","DOI":"10.1186\/s12888-025-06608-7","volume":"25","author":"A Werner","year":"2025","unstructured":"Werner A, Lauberger J, Steckelberg A, Meyer G. Patient and public involvement in basic and clinical psychiatric research: a scoping review of reviews. BMC Psychiatry. 2025;25:283.","journal-title":"BMC Psychiatry"},{"key":"974_CR35","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1177\/16094069241251537","volume":"23","author":"E Wilson","year":"2024","unstructured":"Wilson E, Turner N. Expanding Qualitative Interviewing for Studies Involving Adults With Different Communication Needs: Reflections on Research With People Living With Motor Neurone Disease. Int J Qual Methods. 2024;23:1\u201310.","journal-title":"Int J Qual Methods"},{"key":"974_CR36","doi-asserted-by":"publisher","first-page":"26","DOI":"10.1186\/s40900-026-00846-z","volume":"12","author":"D Holford","year":"2026","unstructured":"Holford D, Hemingway C, Ozano Kat al. Patient and public involvement, engagement, and participation in practice: co-production of a creative health approach and theory of change through the ReCITE consortium-building project in Liverpool. Res Involv Engagem. 2026;12:26.","journal-title":"Res Involv Engagem"}],"container-title":["Research Involvement and Engagement"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s40900-026-00974-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s40900-026-00974-6","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s40900-026-00974-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T07:51:02Z","timestamp":1789631462000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1186\/s40900-026-00974-6"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,9,17]]},"references-count":35,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,12]]}},"alternative-id":["974"],"URL":"https:\/\/doi.org\/10.1186\/s40900-026-00974-6","relation":{},"ISSN":["2056-7529"],"issn-type":[{"value":"2056-7529","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,9,17]]},"assertion":[{"value":"23 April 2026","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"28 August 2026","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"17 September 2026","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Informed consent was signed by all participants. This study was approved by Health and Care Research Wales (IRAS 360194; REC 25\/PR\/1142) on 1\/12\/2025.","order":1,"name":"Ethics","label":"Ethics approval and consent to participate","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Not applicable.","order":2,"name":"Ethics","label":"Consent for publication","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors declare no competing interests.","order":3,"name":"Ethics","label":"Competing interests","group":{"name":"EthicsHeading","label":"Declarations"}}],"article-number":"147"}}