{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,4]],"date-time":"2026-06-04T05:09:19Z","timestamp":1780549759622,"version":"3.54.1"},"reference-count":51,"publisher":"SAGE Publications","issue":"3","license":[{"start":{"date-parts":[[2025,12,16]],"date-time":"2025-12-16T00:00:00Z","timestamp":1765843200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2025,12,16]],"date-time":"2025-12-16T00:00:00Z","timestamp":1765843200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/501100000033","name":"Institute of Musculoskeletal Health and Arthritis","doi-asserted-by":"publisher","award":["OR2-189333"],"award-info":[{"award-number":["OR2-189333"]}],"id":[{"id":"10.13039\/501100000033","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000223","name":"Muscular Dystrophy Canada","doi-asserted-by":"publisher","award":["Network Catalyst Grant: NG2-170044"],"award-info":[{"award-number":["Network Catalyst Grant: NG2-170044"]}],"id":[{"id":"10.13039\/501100000223","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Journal of Neuromuscular Diseases"],"published-print":{"date-parts":[[2026,5]]},"abstract":"<jats:p>Patient-oriented research is increasingly recognized as an important methodology in health sciences. Benefits of patient engagement include aligning research priorities to those living with health conditions, developing better recruitment strategies and protocols, and integrating findings more meaningfully. However, for research teams to work well with patient-partners, training for all stakeholders is needed. While training exists, none consider the uniqueness of the neuromuscular disease experience. To address this gap, our team of researchers, clinicians, and patient-partners collaborated to increase the capacity for patient-engagement in neuromuscular disease research. Our methods included: 1) conducting a landscape of available resources, and 2) using adult education principles and a backwards design process to develop unique training modules. The result is an online platform with three modules focusing on the neuromuscular disease context, addressing the inclusion, diversity, equity, and accessibility needs of those with neuromuscular diseases, and building teamwork skills. Early evaluation of the first two modules indicates high satisfaction and knowledge gain. Through this process, we have learned about the barriers of patient-oriented research, how to support the required system culture shift, and how to plan for long-term sustainability.<\/jats:p>","DOI":"10.1177\/22143602251405819","type":"journal-article","created":{"date-parts":[[2025,12,16]],"date-time":"2025-12-16T16:03:53Z","timestamp":1765901033000},"page":"462-473","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Building capacity for patient-engagement in neuromuscular disease research: A network project"],"prefix":"10.1177","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6071-4337","authenticated-orcid":false,"given":"Patricia","family":"Mortenson","sequence":"first","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"},{"name":"Department of Occupational Science &amp; Occupational Therapy, University of British Columbia, Vancouver, Canada"},{"name":"British Columbia Children's Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-4225-9075","authenticated-orcid":false,"given":"Homira","family":"Osman","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"},{"name":"Muscular Dystrophy Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-5392-7915","authenticated-orcid":false,"given":"Erin","family":"Beattie","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Corinne","family":"Kagan","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Victoria","family":"Larocca","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"},{"name":"Muscular Dystrophy Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claudia","family":"Maltais","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Linda","family":"Niksic","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Margo","family":"Thompson","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9239-1704","authenticated-orcid":false,"given":"Kathryn","family":"Selby","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario Research Institute"},{"name":"British Columbia Children's Hospital"},{"name":"University of British Columbia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2025,12,16]]},"reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-022-02357-y"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1177\/2054358120979255"},{"key":"e_1_3_3_4_2","doi-asserted-by":"publisher","DOI":"10.1377\/hlthaff.2018.05067"},{"key":"e_1_3_3_5_2","doi-asserted-by":"publisher","DOI":"10.2215\/CJN.16561221"},{"key":"e_1_3_3_6_2","first-page":"1","article-title":"Patient engagement in Canada: a scoping review of the \u2018how\u2019and \u2018what\u2019of patient engagement in health research","volume":"16","author":"Manafo E","year":"2018","unstructured":"Manafo E, Petermann L, Mason-Lai P, et al. Patient engagement in Canada: a scoping review of the \u2018how\u2019and \u2018what\u2019of patient engagement in health research. Health Res Policy Syst 2018; 16: 1\u201311.","journal-title":"Health Res Policy Syst"},{"key":"e_1_3_3_7_2","first-page":"7","article-title":"Research is care","volume":"42","author":"King N","year":"2012","unstructured":"King N. Research is care. Hastings Cent Rep 2012; 42: 7.","journal-title":"Hastings Cent Rep"},{"key":"e_1_3_3_8_2","volume-title":"Understanding disability: from theory to practice","author":"Oliver M","year":"2018","unstructured":"Oliver M. Understanding disability: from theory to practice. 2nd ed. London: Bloomsbury publishing, 2018.","edition":"2"},{"key":"e_1_3_3_9_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-019-0141-7"},{"key":"e_1_3_3_10_2","unstructured":"Canadian Institutes of Health Research (CIHR). Strategy for patient-oriented research: patient engagement framework https:\/\/www.cihr-irsc.gc.ca\/e\/41204.html (2015 accessed 23 February 2025)."},{"key":"e_1_3_3_11_2","doi-asserted-by":"publisher","DOI":"10.1177\/2054358117703070"},{"key":"e_1_3_3_12_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-020-0180-0"},{"key":"e_1_3_3_13_2","doi-asserted-by":"publisher","DOI":"10.1136\/bmjopen-2018-022154"},{"key":"e_1_3_3_14_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-021-00319-5"},{"key":"e_1_3_3_15_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-021-00299-6"},{"key":"e_1_3_3_16_2","doi-asserted-by":"publisher","DOI":"10.1111\/hex.12635"},{"key":"e_1_3_3_17_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-018-0110-6"},{"key":"e_1_3_3_18_2","doi-asserted-by":"publisher","DOI":"10.1186\/s12961-024-01106-w"},{"key":"e_1_3_3_19_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-019-1103-8"},{"key":"e_1_3_3_20_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-023-00535-1"},{"key":"e_1_3_3_21_2","first-page":"687","article-title":"Focus: rare disease: patients as partners in rare disease diagnosis and research","volume":"94","author":"McCray AT","year":"2021","unstructured":"McCray AT, LeBlanc K, Network UD. Focus: rare disease: patients as partners in rare disease diagnosis and research. Yale J Biol Med 2021; 94: 687.","journal-title":"Yale J Biol Med"},{"key":"e_1_3_3_22_2","first-page":"24","article-title":"Frailties and critical issues in neuromuscular diseases highlighted by SARS-CoV-2 pandemic: how many patients are still \u201cinvisible\u201d?","volume":"41","author":"Ricci G","year":"2022","unstructured":"Ricci G, Torri F, Bianchi F, et al. Frailties and critical issues in neuromuscular diseases highlighted by SARS-CoV-2 pandemic: how many patients are still \u201cinvisible\u201d? Acta Myol 2022; 41: 24\u201329.","journal-title":"Acta Myol"},{"key":"e_1_3_3_23_2","doi-asserted-by":"publisher","DOI":"10.2217\/nmt-2023-0008"},{"key":"e_1_3_3_24_2","doi-asserted-by":"publisher","DOI":"10.3389\/fpubh.2023.1247931"},{"key":"e_1_3_3_25_2","doi-asserted-by":"publisher","DOI":"10.3390\/ijerph18062991"},{"key":"e_1_3_3_26_2","first-page":"37","article-title":"Measuring the impact of patient-engaged research: how a methods workshop identified critical outcomes of research engagement","volume":"4","author":"Dillon EC","year":"2017","unstructured":"Dillon EC, Tuzzio L, Madrid S, et al. Measuring the impact of patient-engaged research: how a methods workshop identified critical outcomes of research engagement. J Patient Cent Res Rev 2017; 4: 37.","journal-title":"J Patient Cent Res Rev"},{"key":"e_1_3_3_27_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-017-0608-2"},{"key":"e_1_3_3_28_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-021-00253-6"},{"key":"e_1_3_3_29_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-018-0969-1"},{"key":"e_1_3_3_30_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-021-00276-z"},{"key":"e_1_3_3_31_2","doi-asserted-by":"publisher","DOI":"10.1136\/bmjopen-2023-082502"},{"key":"e_1_3_3_32_2","first-page":"199","volume-title":"Qualitative research practice: a guide for social science students and researchers","author":"Spencer L","year":"2003","unstructured":"Spencer L, Ritchie J, O\u2019Connor W. Analysis: practices, principles and processes. In: Ritchie J, Lewis J (eds) Qualitative research practice: a guide for social science students and researchers. London: Sage, 2003, pp.199\u2013218."},{"key":"e_1_3_3_33_2","unstructured":"Maritime SPOR SUPPORT Unit. Patient-oriented research training online practicum https:\/\/mssu.ca\/get-involved\/attend-training\/ (last accessed 23 February 2025)."},{"key":"e_1_3_3_34_2","volume-title":"Understanding by design","author":"Wiggins G","year":"2005","unstructured":"Wiggins G, McTighe J. Understanding by design. 2nd ed. Alexandria, VA: Association for Supervision and Curriculum Development, 2005.","edition":"2"},{"key":"e_1_3_3_35_2","unstructured":"Mortenson P Kagan C Maltais C et al. imPORTND: patient-oriented research training in neuromuscular diseases https:\/\/importnd.neuromuscularnetwork.ca\/ (2023 last accessed 28 February 2025)."},{"key":"e_1_3_3_36_2","first-page":"547","article-title":"Designing online courses in teacher education to enhance adult learner engagement","volume":"31","author":"Ornelles C","year":"2019","unstructured":"Ornelles C, Ray AB, Wells JC. Designing online courses in teacher education to enhance adult learner engagement. Int J Teach Learn Higher Educ 2019; 31: 547\u2013557.","journal-title":"Int J Teach Learn Higher Educ"},{"key":"e_1_3_3_37_2","doi-asserted-by":"publisher","DOI":"10.2105\/AJPH.89.9.1322"},{"key":"e_1_3_3_38_2","unstructured":"The Neuromuscular Disease Network for Canada. Patient oriented research resources https:\/\/neuromuscularnetwork.ca\/cross-cutting-network-themes\/expert-patient-capacity-building\/patient-oriented-research-resources (2023 last accessed 28 February 2025)."},{"key":"e_1_3_3_39_2","doi-asserted-by":"publisher","DOI":"10.1080\/09687599.2015.1037950"},{"key":"e_1_3_3_40_2","unstructured":"Kirkpatrick A Connor J Cooper M. Web content accessibility guidelines (WCAG) 2.1 https:\/\/www.w3.org\/TR\/WCAG20\/ (2018 accessed 22 February 2023)."},{"key":"e_1_3_3_41_2","doi-asserted-by":"publisher","DOI":"10.1017\/cjn.2023.175"},{"key":"e_1_3_3_42_2","doi-asserted-by":"publisher","DOI":"10.3389\/fresc.2022.878231"},{"key":"e_1_3_3_43_2","doi-asserted-by":"publisher","DOI":"10.1136\/bmjopen-2022-061465"},{"key":"e_1_3_3_44_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-021-00296-9"},{"key":"e_1_3_3_45_2","doi-asserted-by":"publisher","DOI":"10.1186\/s12913-017-2463-1"},{"key":"e_1_3_3_46_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2022.07.394"},{"key":"e_1_3_3_47_2","doi-asserted-by":"publisher","DOI":"10.1212\/NXG.0000000000000607"},{"key":"e_1_3_3_48_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-018-0117-z"},{"key":"e_1_3_3_49_2","doi-asserted-by":"publisher","DOI":"10.1158\/2159-8290.CD-21-0693"},{"key":"e_1_3_3_50_2","doi-asserted-by":"publisher","DOI":"10.1186\/s40900-022-00341-1"},{"key":"e_1_3_3_51_2","doi-asserted-by":"publisher","DOI":"10.1177\/0163278719896392"},{"key":"e_1_3_3_52_2","doi-asserted-by":"publisher","DOI":"10.2217\/cer-2015-0023"}],"container-title":["Journal of Neuromuscular Diseases"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/22143602251405819","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/22143602251405819","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/22143602251405819","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,4]],"date-time":"2026-06-04T04:58:51Z","timestamp":1780549131000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/22143602251405819"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,12,16]]},"references-count":51,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2026,5]]}},"alternative-id":["10.1177\/22143602251405819"],"URL":"https:\/\/doi.org\/10.1177\/22143602251405819","relation":{},"ISSN":["2214-3599","2214-3602"],"issn-type":[{"value":"2214-3599","type":"print"},{"value":"2214-3602","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,12,16]]}}}