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Multiple disease modifying therapies are in clinical trials for DM1 and show promise in improving skeletal muscle weakness and myotonia. Testing the effects of these medicines, or others, in the heart is of critical importance, but requires identification of endpoints of cardiac function that accurately reflect the state of DM1 cardiac disease. To better define cardiac endpoints, the Myotonic Dystrophy Clinical Research Network (DMCRN) and the Myotonic Dystrophy Foundation (MDF) convened a workshop entitled \u2018\u2018Cardiac Endpoint Workshop\u2019\u2019 in May 2025 at the Myotonic Dystrophy Foundation International Conference. Here, we summarize the discussion at the workshop and perform secondary analysis of cardiac outcomes in the published literature to evaluate cardiac endpoints for clinical impact and trial feasibility. This analysis demonstrates that major cardiac events are too infrequent (&lt;1% annual incidence), and alternatives such as composite endpoints or progression of cardiac conduction prolongation would likely be underpowered in a conventional clinical trial. Given these limitations, we identify areas for further natural history study to better describe longitudinal cardiac structural and functional changes to inform specialized patient selection or identify alternative measures with sensitivity to detect therapeutic impact in a trial.<\/jats:p>","DOI":"10.1177\/22143602261433484","type":"journal-article","created":{"date-parts":[[2026,4,7]],"date-time":"2026-04-07T10:10:14Z","timestamp":1775556614000},"page":"1567-1579","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":1,"title":["Developing endpoints for the cardiac burden in myotonic dystrophy type 1: A workshop report"],"prefix":"10.1177","volume":"13","author":[{"given":"Julia M","family":"Hartman","sequence":"first","affiliation":[{"name":"Virginia Commonwealth University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0114-495X","authenticated-orcid":false,"given":"Samuel","family":"Carrell","sequence":"additional","affiliation":[{"name":"Virginia Commonwealth University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William J","family":"Groh","sequence":"additional","affiliation":[{"name":"Ralph H. Johnson VA Medical Center and Medical University of South Carolina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas A","family":"Cooper","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3397-0535","authenticated-orcid":false,"given":"Jordana","family":"Kron","sequence":"additional","affiliation":[{"name":"Virginia Commonwealth University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Greg","family":"Hundley","sequence":"additional","affiliation":[{"name":"Virginia Commonwealth University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1001-1849","authenticated-orcid":false,"given":"Jennifer","family":"Jordan","sequence":"additional","affiliation":[{"name":"Virginia Commonwealth University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amy","family":"Ladd","sequence":"additional","affiliation":[{"name":"Virginia Commonwealth University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Man","family":"Hung","sequence":"additional","affiliation":[{"name":"Roseman University of Health Sciences"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicholas E","family":"Johnson","sequence":"additional","affiliation":[{"name":"Virginia Commonwealth University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"on behalf of the Myotonic Dystrophy Clinical Research Network (DMCRN) and the Myotonic Dystrophy Foundation (MDF)","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2026,4,7]]},"reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000011425"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0008-6363(96)00163-0"},{"key":"e_1_3_3_4_2","volume-title":"Myotonic Dystrophy","author":"Harper PS","year":"2001","unstructured":"Harper PS. 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Usefulness of clinical and electrocardiographic data for predicting adverse cardiac events in patients with myotonic dystrophy. 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