{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,15]],"date-time":"2026-10-15T20:46:51Z","timestamp":1792097211530,"version":"4.4.0"},"reference-count":46,"publisher":"American Society for Clinical Investigation","issue":"9","funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01AR068286"],"award-info":[{"award-number":["R01AR068286"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01AG059605"],"award-info":[{"award-number":["R01AG059605"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000875","name":"Pew Charitable Trusts","doi-asserted-by":"crossref","award":["2015"],"award-info":[{"award-number":["2015"]}],"id":[{"id":"10.13039\/100000875","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,5,7]]},"abstract":"<jats:p>\n                    Muscle progenitor cell fusion is required for the formation and regeneration of multinucleated skeletal muscle fibers. Chronic muscle regeneration in Duchenne muscular dystrophy (DMD) is characterized by ongoing fusion of satellite cell (SC) progeny, but the effects of fusion on disease and the mechanisms by which fusion is accomplished in this setting are not fully understood. Using the\n                    <jats:italic>mdx<\/jats:italic>\n                    mouse model of DMD, we deleted the fusogenic protein Myomaker in SCs or myofibers. Following deletion in SCs, mice displayed a complete lack of myocyte fusion, resulting in severe muscle loss, enhanced fibrosis, and significant functional decline. Reduction of Myomaker in mature myofibers in\n                    <jats:italic>mdx<\/jats:italic>\n                    mice, however, led to minimal alterations in fusion dynamics. Unexpectedly, myofiber-specific deletion of Myomaker resulted in improvement of disease phenotype, with enhanced function and decreased muscle damage. Our data indicate that Myomaker has divergent effects on dystrophic disease severity depending upon its compartment of expression. These findings show that myocyte fusion is absolutely required for effective regeneration in DMD, but persistent Myomaker expression in myofibers due to ongoing fusion may have unintended deleterious consequences for muscle integrity. Thus, sustained activation of a component of the myogenic program in dystrophic myofibers exacerbates disease.\n                  <\/jats:p>","DOI":"10.1172\/jci.insight.136095","type":"journal-article","created":{"date-parts":[[2020,4,20]],"date-time":"2020-04-20T16:33:09Z","timestamp":1587400389000},"source":"Crossref","is-referenced-by-count":40,"title":["Myocyte-derived Myomaker expression is required for regenerative fusion but exacerbates membrane instability in dystrophic myofibers"],"prefix":"10.1172","volume":"5","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8009-3224","authenticated-orcid":true,"given":"Michael J.","family":"Petrany","sequence":"first","affiliation":[{"name":"Division of Molecular Cardiovascular Biology, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, Ohio, 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