{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,4]],"date-time":"2026-10-04T15:26:08Z","timestamp":1791127568991,"version":"4.1.0"},"reference-count":34,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2014,1,17]],"date-time":"2014-01-17T00:00:00Z","timestamp":1389916800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"National Health and Medical Research Council Australia","award":["#566818"],"award-info":[{"award-number":["#566818"]}]},{"name":"Association Fran\u00e7aise contre les Myopathies (AFM, France)"},{"DOI":"10.13039\/501100001711","name":"Swiss National Science Foundation","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100001711","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Experimental Physiology"],"published-print":{"date-parts":[[2014,4]]},"abstract":"<jats:title>New Findings<\/jats:title><jats:sec><jats:label\/><jats:p><jats:list list-type=\"bullet\">\n<jats:list-item><jats:p><jats:bold>What is the central question of this study?<\/jats:bold><\/jats:p>\n<jats:p>The Notch signalling pathway plays an important role in muscle regeneration, and activation of the pathway has been shown to enhance muscle regeneration in aged mice. It is unknown whether Notch activation will have a similarly beneficial effect on muscle regeneration in the context of Duchenne muscular dystrophy (DMD).<\/jats:p><\/jats:list-item>\n<jats:list-item><jats:p><jats:bold>What is the main finding and its importance?<\/jats:bold><\/jats:p>\n<jats:p>Although expression of Notch signalling components is altered in both mouse models of DMD and in human DMD patients, activation of the Notch signalling pathway does not confer any functional benefit on muscles from dystrophic mice, suggesting that other signalling pathways may be more fruitful targets for manipulation in treating DMD.<\/jats:p><\/jats:list-item>\n<\/jats:list><\/jats:p><\/jats:sec><jats:sec><jats:title>Abstract<\/jats:title><jats:p>In Duchenne muscular dystrophy (DMD), muscle damage and impaired regeneration lead to progressive muscle wasting, weakness and premature death. The Notch signalling pathway represents a central regulator of gene expression and is critical for cellular proliferation, differentiation and apoptotic signalling during all stages of embryonic muscle development. Notch activation improves muscle regeneration in aged mice, but its potential to restore regeneration and function in muscular dystrophy is unknown. We performed a comprehensive examination of several genes involved in Notch signalling in muscles from dystrophin\u2010deficient <jats:italic>mdx<\/jats:italic> and <jats:italic>dko<\/jats:italic> (utrophin\u2010 and dystrophin\u2010null) mice and DMD patients. A reduction of <jats:italic>Notch1<\/jats:italic> and <jats:italic>Hes1<\/jats:italic> mRNA in tibialis anterior muscles of <jats:italic>dko<\/jats:italic> mice and quadriceps muscles of DMD patients and a reduction of <jats:italic>Hes1<\/jats:italic> mRNA in the diaphragm of the <jats:italic>mdx<\/jats:italic> mice were observed, with other targets being inconsistent across species. Activation and inhibition of Notch signalling, followed by measures of muscle regeneration and function, were performed in the mouse models of DMD. Notch activation had no effect on functional regeneration in C57BL\/10, <jats:italic>mdx<\/jats:italic> or <jats:italic>dko<\/jats:italic> mice. Notch inhibition significantly depressed the frequency\u2013force relationship in regenerating muscles of C57BL\/10 and <jats:italic>mdx<\/jats:italic> mice after injury, indicating reduced force at each stimulation frequency, but enhanced the frequency\u2013force relationship in muscles from <jats:italic>dko<\/jats:italic> mice. We conclude that while Notch inhibition produces slight functional defects in dystrophic muscle, Notch activation does not significantly improve muscle regeneration in murine models of muscular dystrophy. Furthermore, the inconsistent expression of Notch targets between murine models and DMD patients suggests caution when making interspecies comparisons.<\/jats:p><\/jats:sec>","DOI":"10.1113\/expphysiol.2013.077255","type":"journal-article","created":{"date-parts":[[2014,1,18]],"date-time":"2014-01-18T01:16:29Z","timestamp":1390007789000},"page":"675-687","source":"Crossref","is-referenced-by-count":26,"title":["Alterations in Notch signalling in skeletal muscles from <i>mdx<\/i> and <i>dko<\/i> dystrophic mice and patients with Duchenne muscular dystrophy"],"prefix":"10.1113","volume":"99","author":[{"given":"Jarrod E.","family":"Church","sequence":"first","affiliation":[{"name":"Basic and Clinical Myology Laboratory Department of Physiology The University of Melbourne  Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer","family":"Trieu","sequence":"additional","affiliation":[{"name":"Basic and Clinical Myology Laboratory Department of Physiology The University of Melbourne  Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annabel","family":"Chee","sequence":"additional","affiliation":[{"name":"Basic and Clinical Myology Laboratory Department of Physiology The University of Melbourne  Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Timur","family":"Naim","sequence":"additional","affiliation":[{"name":"Basic and Clinical Myology Laboratory Department of Physiology The University of Melbourne  Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stefan M.","family":"Gehrig","sequence":"additional","affiliation":[{"name":"Basic and Clinical Myology Laboratory Department of Physiology The University of Melbourne  Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S\u00e9verine","family":"Lamon","sequence":"additional","affiliation":[{"name":"Centre for Physical Activity and Nutrition Research School of Exercise and Nutrition Sciences Deakin University  Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Corrado","family":"Angelini","sequence":"additional","affiliation":[{"name":"Neurosciences Department IRCCS San Camillo Hospital  Lido Venice Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aaron P.","family":"Russell","sequence":"additional","affiliation":[{"name":"Centre for Physical Activity and Nutrition Research School of Exercise and Nutrition Sciences Deakin University  Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gordon S.","family":"Lynch","sequence":"additional","affiliation":[{"name":"Basic and Clinical Myology Laboratory Department of Physiology The University of Melbourne  Victoria 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