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Mis\u2010splicing of amphiphysin\u20102\/BIN1 is also associated with myotonic dystrophy that shares histopathological hallmarks with CNM. How amphiphysin\u20102 orchestrates nuclear positioning and triad organization and how CNM\u2010associated mutations lead to muscle dysfunction remains elusive. We find that N\u2010WASP interacts with amphiphysin\u20102 in myofibers and that this interaction and N\u2010WASP distribution are disrupted by amphiphysin\u20102 CNM mutations. We establish that N\u2010WASP functions downstream of amphiphysin\u20102 to drive peripheral nuclear positioning and triad organization during myofiber formation. Peripheral nuclear positioning requires microtubule\/Map7\/Kif5b\u2010dependent distribution of nuclei along the myofiber and is driven by actin and nesprins. In adult myofibers, N\u2010WASP and amphiphysin\u20102 are only involved in the maintenance of triad organization but not in the maintenance of peripheral nuclear positioning. Importantly, we confirmed that N\u2010WASP distribution is disrupted in CNM and myotonic dystrophy patients. Our results support a role for N\u2010WASP in amphiphysin\u20102\u2010dependent nuclear positioning and triad organization and in CNM and myotonic dystrophy pathophysiology.<\/jats:p>","DOI":"10.15252\/emmm.201404436","type":"journal-article","created":{"date-parts":[[2014,9,28]],"date-time":"2014-09-28T20:09:29Z","timestamp":1411934969000},"page":"1455-1475","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":128,"title":["N\u2010WASP is required for Amphiphysin\u20102\/BIN1\u2010dependent nuclear positioning and triad organization in skeletal muscle and is involved in the pathophysiology of centronuclear myopathy"],"prefix":"10.1038","volume":"6","author":[{"given":"Sestina","family":"Falcone","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William","family":"Roman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karim","family":"Hnia","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vincent","family":"Gache","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathalie","family":"Didier","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeanne","family":"Lain\u00e9","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frederic","family":"Aurad\u00e9","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabelle","family":"Marty","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ichizo","family":"Nishino","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicolas","family":"Charlet\u2010Berguerand","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Norma Beatriz","family":"Romero","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Giovanna","family":"Marazzi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Sassoon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jocelyn","family":"Laporte","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edgar R","family":"Gomes","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2014,9,29]]},"reference":[{"key":"BFEMMM201404436_CR1","doi-asserted-by":"crossref","first-page":"26","DOI":"10.1186\/2044-5040-1-26","volume":"1","author":"L Al\u2010Qusairi","year":"2011","unstructured":"Al\u2010Qusairi L, Laporte J (2011) T\u2010tubule biogenesis and triad formation in skeletal muscle and implication in human diseases. 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