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We generated a knock\u2010in Dnm2R465W mouse model expressing the most frequent human mutation and recently reported that HTZ mice progressively developed a myopathy. We investigated here the cause of neonatal lethality occurring in homozygous (HMZ) mice. We show that HMZ mice present at birth with a reduced body weight, hypoglycemia, increased liver glycogen content and hepatomegaly, in agreement with a defect in neonatal autophagy. <jats:italic>In vitro<\/jats:italic> studies performed in HMZ embryonic fibroblasts point out to a decrease in the autophagy flux prior to degradation at the autolysosome. We show that starved HMZ cells have a higher number of immature autophagy\u2010related structures probably due to a defect of acidification. Our results highlight the role of Dnm2 in the cross talk between endosomal and autophagic pathways and evidence a new role of Dnm2\u2010dependent membrane trafficking in autophagy which may be relevant in DNM2\u2010related human diseases.<\/jats:bold><\/jats:p>","DOI":"10.1111\/j.1600-0854.2012.01348.x","type":"journal-article","created":{"date-parts":[[2012,2,28]],"date-time":"2012-02-28T11:48:24Z","timestamp":1330429704000},"page":"869-879","source":"Crossref","is-referenced-by-count":70,"title":["A Centronuclear Myopathy \u2013 Dynamin 2 Mutation Impairs Autophagy in Mice"],"prefix":"10.1111","volume":"13","author":[{"given":"Anne\u2010C\u00e9cile","family":"Durieux","sequence":"first","affiliation":[{"name":"UM76 Universit\u00e9 Pierre et Marie Curie\u2010Paris 6  Paris F\u201075013 France"},{"name":"U974 Inserm  Paris F\u201075013 France"},{"name":"UMR 7215 CNRS  Paris France"},{"name":"Institut de Myologie  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"St\u00e9phane","family":"Vassilopoulos","sequence":"additional","affiliation":[{"name":"UM76 Universit\u00e9 Pierre et Marie Curie\u2010Paris 6  Paris F\u201075013 France"},{"name":"U974 Inserm  Paris F\u201075013 France"},{"name":"UMR 7215 CNRS  Paris France"},{"name":"Institut de Myologie  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeanne","family":"Lain\u00e9","sequence":"additional","affiliation":[{"name":"UM76 Universit\u00e9 Pierre et Marie Curie\u2010Paris 6  Paris F\u201075013 France"},{"name":"U974 Inserm  Paris F\u201075013 France"},{"name":"UMR 7215 CNRS  Paris France"},{"name":"Institut de Myologie  Paris France"},{"name":"D\u00e9partement de Physiologie, site Piti\u00e9\u2010Salp\u00eatri\u00e8re Universit\u00e9 Pierre et Marie Curie\u2010Paris 6  Paris F\u201075013 France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bodva\u00ebl","family":"Fraysse","sequence":"additional","affiliation":[{"name":"UM76 Universit\u00e9 Pierre et Marie Curie\u2010Paris 6  Paris F\u201075013 France"},{"name":"U974 Inserm  Paris F\u201075013 France"},{"name":"UMR 7215 CNRS  Paris France"},{"name":"Institut de Myologie  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Bri\u00f1as","sequence":"additional","affiliation":[{"name":"UM76 Universit\u00e9 Pierre et Marie Curie\u2010Paris 6  Paris F\u201075013 France"},{"name":"U974 Inserm  Paris F\u201075013 France"},{"name":"UMR 7215 CNRS  Paris France"},{"name":"Institut de Myologie  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernard","family":"Prudhon","sequence":"additional","affiliation":[{"name":"UM76 Universit\u00e9 Pierre et Marie Curie\u2010Paris 6  Paris F\u201075013 France"},{"name":"U974 Inserm  Paris F\u201075013 France"},{"name":"UMR 7215 CNRS  Paris France"},{"name":"Institut de Myologie  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Josiane","family":"Castells","sequence":"additional","affiliation":[{"name":"Laboratoire de Physiologie de l'Exercice  EA4338 Universit\u00e9 Jean Monnet Saint Etienne France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Damien","family":"Freyssenet","sequence":"additional","affiliation":[{"name":"Laboratoire de Physiologie de l'Exercice  EA4338 Universit\u00e9 Jean Monnet Saint Etienne France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gis\u00e8le","family":"Bonne","sequence":"additional","affiliation":[{"name":"UM76 Universit\u00e9 Pierre et Marie Curie\u2010Paris 6  Paris F\u201075013 France"},{"name":"U974 Inserm  Paris F\u201075013 France"},{"name":"UMR 7215 CNRS  Paris France"},{"name":"Institut de Myologie  Paris France"},{"name":"AP\u2010HP, Groupe Hospitalier Piti\u00e9\u2010Salp\u00eatri\u00e8re U.F. 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