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Mutations in human lipin 1 are a common cause of recurrent rhabdomyolysis in children. Mice with constitutive whole\u2010body lipin 1 deficiency have been used to examine mechanisms connecting lipin 1 deficiency to myocyte injury. However, that mouse model is confounded by lipodystrophy not phenocopied in people. Herein, 2 muscle\u2010specific mouse models were studied:\n                    <jats:italic>1<\/jats:italic>\n                    )\n                    <jats:italic>Lpin1<\/jats:italic>\n                    exon 3 and 4 deletion, resulting in a hypomorphic protein without PAP activity, but which preserved transcriptional coregulatory function; and\n                    <jats:italic>2<\/jats:italic>\n                    )\n                    <jats:italic>Lpin1<\/jats:italic>\n                    exon 7 deletion, resulting in total protein loss. In both models, skeletal muscles exhibited a chronic myopathy with ongoing muscle fiber necrosis and regeneration and accumulation of phosphatidic acid and, paradoxically, diacylglycerol. Additionally, lipin 1\u2013deficient mice had abundant, but abnormal, mitochondria likely because of impaired autophagy. Finally, these mice exhibited increased plasma creatine kinase following exhaustive exercise when unfed. These data suggest that mice lacking lipin 1\u2013mediated PAP activity in skeletal muscle may serve as a model for determining the mechanisms by which lipin 1 deficiency leads to myocyte injury and for testing potential therapeutic approaches.\u2014Schweitzer, G. G., Collier, S. L., Chen, Z., McCommis, K. S., Pittman, S. K., Yoshino, J., Matkovich, S. J., Hsu, F.\u2010F., Chrast, R., Eaton, J. M., Harris, T. E., Weihl, C. C., Finck, B. N. Loss of lipin 1\u2013mediated phosphatidic acid phosphohydrolase activity in muscle leads to skeletal myopathy in mice. FASEB J. 33, 652\u2013667 (2019).\n                    <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"http:\/\/www.fasebj.org\">www.fasebj.org<\/jats:ext-link>\n                  <\/jats:p>","DOI":"10.1096\/fj.201800361r","type":"journal-article","created":{"date-parts":[[2018,7,20]],"date-time":"2018-07-20T14:48:40Z","timestamp":1532098120000},"page":"652-667","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":41,"title":["Loss of lipin 1\u2010mediated phosphatidic acid phosphohydrolase activity in muscle leads to skeletal myopathy in mice"],"prefix":"10.1096","volume":"33","author":[{"given":"George G.","family":"Schweitzer","sequence":"first","affiliation":[{"name":"Division of Geriatrics and Nutritional Sciences Department of Medicine Washington University School of Medicine  St. Louis Missouri USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara 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