biostudies-literatureUnknown14(5)Yuan W<b>Aim:</b> Adipose tissue (AT) dysfunction that occurs in both obesity and lipodystrophy is associated with the development of cardiomyopathy. However, it is unclear how dysfunctional AT induces cardiomyopathy due to limited animal models available. We have identified vacuolar H⁺-ATPase subunit V_od1, encoded by <i>Atp6v0d1</i>, as a master regulator of adipogenesis, and adipose-specific deletion of <i>Atp6v0d1</i> (<i>Atp6v0d1</i>^AKO) in mice caused generalized lipodystrophy and spontaneous cardiomyopathy. Using this unique animal model, we explore the mechanism(s) underlying lipodystrophy-related cardiomyopathy. <b>Methods and Results:</b> <i>Atp6v0d1</i>^AKO mice developed cardiac hypertrophy at 12 weeks, and progressed to heart failure at 28 wTheranostics2246-2264https://www.ebi.ac.uk/biostudies/studies/S-EPMC10945344biostudies-literatureMyocardin reverses insulin resistance and ameliorates cardiomyopathy by increasing IRS-1 expression in a murine model of lipodystrophy caused by adipose deficiency of vacuolar H&lt;sup&gt;+&lt;/sup&gt;-ATPase V0d1 subunit.PMC10945344Yan MLiu JPan YLiu LLu XOuyang KHe QSun YLi YHuang YZhang XLin HZhang CYuan WSong YXu YfalseMyocardin reverses insulin resistance and ameliorates cardiomyopathy by increasing IRS-1 expression in a murine model of lipodystrophy caused by adipose deficiency of vacuolar H&lt;sup&gt;+&lt;/sup&gt;-ATPase V0d1 subunit.<b>Aim:</b> Adipose tissue (AT) dysfunction that occurs in both obesity and lipodystrophy is associated with the development of cardiomyopathy. However, it is unclear how dysfunctional AT induces cardiomyopathy due to limited animal models available. We have identified vacuolar H⁺-ATPase subunit V_od1, encoded by <i>Atp6v0d1</i>, as a master regulator of adipogenesis, and adipose-specific deletion of <i>Atp6v0d1</i> (<i>Atp6v0d1</i>^AKO) in mice caused generalized lipodystrophy and spontaneous cardiomyopathy. Using this unique animal model, we explore the mechanism(s) underlying lipodystrophy-related cardiomyopathy. <b>Methods and Results:</b> <i>Atp6v0d1</i>^AKO mice developed cardiac hypertrophy at 12 weeks, and progressed to heart failure at 28 w2024-01-01T00:00:00Z20242026-07-15T02:25:25.771Z2025-04-06T13:54:08.216ZS-EPMC109453443850562010.7150/thno.93192