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- W2765992815 abstract "Lysosomal storage diseases (LSDs) cause severe cardiomyopathy. However, there is currently no effective treatment. Rag proteins play a critical role in regulating lysosomal functions. Muscle-specific deletion of RagA and RagB (RagA/B) suppresses autophagy and induces cardiomyopathy reminiscent of LSDs. Here, we aimed to investigate the molecular mechanism of cardiomyopathy in RagA/B knockout (KO) mice. We generated cardiac-specific RagA/BKO (RagA/BcKO) mice using αMHC-Cre. Like the muscle-specific KO mice, RagA/BcKO mice showed prominent cardiac hypertrophy (CH) (heart weight (HW)/ tibial length (TL): 12.35 ± 0.41 vs. 7.12 ± 0.15, p< 0.01) at 3 months of age. RagA/BcKO mice exhibited significantly more phospho-histone H3-positive cardiomyocytes (CMs) than control mice, suggesting that CM proliferation is stimulated. However, RagA/BcKO mice exhibited a significantly smaller fractional shortening (%FS: 23.0 ± 0.82 vs. 45.8 ± 1.06, p< 0.01) and significantly higher mortality than control mice (50% vs. 0%, p<..." @default.
- W2765992815 created "2017-11-10" @default.
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- W2765992815 date "2016-11-11" @default.
- W2765992815 modified "2023-09-24" @default.
- W2765992815 title "Abstract 19054: Yap Plays a Crucial Role in the Development of Cardiomyopathy in Lysosomal Storage Diseases" @default.
- W2765992815 hasPublicationYear "2016" @default.
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