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ML-SA5 is a potent TRPML cation channel agonist that activates whole-endolysosomal TRPML1 (ML1) currents in DMD myoblasts at a higher potency than ML-SA1 (EC50 = 285 nM vs 10 μM). ML-SA5 ameliorates muscular dystrophy in mdx, but not ML1-knockout, mice in vivo (2 or 5 mg/kg/day i.p.) by facilitating sarcolemma repair, thereby reducing skeletal and cardiac muscle damage with no apparent toxicity to the animals.
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