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Although ANO5 myopathy is not X-chromosome linked, we performed a meta-analysis of the research literature and found that three-quarters of patients with LGMD-R12 are males. Females are less likely to present with moderate to severe skeletal muscle and\/or cardiac pathology. Because these sex differences could be explained in several ways, we compared males and females in a mouse model of LGMD-R12. This model recapitulates the sex differences in human LGMD-R12. Only male Ano5<jats:sup>\u2212\/\u2212<\/jats:sup> mice had elevated serum creatine kinase after exercise and exhibited defective membrane repair after laser injury. In contrast, by these measures, female Ano5<jats:sup>\u2212\/\u2212<\/jats:sup> mice were indistinguishable from wild type. Despite these differences, both male and female Ano5<jats:sup>\u2212\/\u2212<\/jats:sup> mice exhibited exercise intolerance. Although exercise intolerance of male mice can be explained by skeletal muscle dysfunction, echocardiography revealed that Ano5<jats:sup>\u2212\/\u2212<\/jats:sup> female mice had features of cardiomyopathy that may be responsible for their exercise intolerance. 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