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We investigated the potential involvement of\n                    <jats:italic>VPS13A<\/jats:italic>\n                    deficiency in ferroptosis. The\n                    <jats:italic>VPS13A<\/jats:italic>\n                    gene encodes for chorein, and its deficiency is a molecular cause of chorea\u2010acanthocytosis (ChAc), a Huntington\u2010like disease with neurodegeneration in the striatum. In our previous study, we found male infertility characterized by increased malondialdehyde staining of the spermatozoa in the testes of the ChAc model mice. Thus, in this study we performed metabolome analysis of sperm extracted from the epididymis of the ChAc model mice, which revealed decreased cystine levels, suggesting an association between chorein deficiency and ferroptosis. We then investigated the role of chorein in ferroptosis using\n                    <jats:italic>VPS13A<\/jats:italic>\n                    knockdown (\n                    <jats:italic>VPS13A<\/jats:italic>\n                    \u2010KD) HEK293 cells. We found that\n                    <jats:italic>VPS13A<\/jats:italic>\n                    \u2010KD cells displayed a significantly diminished resistance to tert\u2010Butyl hydroperoxide (tBHP)\u2010induced lipid peroxidation and cell death compared to control cells, which could be rescued by treatment with ferrostatin\u20101. Moreover,\n                    <jats:italic>VPS13A<\/jats:italic>\n                    \u2010KD cells showed Fe(II) accumulation, suggesting an impaired capacity for divalent iron removal. In the cytosolic fraction of\n                    <jats:italic>VPS13A<\/jats:italic>\n                    \u2010KD cells, the protein level of glutathione peroxidase 4 (GPX4) was significantly reduced, suggesting that dysfunction of chorein impairs GPX4 transport, thereby facilitating ferroptosis. These results suggest that ferroptosis may contribute to neurodegeneration in ChAc caused by loss of chorein function.\n                  <\/jats:p>","DOI":"10.1002\/2211-5463.13870","type":"journal-article","created":{"date-parts":[[2024,11,8]],"date-time":"2024-11-08T12:44:13Z","timestamp":1731069853000},"page":"58-68","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Chorein deficiency promotes ferroptosis"],"prefix":"10.1002","volume":"15","author":[{"given":"Yoshiaki","family":"Nishizawa","sequence":"first","affiliation":[{"name":"Department of Psychiatry Kagoshima University Graduate School of Medical and Dental Sciences  Japan"},{"name":"Department of Biochemistry and Genetics Kagoshima University Graduate School of Medical and Dental Sciences  Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hitoshi","family":"Sakimoto","sequence":"additional","affiliation":[{"name":"Department of 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