Joshi Alaumy, Stanfield Rachel A, Spletter Andrew T, Gohil Vishal M
Department of Biochemistry and Biophysics, Texas A&M University, 301 Old Main Drive, MS 3474, College Station, TX 77843, USA.
Genetics. 2025 Feb 5;229(2). doi: 10.1093/genetics/iyae203.
Mitochondrial membrane phospholipid cardiolipin is essential for the stability of several inner mitochondrial membrane protein complexes. We recently showed that the abundance of mitochondrial magnesium channel MRS2 is reduced in models of Barth syndrome, an X-linked genetic disorder caused by a remodeling defect in cardiolipin. However, the mechanism underlying the reduced abundance of MRS2 in cardiolipin-depleted mitochondria remained unknown. In this study, we utilized yeast mutants of mitochondrial proteases to identify an evolutionarily conserved m-AAA protease, Yta10/Yta12, responsible for degrading Mrs2. The activity of m-AAA protease is regulated by the inner mitochondrial membrane scaffolding complex prohibitin, and consistent with this role, we find that Mrs2 turnover is increased in yeast prohibitin mutants. Importantly, we find that deleting Yta10 in cardiolipin-deficient yeast cells restores the steady-state levels of Mrs2 to the wild-type cells, and the knockdown of AFG3L2, a mammalian homolog of Yta12, increases the abundance of MRS2 in a murine muscle cell line. Thus, our work has identified the m-AAA protease/prohibitin complex as an evolutionarily conserved regulator of Mrs2 that can be targeted to restore Mrs2 abundance in cardiolipin-depleted cells.
线粒体膜磷脂心磷脂对于几种线粒体内膜蛋白复合物的稳定性至关重要。我们最近发现,在巴氏综合征模型中,线粒体镁通道MRS2的丰度降低,巴氏综合征是一种由心磷脂重塑缺陷引起的X连锁遗传病。然而,心磷脂耗竭的线粒体中MRS2丰度降低的潜在机制仍然未知。在这项研究中,我们利用线粒体蛋白酶的酵母突变体来鉴定一种进化上保守的m-AAA蛋白酶Yta10/Yta12,它负责降解Mrs2。m-AAA蛋白酶的活性受线粒体内膜支架复合物抑制素的调节,与此作用一致,我们发现在酵母抑制素突变体中Mrs2的周转增加。重要的是,我们发现在心磷脂缺陷的酵母细胞中删除Yta10可将Mrs2的稳态水平恢复到野生型细胞,并且Yta12的哺乳动物同源物AFG3L2的敲低会增加小鼠肌肉细胞系中MRS2的丰度。因此,我们的工作已确定m-AAA蛋白酶/抑制素复合物是Mrs2的一种进化上保守的调节因子,可作为靶点来恢复心磷脂耗竭细胞中Mrs2的丰度。