Departments of Pediatrics, Neurology and Neuroscience, Northwestern University Feinberg School of Medicine, 303 East Superior Street, Chicago, IL 60611, United States.
Ann & Robert H. Lurie Children's Hospital of Chicago, 225 East Chicago Avenue, Chicago, IL 60611, United States.
Hum Mol Genet. 2024 May 18;33(11):969-980. doi: 10.1093/hmg/ddae029.
RNA methylation of N6-methyladenosine (m6A) is emerging as a fundamental regulator of every aspect of RNA biology. RNA methylation directly impacts protein production to achieve quick modulation of dynamic biological processes. However, whether RNA methylation regulates mitochondrial function is not known, especially in neuronal cells which require a high energy supply and quick reactive responses. Here we show that m6A RNA methylation regulates mitochondrial function through promoting nuclear-encoded mitochondrial complex subunit RNA translation. Conditional genetic knockout of m6A RNA methyltransferase Mettl14 (Methyltransferase like 14) by Nestin-Cre together with metabolomic analysis reveals that Mettl14 knockout-induced m6A depletion significantly downregulates metabolites related to energy metabolism. Furthermore, transcriptome-wide RNA methylation profiling of wild type and Mettl14 knockout mouse brains by m6A-Seq shows enrichment of methylation on mitochondria-related RNA. Importantly, loss of m6A leads to a significant reduction in mitochondrial respiratory capacity and membrane potential. These functional defects are paralleled by the reduced expression of mitochondrial electron transport chain complexes, as well as decreased mitochondrial super-complex assembly and activity. Mechanistically, m6A depletion decreases the translational efficiency of methylated RNA encoding mitochondrial complex subunits through reducing their association with polysomes, while not affecting RNA stability. Together, these findings reveal a novel role for RNA methylation in regulating mitochondrial function. Given that mitochondrial dysfunction and RNA methylation have been increasingly implicate in neurodegenerative disorders, our findings not only provide insights into fundamental mechanisms regulating mitochondrial function, but also open up new avenues for understanding the pathogenesis of neurological diseases.
RNA 上的 N6-甲基腺苷(m6A)甲基化作为 RNA 生物学各个方面的基本调控因子而崭露头角。RNA 甲基化直接影响蛋白质的产生,以实现对动态生物过程的快速调节。然而,RNA 甲基化是否调节线粒体功能尚不清楚,尤其是在神经元细胞中,它们需要高能量供应和快速的反应。在这里,我们表明 m6A RNA 甲基化通过促进核编码线粒体复合物亚基 RNA 翻译来调节线粒体功能。Nestin-Cre 条件性基因敲除 m6A RNA 甲基转移酶 Mettl14(类似甲基转移酶 14)并进行代谢组学分析表明,Mettl14 敲除诱导的 m6A 耗竭显著下调与能量代谢相关的代谢物。此外,通过 m6A-Seq 对野生型和 Mettl14 敲除小鼠大脑进行全转录组 RNA 甲基化谱分析显示,甲基化在与线粒体相关的 RNA 上富集。重要的是,m6A 的缺失导致线粒体呼吸能力和膜电位显著降低。这些功能缺陷与线粒体电子传递链复合物的表达减少以及线粒体超复合物组装和活性降低相平行。在机制上,m6A 耗竭通过减少与多核糖体的结合,降低了编码线粒体复合物亚基的甲基化 RNA 的翻译效率,而不影响 RNA 稳定性。总之,这些发现揭示了 RNA 甲基化在调节线粒体功能中的新作用。鉴于线粒体功能障碍和 RNA 甲基化在神经退行性疾病中越来越受到关注,我们的发现不仅为调节线粒体功能的基本机制提供了深入的见解,而且为理解神经疾病的发病机制开辟了新的途径。