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内侧颞叶癫痫的遗传易感性与线粒体功能障碍研究

The Study of Genetic Susceptibility and Mitochondrial Dysfunction in Mesial Temporal Lobe Epilepsy.

作者信息

Yang Haiyan, Yin Fei, Gan Siyi, Pan Zou, Xiao Ting, Kessi Miriam, Yang Zhuangyi, Zhang Victor Wei, Wu Liwen

机构信息

Department of Pediatrics, Xiangya Hospital, Central South University, Changsha, 410008, Hunan, People's Republic of China.

Hunan Intellectual and Developmental Disabilities Research Center of Children, Changsha, 410008, Hunan, China.

出版信息

Mol Neurobiol. 2020 Sep;57(9):3920-3930. doi: 10.1007/s12035-020-01993-4. Epub 2020 Jul 6.

Abstract

The aim of this study is to investigate the mitochondrial dysfunction and pathogenic role of the mitochondrial genome in the progression of mesial temporal lobe epilepsy (MTLE) in vivo and in vitro. Mitochondrial DNA (mtDNA) and nuclear DNA were detected in the hippocampal samples and peripheral blood of patients with MTLE. Mitochondrial functions were detected in vivo and in vitro. In 20 patients with MTLE, mtDNA mutations involving single or multiple deletions in the hippocampus were found in 5 patients but were not detected in the peripheral blood. Two patients carried pathogenic mutations of RELN, both in the hippocampus and blood. A pathogenic mutation of DNA2 was found in the hippocampus of the 2 patients with multiple deletions but not in the blood samples. The mtDNA copy numbers showed dynamic changes in the MTLE models. In MTLE patients, low metabolism in mesial temporal lobe and hippocampus was observed by using PET-CT. Under electron microscope, the mitochondrial cristae were disordered, the density of mitochondrial matrix decreased and even vacuolated in the hippocampus neurons. In the MTLE rat models, there were dynamic changes in mitochondrial morphology; the ATP production rate decreased in the acute phase, the latent phase, and the chronic phase. Mitochondrial enzyme complex I activity decreased in both acute and chronic phases, and there was no significant difference in latent period. Decreased mitochondrial membrane potential and calcium homeostasis were detected in the epileptic cell models. We first identified somatic mutations in mtDNA in MTLE patients and comprehensively evaluated mitochondrial dysfunction in the pathogenesis of MTLE in vivo and in vitro. This evidence supports the environmental and modifying genetic interactions that contribute to the development of MTLE.

摘要

本研究的目的是在体内和体外研究线粒体功能障碍以及线粒体基因组在颞叶内侧癫痫(MTLE)进展中的致病作用。在MTLE患者的海马样本和外周血中检测线粒体DNA(mtDNA)和核DNA。在体内和体外检测线粒体功能。在20例MTLE患者中,5例患者的海马中发现了涉及单个或多个缺失的mtDNA突变,但在外周血中未检测到。2例患者在海马和血液中均携带RELN的致病突变。在2例有多个缺失的患者的海马中发现了DNA2的致病突变,但在血液样本中未发现。mtDNA拷贝数在MTLE模型中呈现动态变化。在MTLE患者中,使用PET-CT观察到颞叶内侧和海马的低代谢。在电子显微镜下,海马神经元中线粒体嵴紊乱,线粒体基质密度降低甚至出现空泡化。在MTLE大鼠模型中,线粒体形态有动态变化;在急性期、潜伏期和慢性期ATP产生率均降低。线粒体酶复合物I活性在急性期和慢性期均降低,潜伏期无显著差异。在癫痫细胞模型中检测到线粒体膜电位降低和钙稳态失衡。我们首次在MTLE患者中鉴定出mtDNA的体细胞突变,并在体内和体外全面评估了线粒体功能障碍在MTLE发病机制中的作用。这一证据支持了环境和修饰基因相互作用对MTLE发展的影响。

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