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雷特综合征与 MeCP2。

Rett syndrome and MeCP2.

机构信息

Regenerative Medicine Program, Department of Biochemistry and Medical Genetics, Faculty of Medicine, University of Manitoba, 745 Bannatyne Avenue, Rm. 627, Basic Medical Sciences Bldg., Winnipeg, MB, R3E 0J9, Canada.

出版信息

Neuromolecular Med. 2014 Jun;16(2):231-64. doi: 10.1007/s12017-014-8295-9. Epub 2014 Mar 11.

DOI:10.1007/s12017-014-8295-9
PMID:24615633
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5798978/
Abstract

Rett syndrome (RTT) is a severe and progressive neurological disorder, which mainly affects young females. Mutations of the methyl-CpG binding protein 2 (MECP2) gene are the most prevalent cause of classical RTT cases. MECP2 mutations or altered expression are also associated with a spectrum of neurodevelopmental disorders such as autism spectrum disorders with recent links to fetal alcohol spectrum disorders. Collectively, MeCP2 relation to these neurodevelopmental disorders highlights the importance of understanding the molecular mechanisms by which MeCP2 impacts brain development, mental conditions, and compromised brain function. Since MECP2 mutations were discovered to be the primary cause of RTT, a significant progress has been made in the MeCP2 research, with respect to the expression, function and regulation of MeCP2 in the brain and its contribution in RTT pathogenesis. To date, there have been intensive efforts in designing effective therapeutic strategies for RTT benefiting from mouse models and cells collected from RTT patients. Despite significant progress in MeCP2 research over the last few decades, there is still a knowledge gap between the in vitro and in vivo research findings and translating these findings into effective therapeutic interventions in human RTT patients. In this review, we will provide a synopsis of Rett syndrome as a severe neurological disorder and will discuss the role of MeCP2 in RTT pathophysiology.

摘要

雷特综合征(RTT)是一种严重且进行性的神经疾病,主要影响年轻女性。甲基化CpG 结合蛋白 2(MECP2)基因突变是经典 RTT 病例最常见的原因。MECP2 突变或表达改变也与一系列神经发育障碍有关,如自闭症谱系障碍,最近与胎儿酒精谱系障碍有关。总之,MeCP2 与这些神经发育障碍的关系突出了理解 MeCP2 影响大脑发育、精神状况和大脑功能受损的分子机制的重要性。由于 MECP2 突变被发现是 RTT 的主要原因,因此在 MeCP2 研究方面取得了重大进展,包括在大脑中的表达、功能和调节及其在 RTT 发病机制中的作用。迄今为止,已经从 RTT 患者中收集的小鼠模型和细胞中,设计了针对 RTT 的有效治疗策略,这些策略取得了显著进展。尽管在过去几十年中,MeCP2 研究取得了重大进展,但在体外和体内研究结果之间仍然存在知识差距,并且这些研究结果难以转化为人类 RTT 患者的有效治疗干预措施。在这篇综述中,我们将提供一个关于严重神经疾病雷特综合征的概要,并讨论 MeCP2 在 RTT 病理生理学中的作用。

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本文引用的文献

1
MeCP2-Related Diseases and Animal Models.与MeCP2相关的疾病及动物模型
Diseases. 2014 Jan 27;2(1):45-70. doi: 10.3390/diseases2010045.
2
Brain region-specific expression of MeCP2 isoforms correlates with DNA methylation within Mecp2 regulatory elements.MeCP2 亚型在脑区的特异性表达与 Mecp2 调控元件内的 DNA 甲基化相关。
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Mice with an isoform-ablating Mecp2 exon 1 mutation recapitulate the neurologic deficits of Rett syndrome.携带Mecp2外显子1突变(该突变可消除异构体)的小鼠重现了雷特综合征的神经功能缺损症状。
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MeCP2 R168X male and female mutant mice exhibit Rett-like behavioral deficits.男性和女性 MeCP2 R168X 突变小鼠表现出类似雷特氏症的行为缺陷。
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Decitabine alters the expression of Mecp2 isoforms via dynamic DNA methylation at the Mecp2 regulatory elements in neural stem cells.地西他滨通过在神经干细胞中的 Mecp2 调节元件上的动态 DNA 甲基化改变 Mecp2 异构体的表达。
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SUMOylation of MeCP2 is essential for transcriptional repression and hippocampal synapse development.MeCP2 的 SUMOylation 对于转录抑制和海马突触发育是必不可少的。
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Global transcriptional and translational repression in human-embryonic-stem-cell-derived Rett syndrome neurons.人胚胎干细胞衍生的瑞特综合征神经元中的全局转录和翻译抑制
Cell Stem Cell. 2013 Oct 3;13(4):446-58. doi: 10.1016/j.stem.2013.09.001.
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Identification of TET1 Partners That Control Its DNA-Demethylating Function.鉴定调控TET1 DNA去甲基化功能的相互作用蛋白
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Role of conserved cis-regulatory elements in the post-transcriptional regulation of the human MECP2 gene involved in autism.在涉及自闭症的人类 MECP2 基因的转录后调控中,保守顺式调控元件的作用。
Hum Genomics. 2013 Sep 16;7(1):19. doi: 10.1186/1479-7364-7-19.