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雷特综合征的故事:从临床到神经生物学。

The story of Rett syndrome: from clinic to neurobiology.

作者信息

Chahrour Maria, Zoghbi Huda Y

机构信息

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA.

出版信息

Neuron. 2007 Nov 8;56(3):422-37. doi: 10.1016/j.neuron.2007.10.001.

Abstract

The postnatal neurodevelopmental disorder Rett syndrome (RTT) is caused by mutations in the gene encoding methyl-CpG binding protein 2 (MeCP2), a transcriptional repressor involved in chromatin remodeling and the modulation of RNA splicing. MECP2 aberrations result in a constellation of neuropsychiatric abnormalities, whereby both loss of function and gain in MECP2 dosage lead to similar neurological phenotypes. Recent studies demonstrate disease reversibility in RTT mouse models, suggesting that the neurological defects in MECP2 disorders are not permanent. To investigate the potential for restoring neuronal function in RTT patients, it is essential to identify MeCP2 targets or modifiers of the phenotype that can be therapeutically modulated. Moreover, deciphering the molecular underpinnings of RTT is likely to contribute to the understanding of the pathogenesis of a broader class of neuropsychiatric disorders.

摘要

产后神经发育障碍雷特综合征(RTT)由编码甲基CpG结合蛋白2(MeCP2)的基因突变引起,MeCP2是一种参与染色质重塑和RNA剪接调控的转录抑制因子。MECP2异常导致一系列神经精神异常,功能丧失和MECP2剂量增加均会导致相似的神经表型。最近的研究表明RTT小鼠模型中的疾病具有可逆性,这表明MECP2障碍中的神经缺陷并非永久性的。为了研究恢复RTT患者神经元功能的可能性,识别可进行治疗性调节的MeCP2靶点或表型修饰因子至关重要。此外,破解RTT的分子基础可能有助于理解更广泛类别的神经精神疾病的发病机制。

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