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雷特综合征小鼠模型中神经缺陷的逆转

Reversal of neurological defects in a mouse model of Rett syndrome.

作者信息

Guy Jacky, Gan Jian, Selfridge Jim, Cobb Stuart, Bird Adrian

机构信息

Wellcome Trust Centre for Cell Biology, Edinburgh University, King's Buildings, Edinburgh EH9 3JR, UK.

出版信息

Science. 2007 Feb 23;315(5815):1143-7. doi: 10.1126/science.1138389. Epub 2007 Feb 8.

Abstract

Rett syndrome is an autism spectrum disorder caused by mosaic expression of mutant copies of the X-linked MECP2 gene in neurons. However, neurons do not die, which suggests that this is not a neurodegenerative disorder. An important question for future therapeutic approaches to this and related disorders concerns phenotypic reversibility. Can viable but defective neurons be repaired, or is the damage done during development without normal MeCP2 irrevocable? Using a mouse model, we demonstrate robust phenotypic reversal, as activation of MeCP2 expression leads to striking loss of advanced neurological symptoms in both immature and mature adult animals.

摘要

雷特综合征是一种自闭症谱系障碍,由神经元中X连锁的MECP2基因突变拷贝的嵌合表达引起。然而,神经元不会死亡,这表明这不是一种神经退行性疾病。对于这种疾病及相关疾病未来的治疗方法而言,一个重要问题涉及表型的可逆性。有活力但有缺陷的神经元能够修复吗?还是在发育过程中因缺乏正常的MeCP2而造成的损伤是不可逆转的?利用小鼠模型,我们证明了强大的表型逆转,因为MeCP2表达的激活导致未成熟和成熟成年动物的晚期神经症状显著消失。

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Reversal of neurological defects in a mouse model of Rett syndrome.雷特综合征小鼠模型中神经缺陷的逆转
Science. 2007 Feb 23;315(5815):1143-7. doi: 10.1126/science.1138389. Epub 2007 Feb 8.

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