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雷特综合征小鼠的脑脂质分析

Brain lipid analysis in mice with Rett syndrome.

作者信息

Seyfried Thomas N, Heinecke Karie A, Mantis John G, Denny Christine A

机构信息

Department of Biology, Boston College, Boston, MA 02467, USA.

出版信息

Neurochem Res. 2009 Jun;34(6):1057-65. doi: 10.1007/s11064-008-9874-7. Epub 2008 Nov 11.

Abstract

Rett syndrome (RS) is an X-linked neurodevelopmental disorder mostly involving mutations in the gene for methyl-CpG-binding protein 2 (MECP2). Ganglioside abnormalities were previously found in cerebrum and cerebellum in RS patients. We evaluated total lipid distribution in cerebrum/brainstem, hippocampus, and cerebellum in male mice carrying either the Mecp2 (tm1.1Bird) knockout mutation or the Mecp2 (308/y) deletion mutation. The concentration of the neuronal enriched ganglioside GD1a was significantly lower in the cerebrum/brainstem of Mecp2 (tm1.1Bird) mice than in that of age matched controls, but was not reduced in the Mecp2 (308/y) mice. No other differences in brain lipid content, including myelin-enriched cerebrosides, were detected in mice with either type of Mecp2 mutation. These findings indicate that the poor motor performance previously reported in the RS mutant mice is not associated with major brain lipid abnormalities and that most previous brain lipid abnormalities observed in RS patients were not observed in the Mecp2 (tm1.1Bird) or the Mecp2 (308/y) RS mice.

摘要

雷特综合征(RS)是一种X连锁神经发育障碍,主要涉及甲基CpG结合蛋白2(MECP2)基因的突变。先前在RS患者的大脑和小脑中发现了神经节苷脂异常。我们评估了携带Mecp2(tm1.1Bird)基因敲除突变或Mecp2(308/y)缺失突变的雄性小鼠大脑/脑干、海马体和小脑中的总脂质分布。在Mecp2(tm1.1Bird)小鼠的大脑/脑干中,富含神经元的神经节苷脂GD1a的浓度显著低于年龄匹配的对照组,但在Mecp2(308/y)小鼠中并未降低。在两种类型的Mecp2突变小鼠中,均未检测到脑脂质含量的其他差异,包括富含髓磷脂的脑苷脂。这些发现表明,先前报道的RS突变小鼠运动能力差与主要的脑脂质异常无关,并且在Mecp2(tm1.1Bird)或Mecp2(308/y)RS小鼠中未观察到先前在RS患者中观察到的大多数脑脂质异常。

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