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Mecp2基因敲除小鼠桶状皮层的结构和功能差异

Structural and functional differences in the barrel cortex of Mecp2 null mice.

作者信息

Lee Li-Jen, Tsytsarev Vassiliy, Erzurumlu Reha S

机构信息

Graduate Institute of Anatomy and Cell Biology, National Taiwan University, Taipei, Taiwan, ROC.

Department of Anatomy and Neurobiology, University of Maryland School of Medicine, Baltimore, Maryland.

出版信息

J Comp Neurol. 2017 Dec 15;525(18):3951-3961. doi: 10.1002/cne.24315. Epub 2017 Sep 15.

Abstract

Functional deficits in sensory systems are commonly noted in neurodevelopmental disorders, such as the Rett syndrome (RTT). Defects in methyl CpG binding protein gene (MECP2) largely accounts for RTT. Manipulations of the Mecp2 gene in mice provide useful models to probe into various aspects of brain development associated with the RTT. In this study, we focused on the somatosensory cortical phenotype in the Bird mouse model of RTT. We used voltage-sensitive dye imaging to evaluate whisker sensory evoked activity in the barrel cortex of mice. We coupled this functional assay with morphological analyses in postnatal mice and investigated the dendritic differentiation of barrel neurons and individual thalamocortical axon (TCA) arbors that synapse with them. We show that in Mecp2-deficient male mice, whisker-evoked activity is roughly topographic but weak in the barrel cortex. At the morphological level, we find that TCA arbors fail to develop into discrete, concentrated patches in barrel hollows, and the complexity of the dendritic branches in layer IV spiny stellate neurons is reduced. Collectively, our results indicate significant structural and functional impairments in the barrel cortex of the Bird mouse line, a popular animal model for the RTT. Such structural and functional anomalies in the primary somatosensory cortex may underlie orofacial tactile sensitivity issues and sensorimotor stereotypies characteristic of RTT.

摘要

感觉系统的功能缺陷在神经发育障碍中很常见,如雷特综合征(RTT)。甲基CpG结合蛋白基因(MECP2)的缺陷在很大程度上导致了RTT。对小鼠Mecp2基因的操作提供了有用的模型,以探究与RTT相关的大脑发育的各个方面。在本研究中,我们聚焦于RTT的Bird小鼠模型中的躯体感觉皮层表型。我们使用电压敏感染料成像来评估小鼠桶状皮层中的触须感觉诱发活动。我们将这种功能测定与出生后小鼠的形态学分析相结合,并研究了桶状神经元和与其形成突触的单个丘脑皮质轴突(TCA)分支的树突分化。我们发现,在缺乏Mecp2的雄性小鼠中,触须诱发的活动在桶状皮层中大致呈拓扑分布,但较弱。在形态学水平上,我们发现TCA分支未能在桶状凹陷中发育成离散、集中的斑块,并且IV层棘状星形神经元的树突分支复杂性降低。总的来说,我们的结果表明,Bird小鼠品系(一种常用的RTT动物模型)的桶状皮层存在明显的结构和功能损伤。初级躯体感觉皮层中的这种结构和功能异常可能是RTT特征性的口面部触觉敏感性问题和感觉运动刻板行为的基础。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aaa0/5994756/e4ff1adc1a1d/nihms971976f1.jpg

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