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小脑浦肯野神经元和颗粒细胞中钠通道Scn8a(NaV1.6)细胞特异性敲除的小鼠运动功能受损。

Impaired motor function in mice with cell-specific knockout of sodium channel Scn8a (NaV1.6) in cerebellar purkinje neurons and granule cells.

作者信息

Levin Stephen I, Khaliq Zayd M, Aman Teresa K, Grieco Tina M, Kearney Jennifer A, Raman Indira M, Meisler Miriam H

机构信息

Dept. of Human Genetics, University of Michigan, School of Medicine, Ann Arbor, MI 48109-0618, USA.

出版信息

J Neurophysiol. 2006 Aug;96(2):785-93. doi: 10.1152/jn.01193.2005. Epub 2006 May 10.

Abstract

The Scn8a gene encodes the voltage-gated Na channel alpha subunit Na(V)1.6, which is widely expressed throughout the nervous system. Global null mutations that eliminate Scn8a in all cells result in severe motor dysfunction and premature death, precluding analysis of the physiological role of Na(V)1.6 in different neuronal types. To test the effect of cerebellar Na(V)1.6 on motor coordination in mice, we used the Cre-lox system to eliminate Scn8a expression exclusively in Purkinje neurons (Purkinje KO) and/or granule neurons (granule KO). Whereas granule KO mice had only minor behavioral defects, adult Purkinje KO mice exhibited ataxia, tremor, and impaired coordination. These disorders were exacerbated in double mutants lacking Scn8a in both Purkinje and granule cells (double KO). In Purkinje cells isolated from adult Purkinje KO and double KO but not granule KO mice, the ratio of resurgent-to-transient tetrodotoxin- (TTX)-sensitive Na current amplitudes decreased from approximately 15 to approximately 5%. In cerebellar slices, Purkinje cell spontaneous and maximal firing rates were reduced 10-fold and twofold relative to control in Purkinje KO and double KO but not granule KO mice. Additionally, short-term plasticity of high-frequency parallel fiber EPSCs was altered relative to control in Purkinje KO and double KO but not granule KO mice. These data suggest that the specialized kinetics of Purkinje Na channels depend directly on Scn8a expression. The loss of these channels leads to a decrease in Purkinje cell firing rates as well as a modification of the synaptic properties of afferent parallel fibers, with the ultimate consequence of disrupting motor behavior.

摘要

Scn8a基因编码电压门控钠通道α亚基Na(V)1.6,其在整个神经系统中广泛表达。在所有细胞中消除Scn8a的全球无效突变会导致严重的运动功能障碍和过早死亡,从而排除了对Na(V)1.6在不同神经元类型中的生理作用进行分析的可能性。为了测试小脑Na(V)1.6对小鼠运动协调的影响,我们使用Cre-lox系统专门消除浦肯野神经元(浦肯野基因敲除)和/或颗粒神经元(颗粒基因敲除)中的Scn8a表达。颗粒基因敲除小鼠只有轻微的行为缺陷,而成人浦肯野基因敲除小鼠表现出共济失调、震颤和协调受损。在浦肯野细胞和颗粒细胞中均缺乏Scn8a的双突变体(双基因敲除)中,这些疾病会加剧。在从成年浦肯野基因敲除和双基因敲除但不是颗粒基因敲除小鼠中分离出的浦肯野细胞中,复苏型与瞬时河豚毒素(TTX)敏感钠电流幅度的比率从约15%降至约5%。在小脑切片中,与对照组相比,浦肯野基因敲除和双基因敲除但不是颗粒基因敲除小鼠的浦肯野细胞自发放电率和最大放电率分别降低了10倍和2倍。此外,相对于对照组,浦肯野基因敲除和双基因敲除但不是颗粒基因敲除小鼠的高频平行纤维兴奋性突触后电流的短期可塑性发生了改变。这些数据表明,浦肯野钠通道的特殊动力学直接取决于Scn8a的表达。这些通道的丧失导致浦肯野细胞放电率降低以及传入平行纤维突触特性的改变,最终导致运动行为受到破坏。

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