Motor deficits and hyperactivity in Dyt1 knockdown mice

被引：70

作者：

Dang, Mai T. ^{[1
]}

Yokoi, Fumiaki ^{[1
]}

Pence, Morgan A. ^{[1
]}

Li, Yuqing ^{[1
]}

机构：

[1] Univ Illinois, Dept Mol & Integrat Physiol, NeuroTech Grp, Beckman Inst Adv Sci & Technol, Urbana, IL 61801 USA

来源：

NEUROSCIENCE RESEARCH | 2006年 / 56卷 / 04期

关键词：

Dyt1; TorsinA; dystonia; knockdown; Tor1A; Dyt1 Delta GAG;

D O I：

10.1016/j.neures.2006.09.005

中图分类号：

Q189 [神经科学];

学科分类号：

071006 ;

摘要：

The DYT1 gene containing a trinucleotide deletion (Delta GAG) is linked to early-onset dystonia, a neurological movement disorder of involuntary muscle contractions. To understand DYT1's contribution to dystonia, we produced and analyzed Dyt1 knockdown (KD) mice that expressed a reduced level of torsinA protein encoded by Dyt1. Knockdown mice exhibited deficits in motor control and a decreased trend in dopamine with a significant reduction in 3,4-dihydroxyphenylacetic acid. These alterations are similar to those displayed by previously reported Dyt1 Delta GAG knockin heterozygous mice, suggesting that the partial loss of torsinA function contributes to the pathology of the disease. (c) 2006 Elsevier Ireland Ltd and the Japan Neuroscience Society. All rights reserved.

引用

收藏

页码：470 / 474

页数：5

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