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Stargazin等位基因系列中的表型异质性。

Phenotypic heterogeneity in the stargazin allelic series.

作者信息

Letts Verity A, Kang Myoung-Goo, Mahaffey Connie L, Beyer Barbara, Tenbrink Heather, Campbell Kevin P, Frankel Wayne N

机构信息

The Jackson Laboratory, 600 Main Street, Bar Harbor, Maine 04609, USA.

出版信息

Mamm Genome. 2003 Aug;14(8):506-13. doi: 10.1007/s00335-003-2268-x.

DOI:10.1007/s00335-003-2268-x
PMID:12925883
Abstract

The stargazer mutant mouse is characterized by its ataxic gait, head tossing, and absence seizures. The mutation was identified in the gamma 2 subunit gene of the high voltage-dependent calcium channel, Cacng2. Subsequently, two allelic variants of stargazer have arisen, waggler and stargazer 3J. In this study, we have compared these new alleles to the original stargazer allele. All three mutations affect the Cacng2 mRNA levels as they all arise from disruptions within the introns of this gene. Our results show that the mutations cause reduced Cacng2 mRNA and protein levels. Stargazer and waggler mice have the least amount of mRNA and undetectable protein, whereas stargazer 3J appears to be the mildest allele, both in terms of the phenotype and protein expression. Electroencephalographic (EEG) analysis confirmed that stargazer has frequent spike-wave discharges (SWDs); the average duration of each discharge burst is 5 seconds and recurs every minute. The waggler allele causes a greater variation in SWD activity depending on the individual mouse, and the stargazer 3J mouse has no SWDs. The preliminary characterization of this heterogeneous allelic series provides a basis to explore more biochemical and physiological parameters relating to the role of the Cacng2 product in calcium channel activity, AMPA receptor localization, and cerebellar disturbances.

摘要

凝视突变小鼠的特征是共济失调步态、头部摆动和失神发作。该突变是在高电压依赖性钙通道的γ2亚基基因Cacng2中发现的。随后,出现了凝视突变的两个等位基因变体,即摇摆者和凝视3J。在本研究中,我们将这些新等位基因与原始的凝视等位基因进行了比较。所有这三种突变都会影响Cacng2 mRNA水平,因为它们均源于该基因内含子内的破坏。我们的结果表明,这些突变导致Cacng2 mRNA和蛋白质水平降低。凝视和摇摆者小鼠的mRNA量最少且无法检测到蛋白质,而凝视3J在表型和蛋白质表达方面似乎是最温和的等位基因。脑电图(EEG)分析证实,凝视小鼠有频繁的棘波放电(SWD);每次放电爆发的平均持续时间为5秒,每分钟重复出现。摇摆者等位基因根据个体小鼠的不同会导致SWD活动有更大的变化,而凝视3J小鼠没有SWD。这个异质等位基因系列的初步表征为探索更多与Cacng2产物在钙通道活性、AMPA受体定位和小脑紊乱中的作用相关的生化和生理参数提供了基础。

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Cerebellar Golgi, Purkinje, and basket cells have reduced gamma-aminobutyric acid immunoreactivity in stargazer mutant mice.在“凝视者”突变小鼠中,小脑的高尔基细胞、浦肯野细胞和篮状细胞的γ-氨基丁酸免疫反应性降低。
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