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代谢型谷氨酸受体 1,GRM1:作为遗传性小脑共济失调候选基因的评估。

The metabotropic glutamate receptor 1, GRM1: evaluation as a candidate gene for inherited forms of cerebellar ataxia.

机构信息

Laboratory of Molecular Genetics, G. Gaslini Institute, Largo G. Gaslini 5, 16148, Genoa, Italy.

出版信息

J Neurol. 2010 Apr;257(4):598-602. doi: 10.1007/s00415-009-5380-3. Epub 2009 Nov 19.

DOI:10.1007/s00415-009-5380-3
PMID:19924463
Abstract

The metabotropic glutamate (mGlu) 1 receptor, coded by the GRM1 gene, is involved in synaptic activities, learning and neuroprotection. Eleven different mouse Grm1 mutations, either induced or spontaneously occurring, have been reported, including one from our group. All the mutations result in a complex phenotype with ataxia and intention tremor in mice. Moreover, autoantibodies against mGlu1 receptor have been associated with paraneoplastic cerebellar ataxia in humans. In spite of the large clinical and genetic heterogeneity displayed by the inherited forms of cerebellar ataxia, forms remain with a yet unknown molecular definition. With the evidence coming out from mouse models and from paraneoplastic ataxia, it seems that GRM1 represents a good candidate gene for early-onset ataxia forms, though no GRM1 mutations have thus far been looked for. The aim of this study was to investigate the possible involvement of GRM1 in early-onset or familial forms of ataxia. We searched for gene mutations in a panel of patients with early-onset ataxia as yet molecularly undefined. No causative mutations were found, though we detected synonymous variants in the exons and changes in flanking intronic sequences which are unlikely to alter correct splicing upon bioinformatics prediction. As for other known forms of inherited ataxias, absence of mutations in GRM1 seems to suggest a relatively low frequency in cerebellar ataxias.

摘要

代谢型谷氨酸(mGlu)1 受体由 GRM1 基因编码,参与突触活动、学习和神经保护。已经报道了 11 种不同的小鼠 Grm1 突变,包括我们小组的一个突变。所有这些突变导致小鼠出现共济失调和意向性震颤的复杂表型。此外,针对 mGlu1 受体的自身抗体与人类副肿瘤性小脑共济失调有关。尽管遗传性小脑共济失调表现出很大的临床和遗传异质性,但仍有一些形式的分子定义尚不清楚。由于来自小鼠模型和副肿瘤性共济失调的证据,GRM1 似乎是早发性共济失调形式的一个很好的候选基因,尽管迄今为止尚未寻找 GRM1 突变。本研究旨在探讨 GRM1 是否可能参与早发性或家族性共济失调。我们在一组尚未分子定义的早发性共济失调患者中搜索基因突变。未发现致病突变,但我们在exon 中检测到同义变体,侧翼内含子序列中的变化不太可能通过生物信息学预测改变正确的剪接。对于其他已知形式的遗传性共济失调,GRM1 突变缺失似乎表明小脑共济失调的频率相对较低。

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