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中国大陆6型脊髓小脑共济失调:四个家系的分子和临床特征

Spinocerebellar ataxia type 6 in Mainland China: molecular and clinical features in four families.

作者信息

Jiang Hong, Tang Beisha, Xia Kun, Zhou Yongxin, Xu Bo, Zhao Guohua, Li Haiyan, Shen Lu, Pan Qian, Cai Fang

机构信息

Department of Neurology, Xiangya Hospital, Central South University, Changsha, Hunan 410008, PR China.

出版信息

J Neurol Sci. 2005 Sep 15;236(1-2):25-9. doi: 10.1016/j.jns.2005.04.009.

Abstract

The hereditary spinocerebellar ataxias (SCAs) are a clinically and genetically heterogeneous group of neurodegenerative disorders. The genes causing 11 of these diseases have been identified. To date, there is no report of SCA type 6 (SCA6) in Mainland Chinese. Using a molecular approach, we investigated SCA6 as well as other SCA subtype in 120 Mainland Chinese families with dominantly inherited ataxias and in 60 Mainland Chinese patients with sporadic ataxias. Clinical and molecular features of SCA6 were further characterized in 13 patients from 4 families. We found that SCA3/MJD was the most common type of autosomal dominant SCA in Mainland Chinese, accounting for 83 patients from 59 families (49.2%), followed by SCA2 (8 [6.7%]), SCA1 (7 [5.8%]), SCA6 (4 [3.3%]), SCA7 (1 [0.8%]), SCA8 (0%), SCA10 (0%), SCA12 (1 [0.8%]), SCA14 (0%), SCA17 (0%) and DRPLA (0%). The genes responsible for 40 (33.3%) of dominantly inherited SCA families remain to be determined. Among the 60 patients with sporadic ataxias in the present series, 3 (5.0%) were found to harbor SCA3 mutations, whereas none were found to harbor SCA6 mutations. In the 4 families with SCA6, we found significant anticipation in the absence of genetic instability on transmission. This is the first report of geographic cluster of families with SCA6 subtype in Mainland China.

摘要

遗传性脊髓小脑共济失调(SCAs)是一组临床和遗传异质性的神经退行性疾病。已确定导致其中11种疾病的基因。迄今为止,中国大陆尚无SCA6型(SCA6)的报道。我们采用分子方法,对120个中国大陆显性遗传性共济失调家系和60例中国大陆散发性共济失调患者进行了SCA6以及其他SCA亚型的研究。对来自4个家系的13例患者的SCA6临床和分子特征进行了进一步分析。我们发现,SCA3/MJD是中国大陆最常见的常染色体显性SCA类型,59个家系中的83例患者(占49.2%)属于该类型,其次是SCA2(8例[6.7%])、SCA1(7例[5.8%])、SCA6(4例[3.3%])、SCA7(1例[0.8%])、SCA8(0例)、SCA10(0例)、SCA12(1例[0.8%])、SCA14(0例)、SCA17(0例)和DRPLA(0例)。40个(33.3%)显性遗传性SCA家系的致病基因仍有待确定。在本研究系列的60例散发性共济失调患者中,3例(5.0%)发现携带SCA3突变,而未发现携带SCA6突变。在4个SCA6家系中,我们发现在传递过程中不存在遗传不稳定性的情况下有明显的遗传早现现象。这是中国大陆首次报道SCA6亚型家系的地理聚集情况。

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