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[中国患者Leber遗传性视神经病变的临床特征及突变情况]

[Clinical features and the mutation of Leber's hereditary optic neuropathy in Chinese patients].

作者信息

Wang Yan, Guo Xiang-ming, Jia Xiao-yun, Li Shi-qiang, Xiao Xue-shan, Guo Li, Zhang Qing-jiong

机构信息

Department of Ocular Genetics and Molecular Biology, Zhongshan Ophthalmic Center, Sun Yat-sen University, Guangzhou, Guangdong, 510060, PR China.

出版信息

Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2005 Jun;22(3):334-6.

Abstract

OBJECTIVE

To analyze the mutation of Leber's hereditary optic neuropathy (LHON) and the clinical features in Chinese patients.

METHODS

The primary mtDNA mutations (3460A, 11778A and 14484C) of 156 patients (110 probands and 46 maternal relatives with LHON) were detected by mutation-specific priming polymerase chain reaction, heteroduplex-single strand conformation polymorphism polymerase chain reaction, restriction fragment length polymorphisms and measurement of DNA sequence. The clinical features were analyzed by retrospective study.

RESULTS

The 11778A mutation was found in 100 probands (90.9%), the 3460A mutation was found in 2 (1.8%), and the 14484C was found in 8 (7.3%) of the 110 probands. The visual acuity at onset of the disease was 0.01 or worse in 44 (17.6%) of 250 eyes with the 11778A mutation, but in none of 79 eyes with the 14484C mutation. The visual acuity was 0.1 or better in 76 (29.6%) of 250 eyes with the 11778A mutation, but in 49 (87.3%) of 56 eyes with the 14484C mutation. And 6.8% of 250 eyes with the 11778A mutation recovered a mean final visual acuity of 0.03, whereas 50% of 56 eyes with the 14484C mutation recovered a mean final visual acuity of 0.8.

CONCLUSION

In Chinese LHON patients the 11778A, 14484C primary mutations are common. The clinical features are associated with the site of primary mutation. The visual acuity at onset of the disease and the visual recovery of the eyes with 14484C mutation were better than the eyes with the 11778A mutation.

摘要

目的

分析中国患者中Leber遗传性视神经病变(LHON)的突变情况及临床特征。

方法

采用突变特异性引物聚合酶链反应、异源双链 - 单链构象多态性聚合酶链反应、限制性片段长度多态性及DNA序列测定等方法,检测156例患者(110例先证者及46例LHON患者的母系亲属)的主要线粒体DNA突变(3460A、11778A和14484C)。通过回顾性研究分析临床特征。

结果

在110例先证者中,100例(90.9%)发现11778A突变,2例(1.8%)发现3460A突变,8例(7.3%)发现14484C突变。在携带11778A突变的250只眼中,44只(17.6%)发病时视力为0.01或更差,但携带14484C突变的79只眼中无一例如此。在携带11778A突变 的250只眼中,76只(29.6%)视力为0.1或更好,而在携带14484C突变的56只眼中,49只(87.3%)视力为0.1或更好。携带11778A突变的250只眼中有6.8%最终平均视力恢复到0.03,而携带14484C突变的56只眼中有50%最终平均视力恢复到0.8。

结论

在中国LHON患者中,11778A、14484C主要突变较为常见。临床特征与主要突变位点相关。携带14484C突变的眼睛发病时的视力及视力恢复情况优于携带11778A突变的眼睛。

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