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[一个具有类似肥厚型心肌病临床特征的法布里病中国家系中的α-半乳糖苷酶A基因突变]

[Alpha-galactosidase A gene mutation in a Chinese family with Fabry disease mimicking clinical features of hypertrophic cardiomyopathy].

作者信息

Liu He-jun, Cao Ke-jiang, Li Cheng-rang, Dai Jian, Ma Ji-zheng, Yong Yong-hong, Sun Wei

机构信息

Department of Cardiology, First Affiliated Hospital of Nanjing Medical University, Nanjing 210029, China.

出版信息

Zhonghua Xin Xue Guan Bing Za Zhi. 2006 Feb;34(2):143-7.

Abstract

OBJECTIVE

To screen gene mutation in alpha-galactosidase A (alpha-Gal A) in a nonconsanguineous Chinese family with Fabry disease (FD) with clinical manifestations similar to hypertrophic cardiomyopathy (HCM).

METHODS

Mutation analysis was performed by using purified PCR products to direct sequence analysis on an ABI-377XL automated DNA sequencer. DNA analysis of alpha-Gal A gene and physical and clinical examinations were performed in a female proband and in her relatives (15 subjects in total).

RESULTS

Three hemizygotes and 6 heterozygotes were diagnosed for FD by the alpha-Gal A gene analysis with a missense mutation in exon 5 of the alpha-Gal A sequence, leading to a TGG32TGA substitution, which may induce the absent of tryptophan's translation (corresponded to TGG) by the terminator codon TGA. Six patients in the family were revealed as HCM by echocardiography.

CONCLUSIONS

Present results show that it is important to differentiate FD from other causes of hypertrophy in patients with cardiac hypertrophy. Screening for alpha-Gal A gene mutations in patients with FD and in their relatives could help to identify all suspected cases within the families.

摘要

目的

在一个临床表现类似于肥厚型心肌病(HCM)的非近亲中国法布里病(FD)家族中筛查α-半乳糖苷酶A(α-Gal A)基因突变。

方法

使用纯化的PCR产物在ABI-377XL自动DNA测序仪上进行直接序列分析以进行突变分析。对一名女性先证者及其亲属(共15名受试者)进行了α-Gal A基因的DNA分析以及体格和临床检查。

结果

通过α-Gal A基因分析诊断出3名半合子和6名杂合子患有FD,α-Gal A序列外显子5存在错义突变,导致TGG32TGA替换,这可能导致终止密码子TGA使色氨酸翻译(对应于TGG)缺失。通过超声心动图显示该家族中有6名患者为HCM。

结论

目前的结果表明,区分FD与心脏肥厚患者的其他肥厚病因很重要。对FD患者及其亲属进行α-Gal A基因突变筛查有助于识别家族内所有疑似病例。

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