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中国人患甲基丙二酸血症合并同型半胱氨酸尿症中 MMACHC 基因突变谱。

Mutation spectrum of MMACHC in Chinese patients with combined methylmalonic aciduria and homocystinuria.

机构信息

Institute of Genetics, National Yang-Ming University, Taipei, Taiwan.

出版信息

J Hum Genet. 2010 Sep;55(9):621-6. doi: 10.1038/jhg.2010.81. Epub 2010 Jul 15.

Abstract

The cblC type of combined methylmalonic aciduria (MMA) and homocystinuria (HC) is the most common inborn error of vitamin B(12) metabolism and is caused by mutations in the MMACHC gene. To elucidate the spectrum of mutations that causes combined MMA and HC in Chinese patients, the MMACHC gene was sequenced in 79 unrelated Chinese patients. Sequence analysis identified 98.1% of disease alleles and found that all patients had at least one MMACHC mutation. A total of 24 mutations were identified. Out of the 24 mutations identified, 9 were novel ones, including missense mutations (c.365A>T and c.452A>G), nonsense mutations (c.315C>G and c.615C>A), deletions (c.99delA and c.277-3_c.303del30), duplications (c.248dupT and c.626dupT) and an insertion (c.445_446insA). The c.609G>A, c.658_660delAAG, c.482G>A, c.394C>T and c.80A>G mutations were the most common mutations and accounted for 80% of disease alleles. Haplotype analysis suggests that the spread of the c.80A>G, c.609G>A and c.658_660delAAG mutations in Chinese patients were caused by a founder effect. The results indicate that defects occurring in the MMACHC gene are the major cause of this disease in Chinese patients with combined MMA and HC, and direct mutation analysis can therefore be used as a rapid confirmatory diagnosis among these Chinese patients.

摘要

cblC 型甲基丙二酸血症(MMA)合并同型半胱氨酸尿症(HC)是最常见的维生素 B(12)代谢先天性错误,由 MMACHC 基因突变引起。为阐明导致中国患者 MMA 和 HC 合并的突变谱,对 79 例无亲缘关系的中国患者进行了 MMACHC 基因测序。序列分析确定了 98.1%的疾病等位基因,并发现所有患者至少有一种 MMACHC 突变。共鉴定出 24 种突变。在鉴定的 24 种突变中,有 9 种是新的突变,包括错义突变(c.365A>T 和 c.452A>G)、无义突变(c.315C>G 和 c.615C>A)、缺失(c.99delA 和 c.277-3_c.303del30)、重复(c.248dupT 和 c.626dupT)和插入(c.445_446insA)。c.609G>A、c.658_660delAAG、c.482G>A、c.394C>T 和 c.80A>G 突变是最常见的突变,占疾病等位基因的 80%。单体型分析表明,c.80A>G、c.609G>A 和 c.658_660delAAG 突变在中国患者中的传播是由一个奠基者效应引起的。结果表明,MMACHC 基因缺陷是中国 MMA 和 HC 合并患者发病的主要原因,因此直接突变分析可作为这些中国患者的快速确诊方法。

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