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E326K突变与戈谢病:突变还是多态性?

The E326K mutation and Gaucher disease: mutation or polymorphism?

作者信息

Park J K, Tayebi N, Stubblefield B K, LaMarca M E, MacKenzie J J, Stone D L, Sidransky E

机构信息

Section on Molecular Neurogenetics, National Institute of Mental Health, NIH, Bethesda, MD 20892, USA.

出版信息

Clin Genet. 2002 Jan;61(1):32-4. doi: 10.1034/j.1399-0004.2002.610106.x.

DOI:10.1034/j.1399-0004.2002.610106.x
PMID:11903352
Abstract

Gaucher disease is caused by mutations in the gene for human glucocerebrosidase, a lysosomal enzyme involved in the intracellular hydrolysis of glucosylceramide. While over 150 different glucocerebrosidase mutations have been identified in patients with Gaucher disease, not all reported mutations have been fully characterized as being causative. One such mutation is the E326K mutation, which results from a G to A nucleotide substitution at genomic position 6195 and has been identified in patients with type 1, type 2 and type 3 Gaucher disease. However, in each instance, the E326K mutation was found on the same allele with another glucocerebrosidase mutation. Utilizing polymerase chain reaction (PCR) screening and restriction digestions of both patients with Gaucher disease and normal controls, we identified the E326K allele in both groups. Of the 310 alleles screened from patients with Gaucher disease, the E326K mutation was detected in four alleles (1.3%). In addition, screening for the E326K mutation among normal controls from a random population revealed that three alleles among 316 screened (0.9%) also carried the E326K mutation. In the normal controls with the E326K allele, the glucocerebrosidase gene was completely sequenced, but no additional mutations were found. Because the E326K mutation may be a polymorphism, we caution that a careful examination of any allele with this mutation should be performed to check for the presence of other glucocerebrosidase mutations.

摘要

戈谢病是由人类葡萄糖脑苷脂酶基因的突变引起的,该酶是一种溶酶体酶,参与葡萄糖神经酰胺的细胞内水解。虽然在戈谢病患者中已鉴定出150多种不同的葡萄糖脑苷脂酶突变,但并非所有报道的突变都被充分表征为致病突变。其中一种突变是E326K突变,它是由基因组位置6195处的G到A核苷酸替换导致的,已在1型、2型和3型戈谢病患者中鉴定出。然而,在每种情况下,E326K突变都与另一种葡萄糖脑苷脂酶突变位于同一等位基因上。利用聚合酶链反应(PCR)筛查以及对戈谢病患者和正常对照进行限制性消化,我们在两组中都鉴定出了E326K等位基因。在从戈谢病患者中筛查的310个等位基因中,在4个等位基因(1.3%)中检测到了E326K突变。此外,在来自随机人群的正常对照中筛查E326K突变发现,在筛查的316个等位基因中有3个等位基因(0.9%)也携带E326K突变。在具有E326K等位基因的正常对照中,对葡萄糖脑苷脂酶基因进行了完全测序,但未发现其他突变。由于E326K突变可能是一种多态性,我们提醒,对于任何具有这种突变的等位基因都应进行仔细检查,以检查是否存在其他葡萄糖脑苷脂酶突变。

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