Department of Medical Genetics, Faculty of Medicine, University of Porto, Alameda Hernâni Monteiro, 4200-319, Porto, Portugal.
J Inherit Metab Dis. 2008 Dec;31 Suppl 2:S405-13. doi: 10.1007/s10545-008-0972-0. Epub 2008 Nov 3.
Subnormal leukocyte α-galactosidase (α-Gal) activity was found during evaluation of an adolescent male with cryptogenic cerebrovascular small-vessel disease. The only molecular abnormality found was the g.1170C>T single-nucleotide polymorphism (SNP) in the 5' untranslated region of exon 1 in the α-Gal gene (GLA). Historically, this polymorphism has been considered to be biologically neutral. To test the hypothesis that the g.1170T allele might be associated with lower α-Gal expression, we genotyped GLA exon 1 and measured leukocyte and plasma α-Gal in the parents, brother and sister of the index case. The g.1170T allele co-segregated with a subnormal leukocyte α-Gal activity in the three siblings. Although plasma enzyme activities were within the normal range in all five relatives, the ranking of their values suggested a dosage effect of the g.1170T allele. Western blotting assays of leukocyte protein extracts showed that the relative expression of α-Gal in both the patient and his sister was significantly lower than in sex-matched hemizygous or homozygous controls for the g.1170C allele, either normalized to the β-actin immunoblot expression or standardized to a known amount of recombinant human α-Gal. These family data, in combination with results from a recent GLA SNP screening study among healthy Portuguese individuals, suggest that the g.1170C>T SNP may be co-dominantly associated with a relatively decreased GLA expression at the transcription and/or translation level. Larger population studies are needed to confirm these findings and to test the hypothesis that the GLA g.1170C>T may contribute to the multifactorial risk of ischaemic small-vessel cerebrovascular disease.
在评估一名患有隐源性脑血管小血管疾病的青少年男性时,发现白细胞α-半乳糖苷酶(α-Gal)活性低下。唯一发现的分子异常是α-Gal 基因(GLA)外显子 1 的 5'非翻译区的 g.1170C>T 单核苷酸多态性(SNP)。从历史上看,这种多态性被认为是生物学中性的。为了验证 g.1170T 等位基因可能与较低的α-Gal 表达相关的假设,我们对 GLA 外显子 1 进行了基因分型,并测量了指数病例的父母、兄弟姐妹的白细胞和血浆α-Gal。g.1170T 等位基因与三兄弟姐妹的白细胞α-Gal 活性低下共分离。尽管所有 5 名亲属的血浆酶活性均在正常范围内,但它们的值排序表明 g.1170T 等位基因存在剂量效应。白细胞蛋白提取物的 Western 印迹分析表明,患者及其姐姐的α-Gal 相对表达明显低于 g.1170C 等位基因的性别匹配半合子或纯合子对照,无论是与β-肌动蛋白免疫印迹表达归一化,还是与已知量的重组人α-Gal 标准化。这些家族数据,结合最近在健康葡萄牙个体中进行的 GLA SNP 筛查研究结果,表明 g.1170C>T SNP 可能与转录和/或翻译水平上相对降低的 GLA 表达呈共显性相关。需要进行更大的人群研究来证实这些发现,并检验 GLA g.1170C>T 是否可能导致缺血性小血管脑血管病的多因素风险增加的假设。