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人α-半乳糖苷酶基因 5'非翻译区 g.1170C>T 多态性与酶表达降低相关——来自家系研究的证据。

The g.1170C>T polymorphism of the 5' untranslated region of the human alpha-galactosidase gene is associated with decreased enzyme expression--evidence from a family study.

机构信息

Department of Medical Genetics, Faculty of Medicine, University of Porto, Alameda Hernâni Monteiro, 4200-319, Porto, Portugal.

出版信息

J Inherit Metab Dis. 2008 Dec;31 Suppl 2:S405-13. doi: 10.1007/s10545-008-0972-0. Epub 2008 Nov 3.

DOI:10.1007/s10545-008-0972-0
PMID:18979178
Abstract

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.

摘要

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。

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本文引用的文献

1
Effect of single-nucleotide polymorphisms of the 5' untranslated region of the human α-galactosidase gene on enzyme activity, and their frequencies in Portuguese caucasians.人类α-半乳糖苷酶基因 5'非翻译区单核苷酸多态性对酶活性的影响及其在葡萄牙白种人中的频率。
J Inherit Metab Dis. 2008 Dec;31 Suppl 2:S247-53. doi: 10.1007/s10545-008-0818-9. Epub 2008 Nov 3.
2
Females with Fabry disease frequently have major organ involvement: lessons from the Fabry Registry.法布里病女性患者常出现主要器官受累:来自法布里病注册研究的经验教训。
Mol Genet Metab. 2008 Feb;93(2):112-28. doi: 10.1016/j.ymgme.2007.09.013. Epub 2007 Nov 26.
3
Appl Clin Genet. 2019 Mar 5;12:35-50. doi: 10.2147/TACG.S146022. eCollection 2019.
4
Alpha-Galactosidase A p.A143T, a non-Fabry disease-causing variant.α-半乳糖苷酶A p.A143T,一种非致法布里病的变异体。
Orphanet J Rare Dis. 2016 May 4;11(1):54. doi: 10.1186/s13023-016-0441-z.
5
Increased glycolipid storage produced by the inheritance of a complex intronic haplotype in the α-galactosidase A (GLA) gene.α-半乳糖苷酶A(GLA)基因中复杂内含子单倍型的遗传导致糖脂储存增加。
BMC Genet. 2015 Sep 3;16:109. doi: 10.1186/s12863-015-0267-z.
6
Variations in the GLA gene correlate with globotriaosylceramide and globotriaosylsphingosine analog levels in urine and plasma.GLA基因的变异与尿液和血浆中的球三糖神经酰胺及球三糖鞘氨醇类似物水平相关。
Clin Chim Acta. 2015 Jul 20;447:96-104. doi: 10.1016/j.cca.2015.06.003. Epub 2015 Jun 9.
7
The Modulatory Effects of the Polymorphisms in GLA 5'-Untranslated Region Upon Gene Expression Are Cell-Type Specific.GLA基因5'非翻译区多态性对基因表达的调节作用具有细胞类型特异性。
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8
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9
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10
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