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人甲羟戊酸激酶基因突变效应的计算机模拟预测:迈向甲羟戊酸激酶缺乏症的预测框架

In Silico Prediction of the Effects of Mutations in the Human Mevalonate Kinase Gene: Towards a Predictive Framework for Mevalonate Kinase Deficiency.

作者信息

Browne Claire, Timson David J

机构信息

School of Biological Sciences, Queen's University Belfast, Medical Biology Centre, 97 Lisburn Road, Belfast, BT9 7BL, UK.

Institute for Global Food Security, Queen's University Belfast, 18-30 Malone Road, Belfast, BT9 5BN, UK.

出版信息

Ann Hum Genet. 2015 Nov;79(6):451-9. doi: 10.1111/ahg.12126. Epub 2015 Sep 29.

Abstract

Mevalonate kinase (MVK) catalyses the phosphorylation of mevalonate. Deficiency of MVK is associated with two rare periodic fever syndromes, mevalonic aciduria (MA), a severe form and hyper-immunoglobulin-D syndrome (HIDS), a milder form. An in silico approach was used to analyse the physicochemical and structural effects of 47 disease-associated variants of MVK. A further 20 variants, which are present in human genome databases, were also analysed. Variants associated with MA are clustered into a "hotspot" consisting of residues 8-35 and 234-338 and tended to result in a prediction of severely reduced protein stability. Four of the uncharacterised variants, p.H24P, p.G198R, p. R253W, and p.G335S, were likely to be associated with MA. This method could be used as the basis for initial predictions of severity when new MVK variants are discovered.

摘要

甲羟戊酸激酶(MVK)催化甲羟戊酸的磷酸化。MVK缺乏与两种罕见的周期性发热综合征相关,即严重形式的甲羟戊酸尿症(MA)和较轻形式的高免疫球蛋白D综合征(HIDS)。采用计算机模拟方法分析了MVK的47种疾病相关变体的物理化学和结构效应。还分析了人类基因组数据库中存在的另外20种变体。与MA相关的变体聚集在一个由8-35位和234-338位残基组成的“热点”区域,并且倾向于导致蛋白质稳定性严重降低的预测。四种未表征的变体,p.H24P、p.G198R、p.R253W和p.G335S,可能与MA相关。当发现新的MVK变体时,该方法可作为严重程度初步预测的基础。

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