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鉴定伊朗黏多糖贮积症 VI 型患者的芳基硫酸酯酶 B 基因突变与临床表现。

Identification of arylsulfatase B gene mutations and clinical presentations of Iranian patients with Mucopolysaccharidosis VI.

机构信息

Diabetes Research Center, Health Research Institute, Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran; Department of Pediatrics, School of Medicine, Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran.

Department of Genetics, Shahid Chamran University of Ahvaz, Ahvaz, Iran.

出版信息

Gene. 2019 Jul 20;706:1-5. doi: 10.1016/j.gene.2019.04.050. Epub 2019 Apr 19.

Abstract

BACKGROUND

Mucopolysaccharidosis (MPS) type VI, also known as Maroteaux-Lamy syndrome, is an autosomal recessive lysosomal storage disorder caused by a deficiency in arylsulfatase B (ARSB) enzyme. Our objectives were to investigate clinical phenotypes and performed molecular studies in Iranian patients with MPS VI, for the first time, in the southwestern Iran.

METHODS

We studied 14 cases from 10 unrelated kindreds with MPS VI that were enrolled during 8 years. The mutational analysis of coding and flanking regions of ARSB gene was performed for the patients and their families using genomic DNA from whole blood by direct sequencing.

RESULTS

All cases had parental consanguinity. Except one who had Fars ethnicity and presented with a very mild degree of coarse face, but normal otherwise, even near normal height, all were from Arab ethnicity with characteristic phenotypes including severe facial changes, cardiac involvement and dysostosis multiplex. Sequencing analysis of ARSB gene revealed four pathogenic homozygote mutations, including a novel nonsense mutation c.281C>A (p.Ser94X) in 9 patients, as well as, a known nonsense mutation c.753C>G (p.Try251X) in 3 cases, and two missense mutations c.904G>A (p.Gly302Arg) and c.454C>T (p.Arg152Trp) in two cases. The type of mutations affected the severity patient's phenotypes.

CONCLUSIONS

These findings increased the genetic databases of Iranian patients with MPS VI and would be so much helpful for the high-risk families to speed the detection of carriers with accuracy and perform the prenatal test of disorder with cost-effective in this population.

摘要

背景

黏多糖贮积症(MPS)VI 型,也称为 Maroteaux-Lamy 综合征,是一种常染色体隐性溶酶体贮积病,由芳基硫酸酯酶 B(ARSB)酶缺乏引起。我们的目的是首次在伊朗西南部调查 MPS VI 型伊朗患者的临床表型,并进行分子研究。

方法

我们研究了 10 个无关家系的 14 例 MPS VI 患者,这些患者在 8 年内被纳入研究。使用来自全血的基因组 DNA 通过直接测序对 ARSB 基因的编码和侧翼区域进行了患者及其家属的突变分析。

结果

所有病例均有父母近亲结婚。除了一个来自法尔斯族,表现出非常轻微的粗面,其他都来自阿拉伯族,具有特征性表型,包括严重的面部变化、心脏受累和多发性骨发育不良。ARSB 基因测序分析显示了四种致病性纯合突变,包括 9 例患者中的新型无义突变 c.281C>A(p.Ser94X),以及 3 例患者中的已知无义突变 c.753C>G(p.Try251X),和两个错义突变 c.904G>A(p.Gly302Arg)和 c.454C>T(p.Arg152Trp)在两例患者中。突变类型影响患者表型的严重程度。

结论

这些发现增加了伊朗 MPS VI 型患者的遗传数据库,将极大地帮助高危家庭在该人群中以经济有效的方式准确检测携带者并进行疾病的产前检测。

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