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鉴定 49 例韩国黏多糖贮积症 II 型患者中的 11 种新突变。

Identification of 11 novel mutations in 49 Korean patients with mucopolysaccharidosis type II.

机构信息

Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, South Korea.

出版信息

Clin Genet. 2012 Feb;81(2):185-90. doi: 10.1111/j.1399-0004.2011.01641.x. Epub 2011 Feb 24.

Abstract

Mucopolysaccharidosis type II (MPS II) or Hunter syndrome is a rare lysosomal storage disorder caused by a deficiency of iduronate-2-sulfatase (IDS). As MPS II is X-linked, patients are usually males with heterogeneous mutations ranging from point mutations to gross deletions and recombination. In 2003, we reported a mutation analysis of 25 patients with MPS II. In this study, 31 mutations in another 49 Korean patients (45 families) with MPS II are reported: 12 missense, nine deletions, four splicing, two nonsense, two insertions, one deletion/insertion, and IDS-IDS2 recombination mutations. Among these mutations, 11 were novel ones (4 missense mutations: Ser61Pro, Pro97Arg, Pro228Ala, and Pro261Ala; 5 deletions: c.344delA, c.420delG, c.768delT, c.1112delC and c.1402delC; 1 deletion/insertion: c.1222delinsTA; and 1 insertion mutation: c.359_360insATCC). The IDS-IDS2 recombination mutations were most frequently observed; all patients with this mutation had the severe MPS II phenotype. However, most of the patients (5/7) with the G374G splicing mutation had an attenuated phenotype, except for two sibling cases with the severe phenotype. Except for a few recurrent mutations such as the G374G, R443X, L522P, and recombination mutations, each patient had a unique individual mutation. Therefore, careful interpretation of genotype-phenotype correlations is warranted.

摘要

黏多糖贮积症 II 型(MPS II)或亨特综合征是一种罕见的溶酶体贮积病,由艾杜糖-2-硫酸酯酶(IDS)缺乏引起。由于 MPS II 是 X 连锁的,患者通常为男性,其突变从点突变到大片段缺失和重组不等。2003 年,我们报道了 25 例 MPS II 患者的突变分析。在本研究中,报告了另 49 例韩国 MPS II 患者(45 个家系)的 31 种突变:12 种错义突变、9 种缺失突变、4 种剪接突变、2 种无义突变、2 种插入突变、1 种缺失/插入突变和 IDS-IDS2 重组突变。在这些突变中,有 11 种是新的突变(4 种错义突变:Ser61Pro、Pro97Arg、Pro228Ala 和 Pro261Ala;5 种缺失突变:c.344delA、c.420delG、c.768delT、c.1112delC 和 c.1402delC;1 种缺失/插入突变:c.1222delinsTA;和 1 种插入突变:c.359_360insATCC)。IDS-IDS2 重组突变最为常见;所有携带这种突变的患者均具有严重的 MPS II 表型。然而,具有 G374G 剪接突变的大多数患者(5/7)表现为轻度表型,除了两例具有严重表型的同胞病例。除了一些反复出现的突变,如 G374G、R443X、L522P 和重组突变外,每个患者都有独特的个体突变。因此,需要仔细解释基因型-表型相关性。

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