Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, South Korea.
Clin Genet. 2012 Feb;81(2):185-90. doi: 10.1111/j.1399-0004.2011.01641.x. Epub 2011 Feb 24.
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.
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。