Kroos M, Manta P, Mavridou I, Muntoni F, Halley D, Van der Helm R, Zaifeiriou D, Van der Ploeg A, Reuser A, Michelakakis H
Department of Clinical Genetics, Erasmus MC, Rotterdam, The Netherlands.
J Inherit Metab Dis. 2006 Aug;29(4):556-63. doi: 10.1007/s10545-006-0280-5.
We present seven cases of Pompe disease (McKusick 232300; glycogen storage disease type II; acid maltase deficiency) from Greece. The onset of symptoms varied from early childhood to late adulthood, and the patients had quite variable duration of disease. All but one of them had muscle weakness and all had mildly to highly elevated serum creatine kinase. The diagnosis in all cases was confirmed by the finding of acid alpha-glucosidase (EC 3.2.1.3/20) deficiency in cultured skin fibroblasts. Thirteen mutant alleles were identified and nine different pathogenic mutations were encountered. Four were new: c.2071_2072insAGCCG leads to frameshift and total loss of function; c.1856G > A (p.Ser619Asn) leads to 90-95% loss of function; and the splice-site mutations c.1552-3C > G and c.2331+4A > G reduce the number of correct splicing events by more than 90%. The splice-site mutation c.-32-13T > G (IVS1-13T > G) was encountered four times and seems equally common among Greek and other caucasians. The other mutations: c.925G > A (p.Gly309Arg), c.[307T > G; 271G > A] (p.Cys103Gly; Asp91Asn), c.271del and c.1655T > C (p.Leu552Pro) have been reported earlier. Our study highlights the heterogeneity of Pompe disease in Greece and provides tools for diagnosis and carrier detection.
我们报告了来自希腊的7例庞贝病(麦库西克编号232300;II型糖原贮积病;酸性麦芽糖酶缺乏症)病例。症状出现的时间从幼儿期到成年晚期不等,患者的病程差异也很大。除1例患者外,其余患者均有肌肉无力症状,且所有患者的血清肌酸激酶均有轻度至高度升高。所有病例均通过在培养的皮肤成纤维细胞中发现酸性α-葡萄糖苷酶(EC 3.2.1.3/20)缺乏得以确诊。共鉴定出13个突变等位基因,发现了9种不同的致病突变。其中4种是新发现的:c.2071_2072insAGCCG导致移码突变和功能完全丧失;c.1856G > A(p.Ser619Asn)导致功能丧失90 - 95%;剪接位点突变c.1552 - 3C > G和c.2331+4A > G使正确剪接事件的数量减少了90%以上。剪接位点突变c.-32-13T > G(IVS1-13T > G)出现了4次,在希腊人和其他白种人中似乎同样常见。其他突变:c.925G > A(p.Gly309Arg)、c.[307T > G; 271G > A](p.Cys103Gly; Asp91Asn)、c.271del和c.1655T > C(p.Leu552Pro)此前已有报道。我们这项研究突出了希腊庞贝病的异质性,并为诊断和携带者检测提供了方法。