Baris Ibrahim, Battaloglu Esra
Department of Molecular Biology and Genetics, Bogazici University, Istanbul, Turkey.
Genet Test Mol Biomarkers. 2009 Feb;13(1):19-22. doi: 10.1089/gtmb.2008.0051.
Rett syndrome (RTT), an X-linked dominant neurodevelopmental disorder, is caused mainly by de novo mutations in the methyl-CpG-binding protein 2 gene (MECP2). Although more than 200 different MECP2 mutations have been identified throughout the gene, 7 of those (p.R133C, p.T158M, p.R168X, p.R255X, p.R270X, p.R294X, and p.R306C) account for up to two-thirds of pathogenic mutations in RTT patients. A rapid and efficient screening strategy for these mutations can be used as a preliminary step for genetic diagnosis of RTT. The current protocols used for this purpose are of high cost and require special equipment. We have designed a simpler multiplex amplification refractory mutation system (ARMS)-PCR strategy that allows identification of these common MECP2 mutant alleles in four PCR reactions. The assay was tested in 14 RTT patients who were previously genotyped using PCR-restriction fragment length polymorphism and DNA sequencing. A complete concordance was observed between the results of the two methods. The multiplex ARMS-PCR does not require any special equipment, and it provides rapid, reproducible, and cost-effective detection of common MECP2 mutations. The assay can be carried out efficiently in a standard molecular genetics laboratory and suitable as a preliminary screen for all patients with RTT diagnosis.
雷特综合征(RTT)是一种X连锁显性神经发育障碍,主要由甲基CpG结合蛋白2基因(MECP2)的新发突变引起。尽管在整个基因中已鉴定出200多种不同的MECP2突变,但其中7种(p.R133C、p.T158M、p.R168X、p.R255X、p.R270X、p.R294X和p.R306C)占RTT患者致病突变的三分之二。针对这些突变的快速高效筛查策略可作为RTT基因诊断的初步步骤。目前用于此目的的方案成本高昂且需要特殊设备。我们设计了一种更简单的多重扩增不应性突变系统(ARMS)-PCR策略,该策略可在四个PCR反应中鉴定这些常见的MECP2突变等位基因。该检测方法在14名先前使用PCR-限制性片段长度多态性和DNA测序进行基因分型的RTT患者中进行了测试。两种方法的结果完全一致。多重ARMS-PCR不需要任何特殊设备,并且能够快速、可重复且经济高效地检测常见的MECP2突变。该检测方法可在标准分子遗传学实验室中高效进行,适合作为所有RTT诊断患者的初步筛查方法。