Kang Seong-Ho, Cho Sung Im, Chae Jong-Hee, Chung Kyu Nam, Ra Eun Kyung, Kim So Yeon, Seong Moon-Woo, Kim Ji Yeon, Park Sung Sup
Department of Laboratory Medicine, Seoul National University Hospital, Seoul, Korea.
Genet Test Mol Biomarkers. 2009 Aug;13(4):511-3. doi: 10.1089/gtmb.2008.0158.
Spinal muscular atrophy (SMA) is an autosomal recessive neuromuscular disorder, and about 95% of SMA patients are homozygous for deletions in the SMN1 gene. Herein, classical polymerase chain reaction and restriction fragment length polymorphism (PCR-RFLP) using DraI yielded false homozygous deletions of SMN1 exon 7 in a patient with SMA, but multiple ligation-dependent probe amplification analysis revealed one remaining copy of SMN1 exon 7. Sequencing showed that this false deletion in the PCR-RFLP resulted from a novel mutation of one SMN1 copy that was not deleted (c.863G > T, p.R288M). This novel sequence variant introduced a mismatch that interfered with primer binding. These findings demonstrate that comprehensive analysis using PCR-RFLP, multiple ligation-dependent probe amplification, and sequencing can reliably and correctly diagnose SMA.
脊髓性肌萎缩症(SMA)是一种常染色体隐性神经肌肉疾病,约95%的SMA患者SMN1基因缺失呈纯合状态。在此,使用DraI的经典聚合酶链反应和限制性片段长度多态性(PCR-RFLP)在一名SMA患者中产生了SMN1基因外显子7的假纯合缺失,但多重连接依赖探针扩增分析显示仍有一个SMN1基因外显子7的拷贝。测序表明,PCR-RFLP中的这种假缺失是由一个未缺失的SMN1拷贝的新突变(c.863G>T,p.R288M)导致的。这种新的序列变异引入了一个错配,干扰了引物结合。这些发现表明,使用PCR-RFLP、多重连接依赖探针扩增和测序进行综合分析可以可靠且正确地诊断SMA。