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多重连接依赖探针扩增技术改善了脊髓性肌萎缩症的诊断。

Multiplex ligation-dependent probe amplification improves diagnostics in spinal muscular atrophy.

作者信息

Arkblad Eva L, Darin Niklas, Berg Kerstin, Kimber Eva, Brandberg Göran, Lindberg Christopher, Holmberg Eva, Tulinius Mar, Nordling Margareta

机构信息

Department of Clinical Genetics, Sahlgrenska University Hospital/East, Göteborg, Sweden.

出版信息

Neuromuscul Disord. 2006 Dec;16(12):830-8. doi: 10.1016/j.nmd.2006.08.011. Epub 2006 Oct 17.

Abstract

Spinal muscular atrophy (SMA) is an autosomal recessive disease caused by decreased levels of survival motor neuron protein (SMN). In the majority of cases, this decrease is due to absence of the SMN1 gene. Multiplex ligation-dependent probe amplification (MLPA) is a modern quantitative molecular method. Applied in SMA cases, it improves diagnostics by simultaneously identifying the number of copies of several target sequences in the SMN1 gene and in nearby genes. Using MLPA in clinical diagnostics, we have identified a previously unreported, partial deletion of SMN1 (exons 1-6) in two apparently unrelated Swedish families. This mutation would not have been detected by conventional diagnostic methods. This paper illustrates the broad clinical and genetic spectrum of SMA and includes reports of MLPA results and clinical descriptions of a patient with homozygous absence of SMN1 and only one SMN2 (prenatal onset SMA type 1), an asymptomatic woman with five SMN2 (lacking SMN1) and representative patients with SMA types 1, 2 and 3.

摘要

脊髓性肌萎缩症(SMA)是一种常染色体隐性疾病,由存活运动神经元蛋白(SMN)水平降低引起。在大多数情况下,这种降低是由于缺乏SMN1基因所致。多重连接依赖探针扩增技术(MLPA)是一种现代定量分子方法。应用于SMA病例时,它通过同时鉴定SMN1基因及附近基因中几个靶序列的拷贝数来改善诊断。在临床诊断中使用MLPA时,我们在两个明显无亲缘关系的瑞典家庭中发现了一种先前未报道的SMN1基因部分缺失(外显子1 - 6)。这种突变用传统诊断方法无法检测到。本文阐述了SMA广泛的临床和遗传谱系,包括MLPA结果报告以及SMN1纯合缺失且仅有一个SMN2的患者(产前发病的1型SMA)、有五个SMN2(缺乏SMN1)的无症状女性以及1型、2型和3型SMA代表性患者的临床描述。

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