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通过下一代测序确定的来自土耳其的一组麦克尔迪氏病患者中的肌磷酸化酶(PYGM)突变。

Myophosphorylase (PYGM) mutations determined by next generation sequencing in a cohort from Turkey with McArdle disease.

作者信息

Inal-Gültekin Güldal, Toptaş-Hekimoğlu Bahar, Görmez Zeliha, Gelişin Özlem, Durmuş Hacer, Ergüner Bekir, Demirci Hüseyin, Sağıroğlu Mahmut Ş, Parman Yeşim, Deymeer Feza, Yılmaz-Aydoğan Hülya, Pençe Sadrettin, Bekircan-Kurt Can Ebru, Tan Ersin, Erdem-Özdamar Sevim, Üstek Duran, Giger Urs, Öztürk Oğuz, Serdaroğlu-Oflazer Piraye

机构信息

Department of Molecular Medicine, Istanbul University, Aziz Sancar Institute of Experimental Medicine, Istanbul, Turkey.

The Scientific and Technological Research Council of Turkey (TUBITAK), Advanced Genomics and Bioinformatics Research Center (IGBAM), Kocaeli, Turkey.

出版信息

Neuromuscul Disord. 2017 Nov;27(11):997-1008. doi: 10.1016/j.nmd.2017.06.004. Epub 2017 Jun 16.

Abstract

This study aimed to identify PYGM mutations in patients with McArdle disease from Turkey by next generation sequencing (NGS). Genomic DNA was extracted from the blood of the McArdle patients (n = 67) and unrelated healthy volunteers (n = 53). The PYGM gene was sequenced with NGS and the observed mutations were validated by direct Sanger sequencing. A diagnostic algorithm was developed for patients with suspected McArdle disease. A total of 16 deleterious PYGM mutations were identified, of which 5 were novel, including 1 splice-site donor, 1 frame-shift, and 3 non-synonymous variants. The p.Met1Val (27-patients/11-families) was the most common PYGM mutation, followed by p.Arg576* (6/4), c.1827+7A>G (5/4), c.772+2_3delTG (5/3), p.Phe710del (4/2), p.Lys754Asnfs (2/1), and p.Arg50* (1/1). A molecular diagnostic flowchart is proposed for the McArdle patients in Turkey, covering the 6 most common PYGM mutations found in Turkey as well as the most common mutation in Europe. The diagnostic algorithm may alleviate the need for muscle biopsies in 77.6% of future patients. A prevalence of any of the mutations to a geographical region in Turkey was not identified. Furthermore, the NGS approach to sequence the entire PYGM gene was successful in detecting a common missense mutation and discovering novel mutations in this population study.

摘要

本研究旨在通过下一代测序(NGS)鉴定来自土耳其的麦克尔迪氏病患者中的PYGM突变。从麦克尔迪氏病患者(n = 67)和无关健康志愿者(n = 53)的血液中提取基因组DNA。使用NGS对PYGM基因进行测序,并通过直接桑格测序验证观察到的突变。为疑似麦克尔迪氏病患者开发了一种诊断算法。共鉴定出16种有害的PYGM突变,其中5种是新突变,包括1个剪接位点供体突变、1个移码突变和3个非同义变体。p.Met1Val(27例患者/11个家系)是最常见的PYGM突变,其次是p.Arg576*(6/4)、c.1827+7A>G(5/4)、c.772+2_3delTG(5/3)、p.Phe710del(4/2)、p.Lys754Asnfs(2/1)和p.Arg50*(1/1)。提出了针对土耳其麦克尔迪氏病患者的分子诊断流程图,涵盖了在土耳其发现的6种最常见的PYGM突变以及欧洲最常见的突变。该诊断算法可能会使未来77.6%的患者无需进行肌肉活检。未确定土耳其任何一个地理区域中这些突变的患病率。此外,在这项人群研究中,对整个PYGM基因进行测序的NGS方法成功检测到了一个常见的错义突变并发现了新的突变。

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本文引用的文献

2
McArdle Disease: Update of Reported Mutations and Polymorphisms in the PYGM Gene.
Hum Mutat. 2015 Jul;36(7):669-78. doi: 10.1002/humu.22806. Epub 2015 Jun 3.
3
Determining the prevalence of McArdle disease from gene frequency by analysis of next-generation sequencing data.
Genet Med. 2015 Dec;17(12):1002-6. doi: 10.1038/gim.2015.9. Epub 2015 Mar 5.
5
FORGE Canada Consortium: outcomes of a 2-year national rare-disease gene-discovery project.
Am J Hum Genet. 2014 Jun 5;94(6):809-17. doi: 10.1016/j.ajhg.2014.05.003.
6
HomSI: a homozygous stretch identifier from next-generation sequencing data.
Bioinformatics. 2014 Feb 1;30(3):445-7. doi: 10.1093/bioinformatics/btt686. Epub 2013 Dec 3.
8
Genotypic and phenotypic features of McArdle disease: insights from the Spanish national registry.
J Neurol Neurosurg Psychiatry. 2012 Mar;83(3):322-8. doi: 10.1136/jnnp-2011-301593. Epub 2012 Jan 16.
9
VarSifter: visualizing and analyzing exome-scale sequence variation data on a desktop computer.
Bioinformatics. 2012 Feb 15;28(4):599-600. doi: 10.1093/bioinformatics/btr711. Epub 2011 Dec 30.
10
Six novel mutations in the myophosphorylase gene in patients with McArdle disease and a family with pseudo-dominant inheritance pattern.
Mol Genet Metab. 2011 Dec;104(4):587-91. doi: 10.1016/j.ymgme.2011.08.012. Epub 2011 Aug 16.

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