| Literature DB >> 28967462 |
Güldal Inal-Gültekin1, Bahar Toptaş-Hekimoğlu1, Zeliha Görmez2, Özlem Gelişin3, Hacer Durmuş3, Bekir Ergüner2, Hüseyin Demirci2, Mahmut Ş Sağıroğlu2, Yeşim Parman3, Feza Deymeer3, Hülya Yılmaz-Aydoğan1, Sadrettin Pençe1, Can Ebru Bekircan-Kurt4, Ersin Tan4, Sevim Erdem-Özdamar4, Duran Üstek5, Urs Giger6, Oğuz Öztürk1, Piraye Serdaroğlu-Oflazer7.
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.Entities:
Keywords: Genomics; Glycogenosis; Molecular screening; Novel mutation; Population specific; Rare muscle disorders
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Year: 2017 PMID: 28967462 PMCID: PMC5698850 DOI: 10.1016/j.nmd.2017.06.004
Source DB: PubMed Journal: Neuromuscul Disord ISSN: 0960-8966 Impact factor: 4.296