Coşkun Salih, Kurtgöz Serkan, Keskin Ece, Sönmez Ferah, Bozkurt Gökay
Medical Faculty, Department of Medical Genetics, Dicle University, 21280 Diyarbakir, Turkey.
J Genet. 2015 Dec;94(4):629-35. doi: 10.1007/s12041-015-0568-z.
Familial Mediterranean fever (FMF) is the most common hereditary inflammatory periodic disease, characterized by recurrent episodes of fever, abdominal pain, synovitis and pleurisy. The aim of this study was to determine the frequency and distribution of Mediterranean fever (MEFV) gene mutations and to investigate the clinical characteristics and genotype-phenotype correlation in patients with FMF in Aydin, a province in western Anatolia, Turkey. Therefore, we retrospectively analysed MEFV gene mutations in 383 patients with suspected FMF and the clinical features of 327 among them. The MEFV gene mutations were investigated using the reverse dot-blot hybridization technique. We detected 26 different genotypes and 11 different mutations. The most common mutations in our cohort were p.M694V (41.15%), p.E148Q (20.35%), p.M680I(G/C) (12.39%) and p.R761H (9.73%). Abdominal pain (86.2%), fever (80.7%), arthralgia (57.2%), vomiting (36.1%), arthritis (34.6%), fatigue (31.5%), anorexia (22.9%) and chest pain (19.0%) were the most prevalent clinical features in our patients. This is the first study from Aydin in which the distribution of MEFV gene mutations and clinical features were evaluated in patients with FMF. We found that the most common mutation was p.M694V in our region, while the frequency of the p.R761H mutation was higher compared to other regions of Turkey with respect to extracted data from previous similar studies. Presented results supported the clinical findings in the literature that the homozygous p.M694V and compound heterozygous genotype were associated with more severe courses in FMF patients.
家族性地中海热(FMF)是最常见的遗传性炎症性周期性疾病,其特征为发热、腹痛、滑膜炎和胸膜炎反复发作。本研究旨在确定地中海热(MEFV)基因突变的频率和分布,并调查土耳其安纳托利亚西部省份艾登的FMF患者的临床特征及基因型-表型相关性。因此,我们回顾性分析了383例疑似FMF患者的MEFV基因突变情况,并分析了其中327例患者的临床特征。采用反向点杂交技术检测MEFV基因突变。我们检测到26种不同的基因型和11种不同的突变。我们队列中最常见的突变是p.M694V(41.15%)、p.E148Q(20.35%)、p.M680I(G/C)(12.39%)和p.R761H(9.73%)。腹痛(86.2%)、发热(80.7%)、关节痛(57.2%)、呕吐(36.1%)、关节炎(34.6%)、疲劳(31.5%)、厌食(22.9%)和胸痛(19.0%)是我们患者中最常见的临床特征。这是艾登首次对FMF患者的MEFV基因突变分布和临床特征进行评估的研究。我们发现,在我们地区最常见的突变是p.M694V,而根据以往类似研究提取的数据,与土耳其其他地区相比,p.R761H突变的频率更高。呈现的结果支持了文献中的临床发现,即纯合子p.M694V和复合杂合子基因型与FMF患者更严重的病程相关。