| Literature DB >> 31522233 |
Hatice Bodur1, Fatma Gül Yurdakul2, Hasan Fatih Çay3, Ülkü Uçar3,4, Yaşar Keskin5, Betül Sargın6,7, Gülcan Gürer6, Ozan Volkan Yurdakul5, Mustafa Çalış8, Hülya Deveci9, Yıldıray Aydın10, Sami Hizmetli11, Remzi Çevik12, Ali Yavuz Karahan13, İsmihan Sunar14, Mehmet Tuncay Duruöz15, Hilal Ecesoy16, Zafer Günendi17, Murat Toprak18, Nesrin Şen19, Duygu Altıntaş20, Ahmet Kıvanç Cengiz21,22, Gökhan Çağlayan11, Ali Nail Demir23, Hüseyin Kaplan8,24, Sertaç Ketenci25, Meltem Alkan Melikoğlu20, Mehmet Nayimoğlu26, Kemal Nas27, Ayşe Banu Sarıfakıoğlu26, İlhan Sezer23.
Abstract
The aims of this study were to investigate the main clinical and laboratory features, including pregnancy and genetic analysis, of Turkish Familial Mediterranean Fever (FMF) patients and to analyze the relationships between genotypic features, age of disease onset, clinical findings, and disease severity. A study was planned within a national network of 22 different centers. Demographics, clinical and laboratory findings, attack characteristics, drugs, pregnancy and birth history, disease severity, and gene mutation analyses were evaluated. Disease severity, assessed using a scoring system developed by Pras et al., was evaluated in relation to gene mutations and age of disease onset. A total of 979 patients (643 females and 336 males; mean age: 35.92 ± 11.97 years) with FMF were included in the study. Of a total of 585 pregnancies, 7% of them resulted in preterm birth and 18.1% resulted in abortions. During pregnancy, there was no FMF attack in 61.4% of patients. Of the MEditerranean FeVer (MEFV) mutations, 150 (24.3%) cases were homozygous, 292 (47.3%) cases were heterozygous, and 175 (28.4%) were compound heterozygous. Patients with homozygous gene mutations had more severe disease activity, earlier age of disease onset, higher rates of joint and skin involvement, sacroiliitis, and amyloidosis. Patients with compound heterozygous genotype displayed severe disease activity in close resemblance to patients with homozygous mutation. In addition, patients with compound heterozygous mutations had higher rates of protracted febrile myalgia and elevated fibrinogen levels. In 63.9% of compound heterozygous patients, age of onset was < 20 years, with greater disease severity, and high rates of attack frequency and colchicine resistance. Our results suggest that indicators for disease severity include early onset of disease and homozygous gene mutations. Furthermore, patients with compound heterozygous mutations displayed significant presentations of severe disease activity.Entities:
Keywords: Amyloidosis; Colchicine; Mutation; Pregnancy
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Year: 2019 PMID: 31522233 DOI: 10.1007/s00296-019-04443-0
Source DB: PubMed Journal: Rheumatol Int ISSN: 0172-8172 Impact factor: 2.631