Literature DB >> 31989427

A comprehensive molecular analysis and genotype-phenotype correlation in patients with familial mediterranean fever.

Burhan Balta1, Murat Erdogan2, Aslıhan Kiraz2, Tayfun Akalın3, Funda Baştug4, Arslan Bayram5.   

Abstract

Familial Mediterranean fever is an auto inflammatory genetic disease involving especially Turks, Armenians, Arabs and non-Ashkenazi Jews and caused by variants in the MEFV gene. In this study, we aimed to evaluate the distribution and frequency of clinical, MEFV gene variants in FMF patients and the relationship between mutations in different exons and phenotype-genotype and clinical findings. 1028 patients diagnosed as FMF were included. The most common genotypes were M694V / R202Q heterozygous (10.4%), M694V homozygous (7.5%), M694V / E148Q / R202Q heterozygous (4.6%), V726A heterozygous (4.5%), M680I heterozygous (4.2%). c.1611-1 G > C, G152R, S104C, R116S, E336K, R461Q mutations were detected in the literature for the first time in FMF patients. We also divided the patients into 4 groups according to whether the MEFV mutations were exon 10 or non-exon 10. The first group consisted of non-exon 10 homozygous or compound heterozygous (n = 180) patients, Group 2 consisted of exon 10- non-exon 10 compound heterozygous (n = 318) patients, Group 3 consisted of exon 10 homozygous or compound heterozygous (n = 256) patients, while Group 4 consisted of heterozygous (n = 227) patients at any exon. There was no significant difference between the groups in terms of abdominal pain, arthritis, arthralgia, vomiting diarrhea, erysipelas like rash, amyloidosis, renal failure family history. There was no difference in fever between Group 1 (55.6%) and 2 (62.3%); however, these two groups were different from Group 3 (75.8%) and 4 (76.7%). Group 3 (18.8%) had the highest rate of appendectomy. In addition, allele frequencies of all mutations detected in the analyses were compared with allele frequencies of healthy people in the gnomad database. It is useful to analyse all exons in the MEFV gene with the next generation sequence analysis in the detection of FMF disease. S104C, R116S, G152R, E336K, R461Q, L508Q and c.1611-1 G > C mutations are also new variants in literature. c.1611-1 G > C is a possible pathogenic variant.

Entities:  

Keywords:  Autoinflammatory disease; FMF; Familial mediterranean fever; MEFV

Mesh:

Substances:

Year:  2020        PMID: 31989427     DOI: 10.1007/s11033-020-05277-x

Source DB:  PubMed          Journal:  Mol Biol Rep        ISSN: 0301-4851            Impact factor:   2.316


  14 in total

1.  Amyloidosis induced, end stage renal disease in patients with familial Mediterranean fever is highly associated with point mutations in the MEFV gene.

Authors:  E Ben-Chetrit; R Backenroth
Journal:  Ann Rheum Dis       Date:  2001-02       Impact factor: 19.103

2.  Genotype-phenotype studies in a large cohort of Armenian patients with familial Mediterranean fever suggest clinical disease with heterozygous MEFV mutations.

Authors:  Mike M Moradian; Tamara Sarkisian; Hasmik Ajrapetyan; Nare Avanesian
Journal:  J Hum Genet       Date:  2010-05-20       Impact factor: 3.172

Review 3.  Evidence-based recommendations for genetic diagnosis of familial Mediterranean fever.

Authors:  Gabriella Giancane; Nienke M Ter Haar; Nico Wulffraat; Sebastiaan J Vastert; Karyl Barron; Veronique Hentgen; Tilmann Kallinich; Huri Ozdogan; Jordi Anton; Paul Brogan; Luca Cantarini; Joost Frenkel; Caroline Galeotti; Marco Gattorno; Gilles Grateau; Michael Hofer; Isabelle Kone-Paut; Jasmin Kuemmerle-Deschner; Helen J Lachmann; Anna Simon; Erkan Demirkaya; Brian Feldman; Yosef Uziel; Seza Ozen
Journal:  Ann Rheum Dis       Date:  2015-01-27       Impact factor: 19.103

4.  Familial Mediterranean fever in childhood: a single-center experience.

Authors:  Kenan Barut; Sezgin Sahin; Amra Adrovic; Ada Bulut Sinoplu; Gozde Yucel; Gizem Pamuk; Aslı Kirectepe Aydın; Selcuk Dasdemir; Eda Tahir Turanlı; Nur Buyru; Ozgur Kasapcopur
Journal:  Rheumatol Int       Date:  2017-08-21       Impact factor: 2.631

Review 5.  The spectrum of Familial Mediterranean Fever (FMF) mutations.

Authors:  I Touitou
Journal:  Eur J Hum Genet       Date:  2001-07       Impact factor: 4.246

6.  The MEFV mutations and their clinical correlations in children with familial Mediterranean fever in southeast Turkey.

Authors:  Aydın Ece; Erdal Çakmak; Ünal Uluca; Selvi Kelekçi; İlyas Yolbaş; Ali Güneş; Servet Yel; İlhan Tan; Velat Şen
Journal:  Rheumatol Int       Date:  2013-09-26       Impact factor: 2.631

7.  Common Familial Mediterranean Fever gene mutations in a Turkish cohort.

Authors:  Munis Dundar; Elif Funda Emirogullari; Aslihan Kiraz; Serpil Taheri; Mevlut Baskol
Journal:  Mol Biol Rep       Date:  2010-12-14       Impact factor: 2.316

Review 8.  Familial Mediterranean fever (FMF) and renal AA amyloidosis--phenotype-genotype correlation, treatment and prognosis.

Authors:  Eldad Ben-Chetrit
Journal:  J Nephrol       Date:  2003 May-Jun       Impact factor: 3.902

9.  Genotype-phenotype correlation in children with familial Mediterranean fever in a Turkish population.

Authors:  Ruhan Duşunsel; Ismail Dursun; Zübeyde Gündüz; M Hakan Poyrazoğlu; Metin Kaya Gürgöze; Munis Dundar
Journal:  Pediatr Int       Date:  2008-04       Impact factor: 1.524

10.  Decreased Chitotriosidase Activity and Levels in Familial Mediterranean Fever.

Authors:  Halef Okan Doğan; Ahmet Omma; Turan Turhan; Nihal Boğdaycıoğlu; Yaşar Karaaslan; Hayrettin Yavuz; Özlem Demirpençe; Hüseyin Aydın; Sevtap Bakır
Journal:  J Korean Med Sci       Date:  2016-12       Impact factor: 2.153

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  2 in total

Review 1.  Multifactorial Rare Diseases: Can Uncertainty Analysis Bring Added Value to the Search for Risk Factors and Etiopathogenesis?

Authors:  Domenica Taruscio; Alberto Mantovani
Journal:  Medicina (Kaunas)       Date:  2021-01-28       Impact factor: 2.430

2.  Phenotypic characterization of Familial Mediterranean Fever patients harboring variants of uncertain significance

Authors:  Alper Sarı; Erdal Bodakçi; Berkan Armağan; Hasan Satış; Nuh Ataş; Nazife Şule Yaşar Bilge; Reyhan Bilici Salman; Gözde Kübra Yardımcı; Hakan Babaoğlu; Levent Kılıç; Mehmet Akif Öztürk; Şeminur Haznedaroğlu; Berna Göker; Umut Kalyoncu; Timuçin Kaşifoğlu; Abdurrahman Tufan
Journal:  Turk J Med Sci       Date:  2021-08-30       Impact factor: 0.973

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