Yıl: 2023 Cilt: 65 Sayı: 6 Sayfa Aralığı: 973 - 979 Metin Dili: İngilizce DOI: 10.24953/turkjped.2023.227 İndeks Tarihi: 22-01-2024

Chest pain in children with familial Mediterranean fever

Öz:
Background. Familial Mediterranean fever (FMF) is the most common and autosomal recessive inherited autoinflammatory disease. The most common signs and symptoms are fever, abdominal pain, chest pain, and arthritis. The aim of this study was to describe the clinical, laboratory and genetic differences between pediatric FMF patients with and without chest pain. Methods. Between January 2006 and January 2022, 1134 patients with FMF were analyzed retrospectively. Patients were divided into two groups including those with and without recurrent chest pain. These groups were compared in demographic, clinical, treatment, and MEFV gene analyses. Results. A hundred and sixty-two (14.3%) patients had recurrent chest pain. In patients with recurrent chest pain, the age of onset of symptoms was younger (p=0.003), and the family history of FMF was higher (p=0.002). Patients with chest pain had a higher annual attack frequency (p<0.001), a longer attack duration (p<0.001), and higher Pras disease activity scores (p<0.001). The colchicine dose used in the treatment was higher in FMF patients with chest pain (p=0.005), and anti-IL-1treatment was higher (p<0.001). M694V homozygous mutation was found more frequently (p=0.001), whereas M694V/V726A mutation was found less frequently in patients with recurrent chest pain (p=0.017). Conclusions. Patients with recurrent chest pain seem to have early onset symptoms, often are more likely to have family history, and have a higher disease severity. In addition, the presence of homozygous M694V mutation is more common in patients with chest pain.
Anahtar Kelime: chest pain familial Mediterranean fever pleurisy pyrin

Belge Türü: Makale Makale Türü: Araştırma Makalesi Erişim Türü: Erişime Açık
  • 1. Sönmez HE, Batu ED, Özen S. Familial Mediterranean fever: current perspectives. J Inflamm Res 2016; 9: 13-20. https://doi.org/10.2147/JIR.S91352
  • 2. Barut K, Sahin S, Adrovic A, et al. Familial Mediterranean fever in childhood: a single-center experience. Rheumatol Int 2018; 38: 67-74. https:// doi.org/10.1007/s00296-017-3796-0
  • 3. Ben-Chetrit E, Levy M. Familial mediterranean fever. Lancet 1998; 351: 659-664. https://doi.org/10.1016/ S0140-6736(97)09408-7
  • 4. Tunca M, Akar S, Onen F, et al. Familial Mediterranean fever (FMF) in Turkey: results of a nationwide multicenter study. Medicine 2005; 84: 1-11. https:// doi.org/10.1097/01.md.0000152370.84628.0c
  • 5. Soylemezoglu O, Kandur Y, Gonen S, et al. Familial Mediterranean fever gene mutation frequencies in a sample Turkish population. Clin Exp Rheumatol 2016; 34: 97-100.
  • 6. French FMF Consortium. A candidate gene for familial Mediterranean fever. Nat Genet 1997; 17: 25-31. https://doi.org/10.1038/ng0997-25
  • 7. International FMF Consortium. Ancient missense mutations in a new member of the RoRet gene family are likely to cause familial Mediterranean fever. Cell 1997; 90: 797-807. https://doi.org/10.1016/ s0092-8674(00)80539-5
  • 8. Yalçinkaya F, Cakar N, Misirlioğlu M, et al. Genotype-phenotype correlation in a large group of Turkish patients with familial mediterranean fever: evidence for mutation-independent amyloidosis. Rheumatology (Oxford) 2000; 39: 67-72. https://doi. org/10.1093/rheumatology/39.1.67
  • 9. Touitou I. The spectrum of Familial Mediterranean Fever (FMF) mutations. Eur J Hum Genet 2001; 9: 473-483. https://doi.org/10.1038/sj.ejhg.5200658
  • 10. Papadopoulos VP, Giaglis S, Mitroulis I, Ritis K. The population genetics of familial mediterranean fever: a meta-analysis study. Ann Hum Genet 2008; 72: 752- 761. https://doi.org/10.1111/j.1469-1809.2008.00471.x
  • 11. Livneh A, Langevitz P, Zemer D, et al. Criteria for the diagnosis of familial Mediterranean fever. Arthritis Rheum 1997; 40: 1879-1885. https://doi.org/10.1002/ art.1780401023
  • 12. Livneh A, Langevitz P, Pras M. Pulmonary associations in familial Mediterranean fever. Curr Opin Pulm Med 1999; 5: 326-331. https://doi. org/10.1097/00063198-199909000-00011
  • 13. Yalçinkaya F, Ozen S, Ozçakar ZB, et al. A new set of criteria for the diagnosis of familial Mediterranean fever in childhood. Rheumatology (Oxford) 2009; 48: 395-398. https://doi.org/10.1093/rheumatology/ ken509
  • 14. Pras E, Livneh A, Balow JE, et al. Clinical differences between North African and Iraqi Jews with familial Mediterranean fever. Am J Med Genet 1998; 75: 216-219. https://doi.org/10.1002/(sici)1096- 8628(19980113)75:2<216::aid-ajmg20>3.0.co;2-r
  • 15. Öztürk K, Coşkuner T, Baglan E, et al. Real-Life data from the largest pediatric familial Mediterranean fever cohort. Front Pediatr 2022; 9: 805919. https:// doi.org/10.3389/fped.2021.805919
  • 16. Demir F, Bolac GL, Merter T, et al. The musculoskeletal system manifestations in children with familial Mediterranean fever. North Clin Istanb 2020; 7: 438-442. https://doi.org/10.14744/ nci.2020.96636
  • 17. Gezgin Yildirim D, Gönen S, Fidan K, Söylemezoğlu O. Does age at onset affect the clinical presentation of Familial Mediterranean Fever in children? J Clin Rheumatol 2022; 28: e125-e128. https://doi. org/10.1097/RHU.0000000000001637
  • 18. Tanatar A, Karadağ ŞG, Çakan M, Sönmez HE, Ayaz NA. Age of onset as an influencing factor for disease severity in children with familial Mediterranean fever. Mod Rheumatol 2021; 31: 219-222. https://doi. org/10.1080/14397595.2020.1719594
  • 19. Padeh S, Livneh A, Pras E, et al. Familial Mediterranean Fever in the first two years of life: a unique phenotype of disease in evolution. J Pediatr 2010; 156: 985-989. https://doi.org/10.1016/j. jpeds.2009.12.010
  • 20. Yalcinkaya F, Özçakar Z, Tanyıldız M. Paediatric rheumatology Familial Mediterranean fever in small children in Turkey. Clin Exp Rheumatol 2011; 29: S87-S90.
  • 21. Sönmezgöz E, Özer S, Gül A, et al. Clinical and demographic evaluation according to MEFV genes in patients with Familial Mediterranean Fever. Biochem Genet 2019; 57: 289-300. https://doi. org/10.1007/s10528-018-9889-y
  • 22. Cefle A, Kamali S, Sayarlioglu M, et al. A comparison of clinical findings of familial Mediterranean fever patients with and without amyloidosis. Rheumatol Int 2005; 25: 442-446. https://doi.org/10.1007/s00296- 004-0471-z
  • 23. Gezgin Yıldırım D, Esmeray Senol P, Söylemezoğlu O. Predictors of persistent inflammation in children with familial Mediterranean fever. Mod Rheumatol 2022; 32: 803-807. https://doi.org/10.1093/mr/roab054
  • 24. Babaoglu H, Armagan B, Bodakci E, et al. Predictors of persistent inflammation in familial Mediterranean fever and association with damage. Rheumatology (Oxford) 2021; 60: 333-339. https://doi.org/10.1093/ rheumatology/keaa378
  • 25. Kasifoglu T, Bilge SY, Sari I, et al. Amyloidosis and its related factors in Turkish patients with familial Mediterranean fever: a multicentre study. Rheumatology (Oxford) 2014; 53: 741-745. https:// doi.org/10.1093/rheumatology/ket400
  • 26. Kilic A, Varkal MA, Durmus MS, et al. Relationship between clinical findings and genetic mutations in patients with familial Mediterranean fever. Pediatr Rheumatol Online J 2015; 13: 59. https://doi. org/10.1186/s12969-015-0057-1
  • 27. Brik R, Gershoni-Baruch R, Shinawi M, Barak L, Bentur L. Pulmonary manifestations and function tests in children genetically diagnosed with FMF. Pediatr Pulmonol 2003; 35: 452-455. https://doi. org/10.1002/ppul.10270
  • 28. Cekin N, Akyurek ME, Pinarbasi E, Ozen F. MEFV mutations and their relation to major clinical symptoms of Familial Mediterranean Fever. Gene 2017; 626: 9-13. https://doi.org/10.1016/j. gene.2017.05.013
APA Sunar Yayla E, Esmeray Şenol P, GEZGİN YİLDİRİM D, Soylemezoglu O (2023). Chest pain in children with familial Mediterranean fever. , 973 - 979. 10.24953/turkjped.2023.227
Chicago Sunar Yayla Emine Nur,Esmeray Şenol Pelin,GEZGİN YİLDİRİM DENİZ,Soylemezoglu Oguz Chest pain in children with familial Mediterranean fever. (2023): 973 - 979. 10.24953/turkjped.2023.227
MLA Sunar Yayla Emine Nur,Esmeray Şenol Pelin,GEZGİN YİLDİRİM DENİZ,Soylemezoglu Oguz Chest pain in children with familial Mediterranean fever. , 2023, ss.973 - 979. 10.24953/turkjped.2023.227
AMA Sunar Yayla E,Esmeray Şenol P,GEZGİN YİLDİRİM D,Soylemezoglu O Chest pain in children with familial Mediterranean fever. . 2023; 973 - 979. 10.24953/turkjped.2023.227
Vancouver Sunar Yayla E,Esmeray Şenol P,GEZGİN YİLDİRİM D,Soylemezoglu O Chest pain in children with familial Mediterranean fever. . 2023; 973 - 979. 10.24953/turkjped.2023.227
IEEE Sunar Yayla E,Esmeray Şenol P,GEZGİN YİLDİRİM D,Soylemezoglu O "Chest pain in children with familial Mediterranean fever." , ss.973 - 979, 2023. 10.24953/turkjped.2023.227
ISNAD Sunar Yayla, Emine Nur vd. "Chest pain in children with familial Mediterranean fever". (2023), 973-979. https://doi.org/10.24953/turkjped.2023.227
APA Sunar Yayla E, Esmeray Şenol P, GEZGİN YİLDİRİM D, Soylemezoglu O (2023). Chest pain in children with familial Mediterranean fever. Turkish Journal of Pediatrics, 65(6), 973 - 979. 10.24953/turkjped.2023.227
Chicago Sunar Yayla Emine Nur,Esmeray Şenol Pelin,GEZGİN YİLDİRİM DENİZ,Soylemezoglu Oguz Chest pain in children with familial Mediterranean fever. Turkish Journal of Pediatrics 65, no.6 (2023): 973 - 979. 10.24953/turkjped.2023.227
MLA Sunar Yayla Emine Nur,Esmeray Şenol Pelin,GEZGİN YİLDİRİM DENİZ,Soylemezoglu Oguz Chest pain in children with familial Mediterranean fever. Turkish Journal of Pediatrics, vol.65, no.6, 2023, ss.973 - 979. 10.24953/turkjped.2023.227
AMA Sunar Yayla E,Esmeray Şenol P,GEZGİN YİLDİRİM D,Soylemezoglu O Chest pain in children with familial Mediterranean fever. Turkish Journal of Pediatrics. 2023; 65(6): 973 - 979. 10.24953/turkjped.2023.227
Vancouver Sunar Yayla E,Esmeray Şenol P,GEZGİN YİLDİRİM D,Soylemezoglu O Chest pain in children with familial Mediterranean fever. Turkish Journal of Pediatrics. 2023; 65(6): 973 - 979. 10.24953/turkjped.2023.227
IEEE Sunar Yayla E,Esmeray Şenol P,GEZGİN YİLDİRİM D,Soylemezoglu O "Chest pain in children with familial Mediterranean fever." Turkish Journal of Pediatrics, 65, ss.973 - 979, 2023. 10.24953/turkjped.2023.227
ISNAD Sunar Yayla, Emine Nur vd. "Chest pain in children with familial Mediterranean fever". Turkish Journal of Pediatrics 65/6 (2023), 973-979. https://doi.org/10.24953/turkjped.2023.227