Case Report
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FMF Hastasında Aseptik Temporomandibular Eklem Sinoviti ile birlikte Seyreden Yaygın Enflamasyon: Vaka Raporu

Year 2022, Volume: 7 Issue: 1, 31 - 36, 01.04.2022

Abstract

Özet:
Ailevi Akdeniz ateşi (FMF), kendini sınırlayan, provoke edilmemiş ateşli ve poliserozit ataklarıyla karakterize sistemik otoinflamatuar hastalıktır. Temporomandibular eklem (TME) tutulumu nadir olsada, yaşam kalitesini önemli ölçüde düşer ve bazı hastalıklar ile benzer semptomlar nedeniyle yanlış teşhis edilebilir. Birkaç vakada farklı cerrahi yaklaşımlar bildirilmesine ragmen, kabul edilmiş tedavi yöntemi yoktur.
Olgu Sunumu
Şiddetli unilateral TME bölgesi ağrısı ve ağız açıklığında kısıtlılık nedeni ile 11 yaşında kadın hasta merkezime baş vurmuştur. Alınan anemnezde kolşisin tedavisi altında olmasına rağmen ayda bir FMF atakları geçirdiği bildirilmiştir. Radyoloji departmanı konsültasyonu sonrası kontrastlı manyetik rezonans görüntüleme (MRI) gerçekleştirildi. MRG raporunun septik artrit ve osteomiyelit gibi enfektif hastalıklara yönlendirmesine rağmen, FMF ataklarının sıklığı nedeniyle diğer enflamatuar süreçler yeniden değerlendirildi. Lokal anestezi altında bakteriyolojik inceleme amaçlı sinovyal sıvı alındı. Artrosentez sonrası semptomlarda hızlı bir rahatlatama gözlemlendi. Sinovyal sıvı incelemesinde bakteri üremesi tespit edilmedi. Kolşisin doz ayarlaması ile altı aylık takipte FMF atağı gözlenmedi ve ilave operasyona ihtiyaç duyulmadı.
SONUÇ
FMF'e bağlı TME artriti çok nadir görülür ve diğer hastalıklarla örtüşen semptomlar yanlış tanıya yol açabilir. Bu durumun tedavisi ile ilgili bir görüş birliği yoktur. Akut ve subakut semptomlar, kolşisin dozunun artırılması ve artrosentez gibi non-invaziv yöntemlerle hızlı bir şekilde giderilebilir.

References

  • 1. Frenkel B, Berg T, Totry A, Rachmiel A. A novel treatment of temporomandibular joint arthritis as a complication in familial Mediterranean fever—literature review and a case report. Oral Maxillofac Surg. 2018;22(4):365–8.
  • 2. Hentgen V, Grateau G, Kone-Paut I, Livneh A, Padeh S, Rozenbaum M, et al. Evidence-based recommendations for the practical management of Familial Mediterranean Fever. Semin Arthritis Rheum [Internet]. 2013;43(3):387–91. Available from: http://dx.doi.org/10.1016/j.semarthrit.2013.04.011
  • 3. Uthman I, Hajj-Ali RA, Arayssi T, Masri AF, Nasr F. Arthritis in familial Mediterranean fever. Rheumatol Int. 2001;20(4):145–8.
  • 4. Berkun Y, Eisenstein EM. Diagnostic criteria of familial Mediterranean fever. Autoimmun Rev [Internet]. 2014;13(4–5):388–90. Available from: http://dx.doi.org/10.1016/j.autrev.2014.01.045
  • 5. Berkun Y, Padeh S, Reichman B. A Single Testing of Serum Amyloid A Levels as a Tool for Diagnosis and. 2007;(6):182–8.
  • 6. Mukhin NA, Kozlovskaya L V., Bogdanova M V., Rameev V V., Moiseev S V., Simonyan AK. Predictors of AA amyloidosis in familial Mediterranean fever. Rheumatol Int. 2015;35(7):1257–61.
  • 7. Jarjour RA, Dodaki R. Arthritis patterns in familial Mediterranean fever patients and association with M694V mutation. Mol Biol Rep. 2011;38(3):2033–6.
  • 8. Migita K, Hisanaga S, Izumi Y, Kawahara C, Shigemitsu Y, Iwanaga N, et al. Protracted arthritis in a Japanese patient with familial Mediterranean fever. Mod Rheumatol. 2017;27(5):905–9.
  • 9. Ozen S, Demirkaya E, Erer B, Livneh A, Ben-Chetrit E, Giancane G, et al. EULAR recommendations for the management of familial Mediterranean fever. Ann Rheum Dis. 2016;75(4):644–51.
  • 10. El Hasbani G, Jawad A, Uthman I. Update on the management of colchicine resistant Familial Mediterranean Fever (FMF). Orphanet J Rare Dis. 2019;14(1):1–12.
  • 11. Dinarello CA, Wolff SM, Goldfinger SE, Dale DC, Alling DW. Colchicine therapy for familial mediterranean fever. A double-blind trial. N Engl J Med [Internet]. 1974 Oct 31;291(18):934–7. Available from: http://www.ncbi.nlm.nih.gov/pubmed/4606353
  • 12. Yalçinkaya F, Çakar N, Misirlioǧlu M, Tümer N, Akar N, Tekin M, et al. Genotype-phenotype correlation in a large group of Turkish patients with familial Mediterranean fever: Evidence for mutation-independent amyloidosis. Rheumatology. 2000;39(1):67–72.
  • 13. Giaglis S, Papadopoulos V, Kambas K, Doumas M, Tsironidou V, Rafail S, et al. MEFV alterations and population genetics analysis in a large cohort of Greek patients with familial Mediterranean fever. Clin Genet. 2007;71(5):458–67.
  • 14. Simon G, Marbach JJ. Familial mediterranean fever with temporomandibular joint arthritis. Pediatrics [Internet]. 1976;57(5):810–2. Available from: https://pediatrics.aappublications.org/content/57/5/810
  • 15. Cooksey DE, Girard K. Temporomandibular joint synovitis with effusion in familial Mediterranean fever. Oral Surgery, Oral Med Oral Pathol. 1979;47(2):123–6.
  • 16. Tovi F, Gatot A, Fliss D. Temporomandibular arthritis in familial mediterranean fever. Head Neck. 1992;14(6):492–5.
  • 17. Tovi F, Barmeir E, Peist M, Bar-Ziv J. Protracted temporomandibular joint arthritis in familial Mediterranean fever. J Oral Maxillofac Surg [Internet]. 1985;43(6):466–8. Available from: http://dx.doi.org/10.1016/S0278-2391(85)80059-8
  • 18. Usluer H, Bircan Z. Protracted familial mediterranean fever arthritis presenting as septic arthritis. Rheumatol Int. 2007;27(11):1083–5.
  • 19. Simon G, Marbach JJ. Familial Mediterranean fever with temporomandibular joint arthritis. Pediatrics. 1976;57(5):810–2.
  • 20. Marzook HAM, Abdel Razek AA, Yousef EA, Attia AAMM. Intra-articular injection of a mixture of hyaluronic acid and corticosteroid versus arthrocentesis in TMJ internal derangement. Vol. 121, Journal of Stomatology, Oral and Maxillofacial Surgery. 2020. p. 30–4.
  • 21. Chuong R, Piper MA. Avascular necrosis of the mandibular condyle-pathogenesis and concepts of management. Oral Surgery, Oral Med Oral Pathol. 1993;75(4):428–32.

Aseptic Arthritis of the Temporomandibular Joint with Severe Inflammation in an FMF Patient: A Case Report

Year 2022, Volume: 7 Issue: 1, 31 - 36, 01.04.2022

Abstract

BACKGROUND
Familial Mediterranean fever (FMF) is a systemic autoinflammatory disorder characterized by self-limited unprovoked inflammatory attacks of fever and polyserositis. Although temporomandibular joint (TMJ) involvement is uncommon, it significantly reduces the quality of life and could be easily misdiagnosed due to overlapping symptoms. Only a few cases have been reported with different surgical approaches, but there are no specific treatment guidelines.
CASE SUMMARY
The case of an 11-year-old female FMF patient with severe unilateral TMJ region pain and limited maximum mouth opening is presented. Although the patient was under colchicine treatment, the frequency of FMF attacks was once a month. After consultation with the radiology department, contrast-enhanced magnetic resonance imaging (MRI) was performed. The differential diagnosis of the MRI report was infectious processes (septic arthritis and osteomyelitis). Nevertheless, other inflammatory processes were evaluated due to the frequency of FMF attacks. Under local anesthesia, synovial fluid was taken for bacteriological examination, and arthrocentesis was performed. Symptoms were relieved immediately after the operation. Bacterial growth was not detected on synovial fluid examination. No new FMF attacks were reported, and no new operations were needed in the six-month follow-up with colchicine dosage adjustment.
CONCLUSION
TMJ arthritis due to FMF is seen very rarely, and overlapping symptoms could lead to misdiagnosis. Although there is no consensus regarding the treatment of TMJ arthritis, acute and subacute symptoms could be relieved very quickly with less invasive methods, such as increasing the dose of colchicine and arthrocentesis.

References

  • 1. Frenkel B, Berg T, Totry A, Rachmiel A. A novel treatment of temporomandibular joint arthritis as a complication in familial Mediterranean fever—literature review and a case report. Oral Maxillofac Surg. 2018;22(4):365–8.
  • 2. Hentgen V, Grateau G, Kone-Paut I, Livneh A, Padeh S, Rozenbaum M, et al. Evidence-based recommendations for the practical management of Familial Mediterranean Fever. Semin Arthritis Rheum [Internet]. 2013;43(3):387–91. Available from: http://dx.doi.org/10.1016/j.semarthrit.2013.04.011
  • 3. Uthman I, Hajj-Ali RA, Arayssi T, Masri AF, Nasr F. Arthritis in familial Mediterranean fever. Rheumatol Int. 2001;20(4):145–8.
  • 4. Berkun Y, Eisenstein EM. Diagnostic criteria of familial Mediterranean fever. Autoimmun Rev [Internet]. 2014;13(4–5):388–90. Available from: http://dx.doi.org/10.1016/j.autrev.2014.01.045
  • 5. Berkun Y, Padeh S, Reichman B. A Single Testing of Serum Amyloid A Levels as a Tool for Diagnosis and. 2007;(6):182–8.
  • 6. Mukhin NA, Kozlovskaya L V., Bogdanova M V., Rameev V V., Moiseev S V., Simonyan AK. Predictors of AA amyloidosis in familial Mediterranean fever. Rheumatol Int. 2015;35(7):1257–61.
  • 7. Jarjour RA, Dodaki R. Arthritis patterns in familial Mediterranean fever patients and association with M694V mutation. Mol Biol Rep. 2011;38(3):2033–6.
  • 8. Migita K, Hisanaga S, Izumi Y, Kawahara C, Shigemitsu Y, Iwanaga N, et al. Protracted arthritis in a Japanese patient with familial Mediterranean fever. Mod Rheumatol. 2017;27(5):905–9.
  • 9. Ozen S, Demirkaya E, Erer B, Livneh A, Ben-Chetrit E, Giancane G, et al. EULAR recommendations for the management of familial Mediterranean fever. Ann Rheum Dis. 2016;75(4):644–51.
  • 10. El Hasbani G, Jawad A, Uthman I. Update on the management of colchicine resistant Familial Mediterranean Fever (FMF). Orphanet J Rare Dis. 2019;14(1):1–12.
  • 11. Dinarello CA, Wolff SM, Goldfinger SE, Dale DC, Alling DW. Colchicine therapy for familial mediterranean fever. A double-blind trial. N Engl J Med [Internet]. 1974 Oct 31;291(18):934–7. Available from: http://www.ncbi.nlm.nih.gov/pubmed/4606353
  • 12. Yalçinkaya F, Çakar N, Misirlioǧlu M, Tümer N, Akar N, Tekin M, et al. Genotype-phenotype correlation in a large group of Turkish patients with familial Mediterranean fever: Evidence for mutation-independent amyloidosis. Rheumatology. 2000;39(1):67–72.
  • 13. Giaglis S, Papadopoulos V, Kambas K, Doumas M, Tsironidou V, Rafail S, et al. MEFV alterations and population genetics analysis in a large cohort of Greek patients with familial Mediterranean fever. Clin Genet. 2007;71(5):458–67.
  • 14. Simon G, Marbach JJ. Familial mediterranean fever with temporomandibular joint arthritis. Pediatrics [Internet]. 1976;57(5):810–2. Available from: https://pediatrics.aappublications.org/content/57/5/810
  • 15. Cooksey DE, Girard K. Temporomandibular joint synovitis with effusion in familial Mediterranean fever. Oral Surgery, Oral Med Oral Pathol. 1979;47(2):123–6.
  • 16. Tovi F, Gatot A, Fliss D. Temporomandibular arthritis in familial mediterranean fever. Head Neck. 1992;14(6):492–5.
  • 17. Tovi F, Barmeir E, Peist M, Bar-Ziv J. Protracted temporomandibular joint arthritis in familial Mediterranean fever. J Oral Maxillofac Surg [Internet]. 1985;43(6):466–8. Available from: http://dx.doi.org/10.1016/S0278-2391(85)80059-8
  • 18. Usluer H, Bircan Z. Protracted familial mediterranean fever arthritis presenting as septic arthritis. Rheumatol Int. 2007;27(11):1083–5.
  • 19. Simon G, Marbach JJ. Familial Mediterranean fever with temporomandibular joint arthritis. Pediatrics. 1976;57(5):810–2.
  • 20. Marzook HAM, Abdel Razek AA, Yousef EA, Attia AAMM. Intra-articular injection of a mixture of hyaluronic acid and corticosteroid versus arthrocentesis in TMJ internal derangement. Vol. 121, Journal of Stomatology, Oral and Maxillofacial Surgery. 2020. p. 30–4.
  • 21. Chuong R, Piper MA. Avascular necrosis of the mandibular condyle-pathogenesis and concepts of management. Oral Surgery, Oral Med Oral Pathol. 1993;75(4):428–32.
There are 21 citations in total.

Details

Primary Language English
Subjects Health Care Administration
Journal Section Olgu Sunumu
Authors

Kıvanç Berke Ak 0000-0002-7623-0473

Muazzez Suzen 0000-0001-5121-9158

Sina Uçkan 0000-0003-1077-7342

Publication Date April 1, 2022
Acceptance Date March 10, 2022
Published in Issue Year 2022 Volume: 7 Issue: 1

Cite

Vancouver Ak KB, Suzen M, Uçkan S. Aseptic Arthritis of the Temporomandibular Joint with Severe Inflammation in an FMF Patient: A Case Report. JAMER. 2022;7(1):31-6.