Skip to main content

Advertisement

Log in

Infliximab therapy for familial Mediterranean fever-related amyloidosis: case series with long term follow-up

  • Case Based Review
  • Published:
Clinical Rheumatology Aims and scope Submit manuscript

Abstract

Familial Mediterranean fever (FMF) is an autosomal recessive disease characterized by recurrent self-limited attacks of fever and polyserositis. Reactive amyloid A amyloidosis is the most devastating complication of FMF, and amyloidosis continues to occur in the colchicine era in untreated and noncompliant patients. Unfortunately, there is no proven effective treatment for established amyloidosis. In this report, we present four FMF-related amyloidosis patients that were treated with long term infliximab therapy with the longest duration of follow-up, together with the literature review. Infliximab was very effective in controlling gastrointestinal system findings and protracted arthritis, and it also had a favorable impact on the clinical findings of nephrotic syndrome in these patients. In conclusion, by controlling debilitating complaints of amyloidosis with infliximab, quality of life increases in these patients, and they get rid of recurrent hospitalizations and return to school or work.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Similar content being viewed by others

References

  1. Lidar M, Livneh A (2007) Familial Mediterranean fever: clinical, molecular and management advancements. Neth J Med 65:318–324

    PubMed  CAS  Google Scholar 

  2. Mamou H, Cattan R (1952) La maladie periodique (sur 14 cas personnels dont 8 compliques de nephropathies). Sem Hop Paris 28:1062–1070

    PubMed  CAS  Google Scholar 

  3. Goldfinger SE (1972) Colchicine for familial Mediterranean fever. N Engl J Med 287:1302

    PubMed  CAS  Google Scholar 

  4. Özkan E, Okur Ö, Ekmekçi A, Özcan R, Tağ T (1972) A new approach to the treatment of periodic fever. Med Bull Istanbul 5:44–49

    Google Scholar 

  5. Tunca M, Akar S, Onen F, Ozdogan H, Kasapcopur O, Yalcinkaya F et al (2005) Familial Mediterranean fever (FMF) in Turkey. Results of nationwide multicenter study. Medicine 84:1–11

    Article  PubMed  Google Scholar 

  6. Touitou I, Sarkisian T, Medlej-Hashim M, Tunca M, Livneh A, Cattan D et al (2007) Country as the primary risk factor for renal amyloidosis in familial Mediterranean fever. Arthritis Rheum 56:1706–1712

    Article  PubMed  Google Scholar 

  7. Cakar N, Yalçinkaya F, Ozkaya N, Tekin M, Akar N, Koçak H et al (2001) Familial Mediterranean fever (FMF)-associated amyloidosis in childhood. Clinical features, course and outcome. Clin Exp Rheumatol 5(Suppl 24):S63–S67

    Google Scholar 

  8. Sayarlıoğlu H, Erkoç R, Sayarlıoğlu M, Dogan E, Soyoral Y (2006) Successful treatment of nephrotic syndrome due to FMF amyloidosis with azathioprine: report of three Turkish case. Rheumatol Int 27:197–199

    Article  PubMed  Google Scholar 

  9. Metyas S, Arkfeld DG, Forrester DM, Ehresmann GR (2004) Infliximab treatment of familial Mediterranean fever and its effect on secondary AA amyloidosis. J Clin Rheumatol 10:134–137

    Article  PubMed  Google Scholar 

  10. Yüksel S, Yalçınkaya F, Acar B, Özçakar ZB, Öztürk B, Ekim M (2006) Clinical improvement with infliximab in a child with amyloidosis secondary to familial Mediterranean fever. Rheumatology 45:1307–1321

    Article  PubMed  Google Scholar 

  11. Bodur H, Seçkin Ü, Eser F, Ergül G, Seçkin S (2008) Coexistence of familial Mediterranean fever and psoriasis in a patient with seronegative spondyloarthropathy. Rheumatol Int 29:107–110

    Article  PubMed  Google Scholar 

  12. Erten S, Erten SF, Altunoğlu A (2012) Successful treatment with anti-tumor necrosis factor (anti-TNF)-alpha of proteinuria in a patient with familial Mediterranean fever (FMF) resistant to colchicine:anti-TNF drugs and FMF. Rheumatol Int 32:1095–1097

    Article  PubMed  CAS  Google Scholar 

  13. Özgöçmen S, Akgül Ö (2011) Anti-TNF agents in familial Mediterranean fever: report of three cases and review of the literature. Mod Rheumatol 21:684–690

    Article  PubMed  Google Scholar 

  14. Dember LM, Hawkins PN, Hazenberg BPC, Gorevic PD, Merlini G, Butrimiene I et al (2007) Eprodisate for the treatment of renal disease in AA amyloidosis. N Eng J Med 356:2349–2360

    Article  CAS  Google Scholar 

  15. Gafni J, Ravid M, Sohar E (1968) The role of amyloidosis in familial Mediterranean fever. A population study. Isr J Med 4:995–999

    CAS  Google Scholar 

  16. Fl Ö, Kaplaman E, Zileli S (1971) Familial Mediterranean fever in Turkey. A report of twenty cases. Am J Med 50:336–339

    Article  Google Scholar 

  17. Falk RH, Comenzo RL, Skinner M (1997) The systemic amyloidoses. N Eng J Med 337:898–909

    Article  CAS  Google Scholar 

  18. Urieli-Shoval S, Linke RP, Matzner Y (2000) Expression and function of serum amyloid A, a major acute-phase protein, in normal and disease states. Curr Opin Hematol 7:64–69

    Article  PubMed  CAS  Google Scholar 

  19. Levalampi T, Honkanen V, Lahdenne P, Nieminen R, Hakala M, Moilanen E (2007) Effects of infliximab on cytokines, myeloperoxidase, and soluble adhesion molecules in patients with juvenile idiopathic arthritis. Scand J Rheumatol 36:189–193

    Article  PubMed  CAS  Google Scholar 

  20. Özgöçmen S, Özçakar L, Ardıçoğlu Ö, Kocakoç E, Kaya A, Kiriş A (2006) Familial Mediterranean fever responds well to infliximab:single case experience. Clin Rheumatol 25:83–87

    Article  PubMed  Google Scholar 

  21. Elkayam O, Hawkins PN, Lachmann H, Yaron M, Caspi D (2002) Rapid and complete resolution of proteinuria due to renal amyloidosis in a patient with rheumatoid arthritis treated with infliximab. Arthritis & Rheumatism 46:2571–2573

    Article  Google Scholar 

  22. Pettersson T, Konttinen YT, Maury CPJ (2008) Treatment strategies for amyloid A amyloidosis. Expert Opin Pharmacother 9:2117–2128

    Article  PubMed  CAS  Google Scholar 

  23. Dember LM (2006) Amyloidosis associated kidney disease. J Am Soc Nephrol 17:3458–3471

    Article  PubMed  CAS  Google Scholar 

  24. Kuroda T, Wada Y, Kobayashi D, Murakami S, Sakai T, Hirose S et al (2009) Effective anti-TNF-α therapy can induce rapid resolution and sustained decrease of gastroduedenal mucosal amyloid deposits in reactive amyloidosis associated with rheumatoid arthritis. J Rheumatol 36:2409–2415

    Article  PubMed  CAS  Google Scholar 

Download references

Disclosures

None.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Fatoş Yalçınkaya.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Özçakar, Z.B., Yüksel, S., Ekim, M. et al. Infliximab therapy for familial Mediterranean fever-related amyloidosis: case series with long term follow-up. Clin Rheumatol 31, 1267–1271 (2012). https://doi.org/10.1007/s10067-012-2009-1

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10067-012-2009-1

Keywords

Navigation