Abstract
The most common arthritic involvement in familial Mediterranean fever (FMF) is acute recurrent monoarthritis; however, sometimes spondyloarthropathy-like findings or typical ankylosing spondylitis may also ensue. Reported here is our favorable experience with infliximab in an FMF patient who had been resistant to colchicine and disease-modifying antirheumatic drugs (sulfasalazine and methotrexate) treatments. A 72-week follow-up of the patient yielded complete remission of the febrile abdominal episodes, and spondylitis responded well. The patient’s bilateral aseptic necrosis of the femoral head deteriorated and caused hip pain, discomfort, and disability. Overall, we believe that tumor necrosis factor (TNF) alpha has an important role in the disease pathogenesis and also that anti-TNF may represent a promising robust treatment alternative in FMF.
References
Langevitz P, Livneh A, Zemer D, Shemer J, Pras M (1997) Seronegative spondyloarthropathy in familial Mediterranean fever. Semin Arthritis Rheum 27:67–72
Majeed HA, Rawashdeh M (1997) The clinical patterns of arthritis in children with familial Mediterranean fever. Q J Med 90:37–43
Brik R, Shinawi M, Kasinetz L, Gershoni-Baruch R (2001) The musculoskeletal manifestations of familial Mediterranean fever in children genetically diagnosed with the disease. Arthritis Rheum 44:1416–1419
Younes M, Kahn MF, Meyer O (2002) Hip involvement in patients with familial Mediterranean fever. A review of ten cases. Joint Bone Spine 69:560–565
Bodur H, Ucan H, Seckin S, Seckin U, Gunduz OH (1999) Protracted familial Mediterranean fever arthritis. Rheumatol Int 19:71–73
Incel NA, Saracoglu M, Erdem HR (2003) Seronegative spondyloarthropathy of familial Mediterranean fever. Rheumatol Int 23:41–43
Kaushik P, el-Sobkie NI, Shehab D, Malaviya AN (1999) Familial Mediterranean fever with HLA B-27 positive ankylosing spondylitis in a young Armenian man. Clin Exp Rheumatol 17:387–388
Uthman I, Hajj-Ali RA, Arayssi T, Masri AF, Nasr F (2001) Arthritis in familial Mediterranean fever. Rheumatol Int 20:145–148
Brandt J, Haibel H, Cornely D, Golder W, Gonzalez J, Reddig J, Thriene W, Sieper J, Braun J (2000) Successful treatment of active ankylosing spondylitis with the anti-tumor necrosis factor alpha monoclonal antibody infliximab. Arthritis Rheum 43:1346–1352
Van den Bosch F, Kruithof E, Baeten D, De Keyser F, Mielants H, Veys EM (2000) Effects of a loading dose regimen of three infusions of chimeric monoclonal antibody to tumour necrosis factor alpha (infliximab) in spondyloarthropathy: an open pilot study. Ann Rheum Dis 59:428–433
Van Den Bosch F, Kruithof E, Baeten D, Herssens A, de Keyser F, Mielants H, Veys EM (2002) Randomized double-blind comparison of chimeric monoclonal antibody to tumor necrosis factor alpha (infliximab) versus placebo in active spondyloarthropathy. Arthritis Rheum 46:755–765
Braun J, Brandt J, Listing J et al (2002) Treatment of active ankylosing spondylitis with infliximab: a randomized controlled multicentre trial. Lancet 359:1187–1193
Garcia GA, Weisman MH (1992) The arthritis of familial Mediterranean fever. Semin Arthritis Rheum 22:139–150
Knocker DC, Malaise IG, Peetermans WE (1989) Ankylosing spondylitis. An unusual manifestation of familial Mediterranean fever. Report of a case complicated by amyloidosis and polyneuropathy. Clin Rheumatol 8:408–412
Kastner DL (2001) Intermittent and periodic arthritic syndromes. In: Koopman WJ (ed) Arthritis and allied conditions. Lippincott Williams & Wilkins, Philadelphia, pp 1400–1441
Savi M, Asinari G, Gaudiano V, Olivetti G, Neri TM (1978) Unusual immunologic findings in familial Mediterranean fever. Arch Intern Med 138:644–645
Jimenez-Balderas FJ, Martinez-Osuna P, Arellano J, Lara C, Yanez-Sanchez P, Camargo-Coronel A, Fuentes J, Bravo-Gatica C, Fraga A (1997) Does serum rheumatoid factor have an influence on the clinical picture of ankylosing spondylitis? Clin Exp Rheumatol 15:289–293
Onat AM, Ozcakar L, Ureten K, Kiraz S, Ertenl I, Calguneri M (2005) Avascular necrosis of the femoral head foreshadowing familial Mediterranean fever: apropos of three cases. Clin Rheumatol 24:155-157
Sneh E, Pras M, Michaeli D, Shanin N, Gafni J (1977) Protracted arthritis in familial Mediterranean fever. Rheumatol Rehabil 16:102–106
Shatnner A, Gurevitz A, Zemer D, Hahn T (1996) Induced TNF production in vitro as a test for familial Mediterranean fever. Q J Med 89:205–210
Drenth JP, van Deuren M, Meer JW (1995) Cytokine activation during attacks of the hyperimmunoglobulinemia D and periodic fever syndrome. Blood 85:3586–3593
Mege JL, Dilsen N, Sanguedolce V et al (1993) Overproduction of monocyte derived TNF-a, IL-6, IL-8 and increased neutrophil superoxide generation in Behcet’s disease. A comparative study with familial Mediterranean fever and healthy subjects. J Rheumatol 20:1544–1549
Baykal Y, Saglam K, Yilmaz MI, Taslipinar A, Akinci SB, Inal A (2003) Serum sIL-2r, IL-6, IL10 and TNF-a level in familial Mediterranean fever patients. Clin Rheumatol 22:99–101
Gang N, Drenth JP, Langevitz P, Zemer D, Brezniak N, Pras M, van der Meer JW, Livneh A (1999) Activation of the cytokine network in familial Mediterranean fever. J Rheumatol 26:890–897
Centola M, Wood G, Frucht DM, Galon J, Aringer M, Farrell C, Kingma DW, Horwitz ME, Mansfield E, Holland SM, O’Shea JJ, Rosenberg HF, Malech HL, Kastner DL (2000) The gene for familial Mediterranean fever, MEFV, is expressed in early leukocyte development and is regulated in response to inflammatory mediators. Blood 95:3223–3231
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Ozgocmen, S., Özçakar, L., Ardicoglu, O. et al. Familial Mediterranean fever responds well to infliximab: single case experience. Clin Rheumatol 25, 83–87 (2006). https://doi.org/10.1007/s10067-005-1122-9
Received:
Revised:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s10067-005-1122-9