The role of Adalimumab in the Treatment of Colchicine-Resistant Familial Mediterranean Fever
DOI:
https://doi.org/10.3823/1947Keywords:
Familial Mediterranean Fever, Adalimumab, Colchicine, Biologic agents, Anti tumor necrosis factorsAbstract
Familial Mediterranean fever (FMF) is an autosomal recessive inherited disorder. Articular manifestations are the second most common presentation in FMF patients after abdominal pain. Colchicine remains the gold standard of therapy for the different aspects of the disease. However, about 5-15% of patients experience resistance to colchicine. Therefore, physicians were encouraged to try different medications and treatment modalities. Biologic agents were used in several FMF patients with various clinical presentations including articular involvement. In the majority of those, there was a good clinical response with improvement in the quality of life. Adalimumab is an anti-TNF alpha agent used conventionally in inflammatory bowel disease and rheumatologic disorders. To date only five cases reported in the literature using adalimumab for severe refractory FMF.
Our case report details a young female with severe disabling colchicine-resistant FMF associated with significant articular inflammation. She was treated with adalimumab with remarkable clinical improvement and complete response
References
Grateau G, Duruoz MT. Autoinflammatory conditions: when to suspect? How to treat? Best Pract Res Clin Rheumatol. 2010;24(3):401-11 [PMID:20534373 DOI: 10.1016/j.berh.2009.12.009]
Ben-Chetrit E, Levy M. Familial Mediterranean fever. Lancet. 1998;351(9103):659-64 [PMID: 9500348 DOI: 10.1016/S0140-6736(97)09408-7]
Berkun Y, Ben-Chetrit E, Klar A. Peritoneal adhesions and intestinal obstructions in patients with familial Mediterranean fever- are they more frequent? Semin Arthritis Rheum. 2007;36(5):316-21 [PMID: 17240429 DOI: 10.1016/j.semarthrit.2006.11.002]
Akkoc N, Gul A. Familial Mediterranean fever and seronegative arthritis. Curr Rheumatol Rep 2011;13(5):388-94 [PMID: 1695514 DOI:10.1007/s11926-011-0191-9]
Sneh E, Pras M, Michaeli D, Shanin N, Gafni J. Protracted arthritis in familial Mediterranean fever. Rheumatol Rehabil. 1977;16(2):102-6 [PMID: 866903 DOI: 10.1093/rheumatology/16.2.102]
Kaushansky K, Finerman GA, Schwabe AD. Chronic destructive arthritis in familial Mediterranean fever: the predominance of hip involvement and its management. Clin Orthop Relat Res. 1981;(155): 156-161 [PMID:7226609]
Salai M, Langevitz P, Blankstein A, Zemmer D, Chechick A, Pras M, Horoszowski H. Total hip replacement in familial Mediterranean fever. Bull Hosp Jt Dis. 1993;53(1): 25-8 [PMID: 8374487]
kasifoglu T, Calisir C, Cansu DU, Korkmaz C. The frequency of sacroiliitis in familial Mediterranean fever and the role of HLA-B27 and MEFV mutations in the development of sacroiliitis. Clin Rheumatol 2009;28(1): 41-6 [PMID: 18795391 DOI: 10.1007/s10067-008-0980-3]
Langevitz P, Livneh A, Zemer D, Shemer J, Mordechai P. Seronegative spondyloarthropathy in familial Mediterranean fever. Semin Arthritis Rheum 1997;27(2): 67-72 [PMID: 9355205 DOI: 10.1016/S0049-0172(97)80007-8]
Brik R, Shinawi M, Kepten I, Berant M, Gershoni-Baruch R. Familial Mediterranean fever: clinical and genetic characterization n a mixed pediatric population of Jewish and Arab patients. Pediatrics. 1999;103(5): e70 [PMID:10224214]
Tunca M, Akar S, Onen F, Ozdogan H, Kasapcopur O, Yalcinkaya F, et al. Familial Mediterranean fever (FMF) in Turkey: results of a nationwide multicenter study. Medicine (Baltimore). 2005;84(1): 1-11 [PMID: 15643295 DOI: 10.1097/01.md.0000152370.84628.0c]
Livneh A, Langevitz P, Zemer D, Padeh S, Migdal A, Sohar E, et al. The changing face of familial Mediterranean fever. Semin Arthritis Rheum. 1996;26(3): 612-27 [PMID: 8989806]
Cerquaglia C, Diaco M, Nucera G, La Regina M, Montalto M, Manna R. Pharmacological and clinical basis of treatment of familial Mediterranean fever (FMF) with colchicine or analogues: an update. Curr Drug Targets Inflamm Allergy. 2005;4(1): 117-24 [PMID: 15720245 DOI: 10.2174/ 1568010053622984]
Van den Bosch F, Kruithof E, Baeten D, De Keyser F, Mielants H, Veys EM. Effects of a loading dose regimen of three infusions of chimeric monoclonal antiobody to tumor necrosis factor alpha (infliximab) in spondyloarhtopathy: an open pilot study. Ann Rheum Dis. 2000;59(6): 428-433 [PMID: 10834859 DOI: 10.1136/ard.59.6.428]
Van den Bosch F, Kruithof E, Baeten D, Herssens A, De Keyser F, Mielants H, et al. Randomized double-blind comparison of chimeric monoclonal antibody to tumor necrosis factor alpha (infliximab) versus placebo in active spondyloarhtropathy. Arthritis Rheum. 2002;46(3): 755-765 [PMID: 11920412]
Bilgen SA, Kilic L, Akdogan A, Kiraz S, Kalyoncu U, Karadaq O, et al. Effects of anti-tumor necrosis factor agents for Familial Mediterranean fever patients with chronic arthritis and/or sacroiliitis who were resistant to colchicine treatment. J Clin Rheumatol 2011;17(7): 358-62 [PMID: 21946459 DOI: 10.1097/RHU.0b013e31823682f5]
Ozgocmen S, Akgul O. Anti-TNF agents in familial Mediterranean fever: report of three cases and review of the literature. Mod Rheumatol 2011;21(6): 684-90 [PMID: 21567247 DOI: 10.1007/s10165-011-0463-2]
Soriano A, Verecchia E, Afeltra A, Landolfi R, Manna R. IL-1β biological treatment of familial Mediterranean fever. Clinic Rev Allerg Immunol 2013;45(1): 117-130 [PMID: 23322405 DOI: 10.1007/s12016-013-8358-y]
Ortancil O, Sanli A, SapmazBulmus P, Sarikaya S. A case of Familial Mediterranean fever which responds well to adalimumab treatment. Turk J Rheumatol 2011;26(2): 158-162 [DOI: 10.5606/tjr.2011.024]
Kaly L, Rimar D, Slobodin G, Jiries N, Ronser I, Rozenbaum M. Anti-TNF agents in intractable FMF: four cases. Pediatric Rheumatology 2013;11(S1): A14 [DOI: 10.1186/1546-0096-11-S1-A14]
Garcia-Gonzalez A, Weisman MH. The arthritis of Familial Mediterranean fever. Semin Arhtritis Rheum. 1992;22(3): 139-50 [PMID: 1295087]
Brik R, Shinawi M, Kasinetz L, Gershoni-Baruch R. The musculoskeletal manifestations of familial Mediterranean fever in children genetically diagnosed with the disease. Arthritis Rheum. 2001;44(6): 1416-9 [PMID: 11407703]
Lidar M, Scherrmann JM, Shinar Y, Chetrit A, Niel E, Gershoni-Baruch R, et al. Colchicine nonresponsiveness in familial Mediterranean fever: clinical, genetical, pharmacokinetic, and socioeconomic characterization. Semin Arthritis Rheum. 2004;33(4): 273-282 [PMID: 14978665]
Zemer D, Pras M, Sohar E, Modan M, Cabili S, Gafni J. Colchicine in the prevention and the treatment of the amyloidosis of familial Mediterranean fever. N Engl J Med. 1986;314(16): 1001-5 [PMID: 3515182]
Ben-Chetrit E, Ozdogan H. Non-response to colchicine in FMF- definition, causes and suggested solutions. Clin Exp Rheumatol. 2008;26(4suppl50): 1001-5 [PMID: 19026114]
Tufan A, Babaoglu MO, Akdogan A, Yasar U, Calquneri M, Kalyoncu U, et al. Association of drug transporter gene ABCB1 (MDR1) 3435C to T polymorphism with colchicine response in familial Mediterranean fever. J RHEUMATOL. 2007;34(7): 1540-4 [PMID: 17610314]
Chae JJ, Cho YH, Lee GS, Cheng J, Liu PP, Feigenbaum L, et al. Gain-of-function Pyrin mutations induce induce NLRP3 protein-independent interleukin-1β activation and severe autoinflammation in mice. Immunity. 2011;34(5): 755-768 [PMID: 21600797 DOI: 10.1016/j.immuni.2011.02.020]
Ryan JG, Masters SL, Booty MG, Habal N, Alexander JD, Barham BK, et al. Clinical features and functional significance of the P369S/R408Q variant in pyrin, the familial Mediterranean fever protein. Ann.Rheum.Dis. 2010;69(7): 1383-1388 [PMID: 19934105 DOI: 10.1136/ard.2009.113415]
Baykal Y, Saglam K, Yilmaz MI, Taslipinar A, Akinci SB, Inal A. Serum sIL-2r, IL-6, IL-10 and TNF-alpha level in familial Mediterranean fever patients. Clin Rheumatol. 2003;22(2): 99-101 [PMID: 12740672]
Gang N, Drenth JP. Langevitz P, Zemer D, Brezniak N, Pras M, et al. Activation of the cytokine network in familial Mediterranean fever. J Rheumatol. 1999;26(4): 890-7 [PMID: 10229412]
Silva LC, Ortigosa LC, Bernard G. Anti-TNF-alpha agents in the treatment of immune-mediated inflammatory diseases: mechanisms of action and pitfalls. Immunotherapy. 2010;2(6): 817-33 [PMID: 21091114 DOI: 10.2217/imt.10.67]
Kineret (Anakinra) in adult patients with colchicine-resistant familial Mediterranean fever. NCT01705756.
Evaluation of the safety and efficacy of canakinumab in pediatric patients with colchicine intolerant or colchicine-resistant familial Mediterranean fever (FMF) (CONTROL FMF). NCT01148797.
Efficacy and safety, of canakinumab in patients with colchicine-resistant familial Mediterranean fever. NCT01088880.
Rilonacept for treatment of familial Mediterranean fever (FMF). NCT00582907.
Interleukin-1 trap to treat autoinflammatory disease. NCT00094900.
Downloads
Published
Issue
Section
License
Authors who publish with this journal agree to the following terms:
- Authors retain copyright and grant the journal right of first publication with the work simultaneously licensed under a Creative Commons Attribution License that allows others to share the work with an acknowledgement of the work's authorship and initial publication in this journal.
- Authors are able to enter into separate, additional contractual arrangements for the non-exclusive distribution of the journal's published version of the work (e.g., post it to an institutional repository or publish it in a book), with an acknowledgement of its initial publication in this journal.
- Authors are permitted and encouraged to post their work online (e.g., in institutional repositories or on their website) prior to and during the submission process, as it can lead to productive exchanges, as well as earlier and greater citation of published work (See The Effect of Open Access and Benefits of Publishing Open Access).
Â
Â
