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Good response to infliximab in a patient with deep vein thrombosis associated with Behçet disease

  • Case Report
  • Published:
Modern Rheumatology

Abstract

Vascular involvement is a lethal complication in Behçet disease. It is often refractory to conventional therapy such as steroids and immunosuppressants in addition to anticoagulants. We describe here successful treatment with the anti-tumor necrosis factor-alpha (anti-TNF-α) antibody, infliximab, in a patient with Behçet disease presenting with deep vein thrombosis. A 60-year-old man with Behçet disease complained of edema and pain in the lower extremities. Computed tomography revealed a thrombosis extending from the popliteal vein to the inferior vena cava at the level of the renal vein and which recurred despite combination therapy of steroid and immunosuppressants such as cyclosporine, azathioprine, and methotrexate. The patient was then administered infliximab (5 mg/kg) in weeks 0 and 2 and every 4 weeks thereafter. Clinical and laboratory findings improved after the infliximab therapy. Computed tomography of the abdomen and lower extremities showed a reduction of the thrombosis. No severe adverse events occurred during the clinical course. Although further studies are needed to confirm the efficacy and safety of its use, anti-TNF-α antibody may be worth considering as treatment for refractory venous thrombosis in patients with Behçet disease.

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References

  1. Criteria for diagnosis for Behçet’s disease. International Study Group for Behçet’s Disease. Lancet. 1990; 335:1078–80.

    Google Scholar 

  2. Sakane T, Takeno M, Suzuki N, Inaba G. Behçet’s disease. N Engl J Med. 1999;341:1284–91.

    Article  PubMed  CAS  Google Scholar 

  3. Tomasson G, Monach PA, Merkel PA. Thromboembolic disease in vasculitis. Curr Opin Rheumatol. 2009;21:41–6.

    Article  PubMed  Google Scholar 

  4. Hatemi G, Silman A, Bang D, Bodaghi B, Chamberlain AM, Gul A, Houman MH, Kötter I, Olivieri I, Salvarani C, Sfikakis PP, Siva A, Stanford MR, Stübiger N, Yurdakul S, Yazici H, EULAR Expert Committee. EULAR recommendations for the management of Behçet disease. Ann Rheum Dis. 2008;67:1656–62.

    Article  PubMed  CAS  Google Scholar 

  5. Sfikakis PP, Theodossiadis PG, Katsiari CG, Kaklamanis P, Markomichelakis NN. Effect of infliximab on sight-threatening panuveitis in Behçet’s disease. Lancet. 2001;358:295–6.

    Article  PubMed  CAS  Google Scholar 

  6. Ohno S, Nakamura S, Hori S, Shimakawa M, Kawashima H, Mochizuki M, Sugita S, Ueno S, Yoshizaki K, Inaba G. Efficacy, safety, and pharmacokinetics of multiple administration of infliximab in Behçet’s disease with refractory uveoretinitis. J Rheumatol. 2004;31:1362–8.

    PubMed  CAS  Google Scholar 

  7. Tugal-Tutkun I, Mudun A, Urgancioglu M, Kamali S, Kasapoglu E, Inanc M, Gül A. Efficacy of infliximab in the treatment of uveitis that is resistant to treatment with the combination of azathioprine, cyclosporine, and corticosteroids in Behçet’s disease: an open-label trial. Arthritis Rheum. 2005;52:2478–84.

    Article  PubMed  CAS  Google Scholar 

  8. Kikuchi H, Aramaki K, Hirohata S. Effect of infliximab in progressive neuro-Behçet’s syndrome. J Neurol Sci. 2008;272:99–105.

    Article  PubMed  CAS  Google Scholar 

  9. Lie JT. Vascular involvement in Behçet’s disease: arterial and venous and vessels of all sizes. J Rheumatol. 1992;19:341–3.

    PubMed  CAS  Google Scholar 

  10. Koç Y, Güllü I, Akpek G, Akpolat T, Kansu E, Kiraz S, Batman F, Kansu T, Balkanci F, Akkaya S, et al. Vascular involvement in Behçet disease. J Rheumatol. 1992;19:402–10.

    PubMed  Google Scholar 

  11. Boumpas DT, Austin HA 3rd, Vaughan EM, Yarboro CH, Klippel JH, Balow JE. Risk for sustained amenorrhea in patients with systemic lupus erythematosus receiving intermittent pulse cyclophosphamide therapy. Ann Intern Med. 1993;119:366–9.

    PubMed  CAS  Google Scholar 

  12. Boffetta P, Kaldor JM. Secondary malignancies following cancer chemotherapy. Acta Oncol. 1994;33:591–8.

    Article  PubMed  CAS  Google Scholar 

  13. Masala A, Faedda R, Alagna S, Satta A, Chiarelli G, Rovasio PP, Ivaldi R, Taras MS, Lai E, Bartoli E. Use of testosterone to prevent cyclophosphamide-induced azoospermia. Ann Intern Med. 1997;126:292–5.

    PubMed  CAS  Google Scholar 

  14. Iwata S, Saito K, Yamaoka K, Tsujimura S, Nawata M, Suzuki K, Tanaka Y. Effects of anti-TNF-alpha antibody infliximab in refractory entero-Behcet’s disease. Rheumatology (Oxford). 2009;48:1012–3.

    Article  CAS  Google Scholar 

  15. Sfikakis PP, Markomichelakis N, Alpsoy E, Assaad-Khalil S, Bodaghi B, Gul A, Ohno S, Pipitone N, Schirmer M, Stanford M, Wechsler B, Zouboulis C, Kaklamanis P, Yazici H. Anti-TNF therapy in the management of Behcet’s disease—review and basis for recommendations. Rheumatology (Oxford). 2007;46:736–41.

    Article  CAS  Google Scholar 

  16. Lee SW, Lee SY, Kim KN, Jung JK, Chung WT. Adalimumab treatment for life threatening pulmonary artery aneurysm in Behçet disease: a case report. Clin Rheumatol. 2010;29:91–3.

    Article  PubMed  Google Scholar 

  17. Baki K, Villiger PM, Jenni D, Meyer T, Beer JH. Behçet’s disease with life-threatening haemoptoe and pulmonary aneurysms: complete remission after infliximab treatment. Ann Rheum Dis. 2006;65:1531–2.

    Article  PubMed  CAS  Google Scholar 

  18. Endo LM, Rowe SM, Romp RL, Buckmaster MA, Atkinson TP. Pulmonary aneurysms and intracardiac thrombi due to Behçet’s disease in an African-American adolescent with oculocutaneous albinism. Clin Rheumatol. 2007;26:1537–9.

    Article  PubMed  Google Scholar 

  19. Seyahi E, Hamuryudan V, Hatemi G, Melikoglu M, Celik S, Fresko I, Yurdakul S, Yazici H. Infliximab in the treatment of hepatic vein thrombosis (Budd–Chiari syndrome) in three patients with Behcet’s syndrome. Rheumatology (Oxford). 2007;46:1213–4.

    Article  CAS  Google Scholar 

  20. Mege JL, Dilsen N, Sanguedolce V, Gul A, Bonqrand P, Roux H, Ocal L, Inanç M, Capo C. Over production of monocyte derived tumor necrosis factor alpha, interleukin IL-6, IL-8 and increased neutrophil superoxide generation in Behçet’s disease. A comparative study with familial Mediterranean fever and healthy subjects. J Rheumatol. 1993;20:1544–9.

    PubMed  CAS  Google Scholar 

  21. Durmazlar SP, Ulkar GB, Eskioglu F, Tatlican S, Mert A, Akgul A. Significance of serum interleukin-8 levels in patients with Behcet’s disease: high levels may indicate vascular involvement. Int J Dermatol. 2009;48:259–64.

    Article  PubMed  CAS  Google Scholar 

  22. Aki T, Karincaoglu Y, Seyhan M, Batcioglu K. Serum substance P and calcitonin gene-related peptide levels in Behçet’s disease and their association with disease activity. Clin Exp Dermatol. 2006;31:583–7.

    Article  PubMed  CAS  Google Scholar 

  23. Seyahi E, Yurdakul S. Behçet’s syndrome and thrombosis. Mediterr J Hematol Infect Dis. 2011;3:e2011026.

    Article  PubMed  Google Scholar 

  24. Gur-Toy G, Lenk N, Yalcin B, Aksaray S, Alli N. Serum interleukin-8 as a serologic marker of activity in Behçet disease. Int J Dermatol. 2005;44:657–60.

    Article  PubMed  CAS  Google Scholar 

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Authors and Affiliations

  1. First Department of Internal Medicine, Osaka Medical College, Daigaku-Machi 2-7, Takatsuki, Osaka, 569-8686, Japan

    Shuzo Yoshida, Tohru Takeuchi, Ayaka Yoshikawa, Takuro Ozaki, Yohei Fujiki, Kenichiro Hata, Shigeki Makino & Toshiaki Hanafusa

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  1. Shuzo Yoshida
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  2. Tohru Takeuchi
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  3. Ayaka Yoshikawa
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  4. Takuro Ozaki
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  5. Yohei Fujiki
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  6. Kenichiro Hata
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  7. Shigeki Makino
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  8. Toshiaki Hanafusa
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Correspondence to Shuzo Yoshida.

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Yoshida, S., Takeuchi, T., Yoshikawa, A. et al. Good response to infliximab in a patient with deep vein thrombosis associated with Behçet disease. Mod Rheumatol 22, 791–795 (2012). https://doi.org/10.1007/s10165-011-0585-6

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  • DOI: https://doi.org/10.1007/s10165-011-0585-6

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