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A case report of a patient with refractory adult-onset Still’s disease who was successfully treated with tocilizumab over 6 years

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Modern Rheumatology

Abstract

Interleukin-6 overproduction is pathologically involved in adult onset Still’s disease (AOSD). We successfully treated a man with refractory AOSD utilizing tocilizumab. Tocilizumab was discontinued after 15 doses due to intestinal bleeding, but the efficacy was sustained over 21 months. Tocilizumab was readministered safely upon recurrence and showed similar efficacy over 6 years. Corticosteroid and NSAIDs could be discontinued and intestinal bleeding was no more observed. Tocilizumab can be a therapeutic option for AOSD.

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References

  1. Bywaters EGL. Still’s disease in the adult. Ann Rheum Dis. 1971;30:121–33.

    Article  PubMed  CAS  Google Scholar 

  2. Yamaguchi M, Ohta A, Tsunematsu T, Kasukawa R, Mizushima Y, Kashiwagi H, et al. Preliminary criteria for classification of adult Still’s disease. J Rheumatol. 1992;29:424–30.

    Google Scholar 

  3. Chen DY, Lan JL, Lin FJ, Hsieh TY. Proinflammatory cytokine profiles in sera and pathological tissues of patients with active untreated adult onset Still’s disease. J Rheumatol. 2004;31:2189–98.

    PubMed  CAS  Google Scholar 

  4. Scheinberg MA, Chapira E, Fernandes ML, Hubscher O. Interleukin 6: a possible marker of disease activity in adult onset Still’s disease. Clin Exp Rheumatol 1992; 653–5.

  5. Nishimoto N, Kishimoto T, Yoshizaki K. Anti-interleukin 6 receptor antibody treatment in rheumatoid arthritis. Ann Rheum Dis. 2000;59(suppl 1):i21–7.

    Article  PubMed  CAS  Google Scholar 

  6. Sato K, Tsuchiya M, Saldanha J, Koishihara Y, Ohsugi Y, Kishimoto T, et al. Reshaping a human antibody to inhibit the interleukin-6-dependent tumor cell growth. Cancer Res. 1993;53:851–6.

    PubMed  CAS  Google Scholar 

  7. Yokota S, Miyamae T, Imagawa T, Iwata N, Katakura S, Mori M, et al. Therapeutic efficacy of humanized recombinant anti-interleukin-6 receptor antibody in children with systemic-onset juvenile idiopathic arthritis. Arthritis Rheum. 2005;52:818–25.

    Article  PubMed  CAS  Google Scholar 

  8. Woo P, Wilkinson N, Prieur AM, Southwood T, Leone V, Livermore P, et al. Open-label phase II trial of single, ascending doses of MRA in Caucasian children with severe systemic juvenile idiopathic arthritis: proof of principle of the efficacy of IL-6 receptor blockade in this type of arthritis and demonstration of prolonged clinical improvement. Arthritis Res Ther. 2005;7:R1281–8.

    Article  PubMed  CAS  Google Scholar 

  9. Yokota S, Imagawa T, Mori M, Miyamae T, Aihara Y, Takei S, et al. Efficacy and safety of tocilizumab in patients with systemic-onset juvenile idiopathic arthritis: a randomized, double-blind, placebo-controlled, withdrawal phase III trial. Lancet. 2008;371:998–1006.

    Article  PubMed  CAS  Google Scholar 

  10. Iwamoto M, Nara H, Hirata D, Minota S, Nishimoto N, Yoshizaki K. Humanized monoclonal anti-interleukin 6 receptor antibody for treatment of intractable adult-onset Still’s disease. Arthritis Rheum. 2002;46:3388–9.

    Article  PubMed  CAS  Google Scholar 

  11. Nishimoto N, Hashimoto J, Miyasaka N, Yamamoto K, Kawai S, Takeuchi T, et al. Study of active controlled monotherapy used for rheumatoid arthritis, an IL-6 inhibitor (SAMURAI): evidence of clinical and radiographic benefit from an x ray reader-blinded randomized controlled trial of tocilizumab. Ann Rheum Dis. 2007;66:1162–7.

    Article  PubMed  CAS  Google Scholar 

  12. Information for proper use of infliximab (in Japanese) by Tanabe Seiyaku. http://medical.tanabe.co.jp/mstaff/remicade/index_remicade.shtml.

  13. Saiki O, Uda H, Nshimoto N, Miwa T, Mima T, Ogawara T, et al. Adult Still’s disease reflects a Th2 rather than a Th1 cytokine profile. Clin Immunol. 2004;112:120–5.

    Article  PubMed  CAS  Google Scholar 

  14. Olee T, Hashimoto S, Quach J, Lotz M. IL-18 is produced by articular chondrocytes and induces proinflammatory and catabolic responses. J Immunol. 1999;162:1096–100.

    PubMed  CAS  Google Scholar 

  15. Jablonska E, Jablonski J. Effect of IL-18 on the release of IL-6 and its soluble receptors: sIL-6Rα and sgp 130 by human neutrophils. Immunological Investigations. 2002;31:159–67.

    Article  PubMed  CAS  Google Scholar 

  16. Yamamura M, Kawashima M, Taniai M, Yamauchi H, Tanimoto T, Kurimoto M, et al. Interferon-γ-inducing activity of interleukin-18 in the joint with rheumatoid arthritis. Arthritis Rheum. 2001;44:275–85.

    Article  PubMed  CAS  Google Scholar 

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Acknowledgments

We thank Dr. Ichiro Kawase and Dr. Toshio Tanaka for clinical support. This work was supported by Chugai Pharmaceutical Co., Ltd. N. Nishimoto and T. Mima received a consulting fee from Chugai Pharmaceutical Co., Ltd. as medical advisors.

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Authors and Affiliations

  1. Laboratory of Immune Regulation, Graduate School of Frontier Biosciences, Osaka University, 1-3 Yamada-oka, Suita, Osaka, 565-0871, Japan

    Hideko Nakahara, Toru Mima, Naoko Yoshio-Hoshino & Norihiro Nishimoto

  2. NTT West Osaka Hospital, 2-6-40 Karasugatsuji, Tennoji-ku, Osaka, Osaka, 543-8922, Japan

    Hideko Nakahara

  3. Division of Rheumatology, National Hospital Organization, Osaka Minami Medical Center, 2-1 Kidohigashi-cho, Kawachinagano, Osaka, 586-8521, Japan

    Masato Matsushita

  4. Department of Orthopaedics, Osaka University Graduate School of Medicine, 2-2 Yamada-oka, Suita, Osaka, 565-0871, Japan

    Jun Hashimoto

Authors
  1. Hideko Nakahara
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  2. Toru Mima
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  3. Naoko Yoshio-Hoshino
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  4. Masato Matsushita
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  5. Jun Hashimoto
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  6. Norihiro Nishimoto
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Correspondence to Norihiro Nishimoto.

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Nakahara, H., Mima, T., Yoshio-Hoshino, N. et al. A case report of a patient with refractory adult-onset Still’s disease who was successfully treated with tocilizumab over 6 years. Mod Rheumatol 19, 69–72 (2009). https://doi.org/10.1007/s10165-008-0116-2

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  • DOI: https://doi.org/10.1007/s10165-008-0116-2

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