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Case report: successful use of short-term add-on tocilizumab for multirefractory systemic flare of adult-onset Still’s disease

Clinical Rheumatology volume 32, pages 103–106 (2013)Cite this article

Abstract

We report on a 64-year-old woman with multirefractory flare of adult-onset Still’s disease successfully treated with six-month course of add-on anti-interleukin 6 receptor antibody, tocilizumab. Before administration of tocilizumab, the combination therapy with 80 mg/day of prednisolone and cyclosporine or tacrolimus for five weeks, two courses of pulse methylprednisolone, and high-dose intravenous immunoglobulin could not control the disease. Add-on tocilizumab dramatically improved her disease state and enabled tapering of corticosteroid and tacrolimus. Furthermore remission has been maintained on low-dose corticosteroid and tacrolimus after withdrawal of tocilizumab. This case report suggests that short-term add-on tocilizumab might be a useful therapeutic option for patients with multirefractory flare of polycyclic systemic adult-onset Still’s disease.

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References

  1. Efthimiou P, Paik PK, Bielory L (2006) Diagnosis and management of adult onset Still’s disease. Ann Rheum Dis 65:564–572

    PubMed  Article  CAS  Google Scholar 

  2. Cush JJ, Medsger TA Jr, Christy WC, Herbert DC, Cooperstein LA (1987) Adult-onset Still’s disease. Clinical course and outcome. Arthritis Rheum 30:186–194

    PubMed  Article  CAS  Google Scholar 

  3. Félix FH, Leal LK, Fontenele JB (2009) Cloak and dagger: the case for adult onset still disease and hemophagocytic lymphohistiocytosis. Rheumatol Int 29:973–974

    PubMed  Article  Google Scholar 

  4. Yamaguchi M, Ohta A, Tsunematsu T, Kasukawa R, Mizushima Y, Kashiwagi H, Kashiwazaki S, Tanimoto K, Matsumoto Y, Ota T et al (1992) Preliminary criteria for classification of adult Still’s disease. J Rheumatol 19:424–430

    PubMed  CAS  Google Scholar 

  5. Efthimiou P, Georgy S (2006) Pathogenesis and management of adult-onset Still’s disease. Semin Arthritis Rheum 36:144–152

    PubMed  Article  CAS  Google Scholar 

  6. Yokota S, Imagawa T, Mori M, Miyamae T, Aihara Y, Takei S, Iwata N, Umebayashi H, Murata T, Miyoshi M, Tomiita M, Nishimoto N, Kishimoto T (2008) Efficacy and safety of tocilizumab in patients with systemic-onset juvenile idiopathic arthritis: a randomised, double-blind, placebo-controlled, withdrawal phase III trial. Lancet 371:998–1006

    PubMed  Article  CAS  Google Scholar 

  7. Iwamoto M, Nara H, Hirata D, Minota S, Nishimoto N, Yoshizaki K (2002) Humanized monoclonal anti-interleukin-6 receptor antibody for treatment of intractable adult-onset Still’s disease. Arthritis Rheum 46:3388–3389

    PubMed  Article  CAS  Google Scholar 

  8. Nakahara H, Mima T, Yoshio-Hoshino N, Matsushita M, Hashimoto J, Nishimoto N (2009) 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

    PubMed  Article  Google Scholar 

  9. Matsumoto K, Nagashima T, Takatori S, Kawahara Y, Yagi M, Iwamoto M, Okazaki H, Minota S (2009) Glucocorticoid and cyclosporine refractory adult onset Still’s disease successfully treated with tocilizumab. Clin Rheumatol 28:485–487

    PubMed  Article  Google Scholar 

  10. De Bandt M, Saint-Marcoux B (2009) Tocilizumab for multirefractory adult-onset Still’s disease. Ann Rheum Dis 68:153–154

    PubMed  Article  Google Scholar 

  11. Chen DY, Lan JL, Lin FJ, Hsieh TY (2004) Proinflammatory cytokine profiles in sera and pathological tissues of patients with active untreated adult onset Still’s disease. J Rheumatol 31:2189–2198

    PubMed  CAS  Google Scholar 

  12. Hoshino T, Ohta A, Yang D, Kawamoto M, Kikuchi M, Inoue Y, Kamizono S, Ota T, Itoh K, Oizumi K (1998) Elevated serum interleukin 6, interferon-gamma, and tumor necrosis factor-alpha levels in patients with adult Still’s disease. J Rheumatol 25:396–398

    PubMed  CAS  Google Scholar 

  13. Nishimoto N, Terao K, Mima T, Nakahara H, Takagi N, Kakehi T (2008) Mechanisms and pathologic significances in increase in serum interleukin-6 (IL-6) and soluble IL-6 receptor after administration of an anti-IL-6 receptor antibody, tocilizumab, in patients with rheumatoid arthritis and Castleman disease. Blood 112:3959–3964

    PubMed  Article  CAS  Google Scholar 

  14. Comoli P, Labirio M, Basso S, Baldanti F, Grossi P, Furione M, Viganò M, Fiocchi R, Rossi G, Ginevri F, Gridelli B, Moretta A, Montagna D, Locatelli F, Gerna G, Maccario R (2002) Infusion of autologous Epstein–Barr virus (EBV)-specific cytotoxic T cells for prevention of EBV-related lymphoproliferative disorder in solid organ transplant recipients with evidence of active virus replication. Blood 99:2592–2598

    PubMed  Article  CAS  Google Scholar 

  15. Davis JE, Sherritt MA, Bharadwaj M, Morrison LE, Elliott SL, Kear LM, Maddicks-Law J, Kotsimbos T, Gill D, Malouf M, Falk MC, Khanna R, Moss DJ (2004) Determining virological, serological and immunological parameters of EBV infection in the development of PTLD. Int Immunol 16:983–989

    PubMed  Article  CAS  Google Scholar 

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Affiliations

  1. Division of Rheumatology, Nagoya City University Hospital and the Department of Medical Oncology and Immunology, Nagoya City University Graduate School of Medical Sciences, Nagoya, Aichi, 467-8601, Japan

    Taio Naniwa, Rei Ito, Maiko Watanabe, Yoshihito Hayami, Shinji Maeda, Kaneshige Sasaki & Shiho Iwagaitsu

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  1. Taio Naniwa
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  2. Rei Ito
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  3. Maiko Watanabe
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  4. Yoshihito Hayami
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  5. Shinji Maeda
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  6. Kaneshige Sasaki
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  7. Shiho Iwagaitsu
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Correspondence to Taio Naniwa.

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Naniwa, T., Ito, R., Watanabe, M. et al. Case report: successful use of short-term add-on tocilizumab for multirefractory systemic flare of adult-onset Still’s disease. Clin Rheumatol 32, 103–106 (2013). https://doi.org/10.1007/s10067-010-1562-8

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  • DOI: https://doi.org/10.1007/s10067-010-1562-8

Keywords

  • adult-onset Still’s disease
  • interleukin 6
  • interleukin 18
  • tocilizumab