Abstract
Adult-onset Still’s disease (AOSD) is an uncommon systemic inflammatory disease of unknown aetiology. Up to 80% of AOSD cases can be controlled with corticosteroids; however, reports on those unresponsive to corticosteroids, conventional disease modifying drugs and biological agents, including anti-IL1 inhibitors, are emerging. We present a case of AOSD with severe poylarthritis unresponsive to corticosteroids, methotrexate, anakinra and etanercept, but successfully stabilised with a humanized monoclonal anti-IL-6 receptor antibody, tocilizumab, administered once monthly. Thereafter, we compare our case with case reports available in the literature and suggest that for anakinra refractive AOSD patients with arthritis, tocilizumab could be the drug of choice.
References
Fautrel B (2008) Adult-onset Still’s disease. Best Pract Res Clin Rheumatol 22:773–792
Kontzias A, Efthimiou P (2008) Adult-onset Still’s disease: pathogenesis, clinical manifestations and therapeutic advances. Drugs 68:319–337
Yamaguchi M, Ohta A, Tsunematsu T, Kasukawa R, Mizushima Y, Kashiwagi H (1992) Preliminary criteria for classification of adult Still’s disease. J Rheumatol 29:424–430
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
De Bandt M, Saint-Marcoux B (2009) Tocilizumab for multi-refractory adult-onset Still’s disease. Ann Rheum Dis 68:153–154
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
Matsumoto K, Nagashima T, Takatori S, Kawahara Y, Yagi M, Iwamoto M et al (2009) Glucocorticoid and cyclosporine refractory adult onset Still’s disease successfully treated with tocilizumab. Clin Rheumatol 28:485–487
Pascual V, Allantaz F, Arce E, Punaro M, Banchereau J (2005) Role of interleukin-1 (IL-1) in the pathogenesis of systemic onset juvenile idiopathic arthritis and clinical response to IL-1 blockade. J Exp Med 201:1479–1486
De Benedetti F (2009) Targeting interleukin-6 in pediatric rheumatic diseases. Curr Opin Rheumatol 21:533–537
Disclosures
None
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Perdan-Pirkmajer, K., Praprotnik, S. & Tomšič, M. A case of refractory adult-onset Still’s disease successfully controlled with tocilizumab and a review of the literature. Clin Rheumatol 29, 1465–1467 (2010). https://doi.org/10.1007/s10067-010-1553-9
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s10067-010-1553-9