Abstract
In this prospective, non-comparative case series, four patients with severe and highly active adult-onset Still’s disease (AOSD), refractory to high doses of corticosteroids (which had been combined with methotrexate in three of them) and methotrexate were treated with infliximab (initial dose 3–5 mg/kg, continuing at intervals depending on the patient’s individual disease activity). Resolution of their symptoms, which was evident within few days after the first infusion, and a parallel rapid improvement of the acute inflammatory response indices were observed in all. Concomitant corticosteroid treatment was reduced after the first courses of treatment with infliximab, which was well tolerated, and complete disease remission was sustained during a 5–18-month follow-up period. Although further studies to confirm long-term efficacy and safety in larger numbers of patients are needed, we suggest that administration of infliximab with observation for objective improvement is the treatment of choice in cases of AOSD refractory to conventional treatment.
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Abbreviations
- AOSD:
-
Adult-onset Still’s disease
- CRP:
-
C-reactive protein
- ESR:
-
Erythrocyte sedimentation rate
- LDH:
-
Lactic dehydrogenase
- MTX:
-
Methotrexate
- NSAID:
-
Non-steroidal anti-inflammatory drugs
References
Fautrel B, Le Moel G, Saint-Marcoux B et al. (2001) Diagnostic value of ferritin and glycosylated ferritin in adult onset Still’s disease. J Rheumatol 28:322–329
Wouters JM, van de Putte LB (1986) Adult-onset Still’s disease; clinical and laboratory features, treatment and progress of 45 cases. Q J Med 61:1055–1065
Sato M, Takeda A, Honzu H, Saku N, Minato N, Kano S (1993) Adult Still’s disease with Sjogren’s syndrome successfully treated with intravenous pulse methylprednisolone and oral cyclophosphamide. Intern Med 32:730–732
Iglesias J, Sathiraju S, Marik PE (1999) Severe systemic inflammatory response syndrome with shock and ARDS resulting from Still’s disease: clinical response with high-dose pulse methylprednisolone therapy. Chest 115:1738–1740
Kumakura S, Ishikura H, Munemasa S, Adachi T, Murakawa Y, Kobayashi S (1997) Adult onset Still’s disease associated hemophagocytosis. J Rheumatol 24:1645–1648
Beckham JC, Caldwell DS, Peterson BL et al. (1992) Disease severity in rheumatoid arthritis: relationship of plasma tumor necrosis factor-alpha, soluble interleukin 2-receptor, soluble CD4/CD8 ratio, neopterin, and fibrin D-dimer to traditional severity and functional measures. J Clin Immunol 12:353–361
Mangge H, Kenzian H, Gallistl S et al. (1995) Serum cytokines in juvenile rheumatoid arthritis. Correlation with conventional inflammation parameters and clinical subtypes. Arthritis Rheum 38:211–220
Lange U, Teichmann J, Stracke H (2000) Correlation between plasma TNF-alpha, IGF-1, biochemical markers of bone metabolism, markers of inflammation/disease activity, and clinical manifestations in ankylosing spondylitis. Eur J Med Res 5:507–511
Van Deventer SJH (1997) Tumour necrosis factor and Crohn’s disease. Gut 40:443–448
Sfikakis PP (2002) Behcet’s disease: a new target for anti-tumour necrosis factor treatment. Ann Rheum Dis 61 [Suppl 2]:ii51–53
Kavanaugh A, St Clair EW, McCune WJ, Braakman T, Lipsky P (2000) Chimeric anti-tumor necrosis factor-alpha monoclonal antibody treatment of patients with rheumatoid arthritis receiving methotrexate therapy. J Rheumatol 27:841–850
Lovell DJ, Giannini EH, Reiff A et al. (2000) Etanercept in children with polyarticular juvenile rheumatoid arthritis. Pediatric Rheumatology Collaborative Study Group. N Engl J Med 342:763–769
Breban M, Vignon E, Claudepierre P et al. (2002) Efficacy of infliximab in refractory ankylosing spondylitis: results of a six-month open-label study. Rheumatology (Oxford) 41:1280–1285
Bell S, Kamm MA (2000) Antibodies to tumor necrosis factor alpha as treatment for Crohn’s disease. Lancet 355:858–860
Furst DE, Breedveld FC, Kalden JR et al. (2002) Updated consensus statement on biological agents for the treatment of rheumatoid arthritis and other rheumatic diseases (May 2002). Ann Rheum Dis 61 [Suppl 2]:ii2–7
Hoshino T, Ohta A, Yang D et al. (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
Fujii T, Nojima T, Yasuoka H et al. (2001) Cytokine and immunogenetic profiles in Japanese patients with adult Still’s disease. Association with chronic articular disease. Rheumatology (Oxford) 40:1398–1404
Yamaguchi M, Ohta A, Tsunematsu T et al. (1992) Preliminary criteria for classification of adult Still’s disease. J Rheumatol 19:424–430
Furst DE, Cush J, Kaufmann S, Siegel J, Kurth R (2002) Preliminary guidelines for diagnosing and treating tuberculosis in patients with rheumatoid arthritis in immunosuppressive trials or being treated with biological agents. Ann Rheum Dis 61[Suppl 2]:ii62–63
Reference omitted
Hanauer SB, Sandborn W, Practice Parameters Committee of the American College of Gastroenterology (2001) Management of Crohn’s disease in adults. Am J Gastroenterol 96:635–643
Cavagna L, Caporali R, Epis O, Bobbio-Pallavicini F, Montecucco C (2001) Infliximab in the treatment of adult Still’s disease refractory to conventional therapy. Clin Exp Rheumatol 19:329–332
Kraetsch HG, Antoni C, Kalden JR, Manger B (2001) Successful treatment of a small cohort of patients with adult onset of Still’s disease with infliximab: first experiences. Ann Rheum Dis 60 [Suppl 3]:iii55–57
Huffstutter JE, Sienknecht CW (2002) Treatment of resistant Adult Still’s disease with infliximab-A report of two cases. Arthritis Rheum 46[Suppl]:326
Caramaschi P, Biasi D, Carletto A, Bambara LM (2002) A case of adult onset Still’s disease treated with infliximab. Clin Exp Rheumatol 20:113
Husni ME, Maier AL, Mease PJ et al. (2002) Etanercept in the treatment of adult patients with Still’s disease. Arthritis Rheum 46:1171–1176
Van Deventer SJH (2001) Transmembrane TNF-a, induction of apoptosis, and the efficacy of TNF-targeting therapies in Crohn’s disease. Gastroenterology 121:1242–1246
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Kokkinos, A., Iliopoulos, A., Greka, P. et al. Successful treatment of refractory adult-onset Still’s disease with infliximab. A prospective, non-comparative series of four patients. Clin Rheumatol 23, 45–49 (2004). https://doi.org/10.1007/s10067-003-0775-5
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DOI: https://doi.org/10.1007/s10067-003-0775-5