Abstract
Adult onset Still’s disease (AOSD) is an uncommon disorder of unknown cause. The clinical symptoms of AOSD are a spiking fever, a typical rash, arthralgia or arthritis, sore throat, lymphadenopathy, and splenomegaly. Pleuropulmonary and cardiac involvement are rare. We report a patient with a two-year history of AOSD with myocarditis refractory to cyclosporine and glucocorticoid. Significant congestive heart failure due to left ventricle dysfunction and hyperferritinemia developed during the hospital course. After therapy with etanercept, the patient’s clinical manifestations recovered and she regained normal left ventricular systolic function.
References
Pouchot J, Sampalis JS, Beaudet F, Carette S, Decary F, Salusinsky-Sternbach M et al (1991) Adult Still’s disease: manifestation, disease course and outcome in 62 patients. Medicine 70:118–136
Cush JJ, Medsger TA Jr, Christy WC, Herbert DC, Cooperstein LA (1987) Adult-onset Still’s disease. Arthritis Rheum 30:186–194
Bywaters EG (1971) Still’s disease in the adult. Ann Rheum Dis 30:121–133
Magadur-Joly G, Billaud E, Barries JH, Pennec YL, Masson C, Renou P, Prost A (1995) Epidemiology of adult Still’s disease: estimate of the incidence by a retrospective study in west France. Ann Rheum Dis 54:587–590
Sampalis JS, Medsger TA Jr, Fries JF, Yeadon C, Senecal JL, Myhal D et al (1996) Risk factors for adult Still’s disease. J Rheumatol 23:2049–2054
Wouters JM, Reekers P, Van de Putte LB (1986) Adult-onset Still’s disease: Disease course and HLA associations. Arthritis Rheum 29:415–418
Choi JH, Suh CH, Lee YM, Suh YJ, Lee SK, Kim SS, Nahm DH, Park HS (2003) Serum cytokine profiles in patients with adult onset Still’s disease. J Rheumatol 30:2422–2427
Chen DY, Hsieh TY, Hsieh CW, Lin FJ, Lan JL (2007) Increased apoptosis of peripheral blood lymphocytes and its association with interleukin-18 in patients with active untreated adult-onset Still’s disease. Arthritis Rheum 57:1530–1538
Assy N, Schlesinger S, Hussein O (2001) Acute hepatitis with extremely ferritin levels and Still’s disease. J Hepatol 35:830–831
Akritidis N, Giannakaki Y, Sakkas L (1997) Very high serum ferritin levels in adult onset Still’s disease. Br J Rheumatol 36:608–609
Schiller D, Mittermayer H (1998) Hyperferritinemia as a marker f Still’s disease. Clin Infect Dis 26:534
Watson JP, Bramble MG, Ghosh SK (1998) Massively elevated serum ferritin in an ill man with abnormal liver function tests. Postgrad Med J 74:619–620
Bank I, Marboe CC, Redberg RF, Jacobs J (1985) Myocarditis in adult Still’s disease. Arthritis Rheum 28:452–454
Yoo WH (2008) Adult onset Still’s disease flared with pericardial effusion. Rheumatol Int 28:285–287
Efthimiou P, Paik PK, Bielory L (2006) Diagnosis and management of adult onset Still’s disease. Ann Rheum Dis 65:564–572
Husni ME, Maier AL, Mease PJ, Overman SS, Fraser P, Gravallese EM, Weinblatt ME (2002) Etanercept in the treatment of adult patients with Still’s disease. Arthritis Rheum 46:1171–1176
Fautrel B, Sibilia J, Mariette X, Combe B (2005) Tumour necrosis factor α blocking agents in refaractory adult Still’s disease: an observational study of 20 cases. Ann Rheum Dis 64:262–266
Kristensen LE, Bartosik I (2006) Myocarditis in adult-onset Still’s disease despite significant immunosuppressive therapy. Scand J Rheumatol 35:330–331
Kuek A, Weerakoon A, Ahmed K, Ostor AJK (2007) Adult-onset Still’s disease and myocarditis: successful treatment with intravenous immunoglobulin and maintenance of remission with etanercept. Rheumatology 46:1043–1044
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Yang, DH., Chang, DM., Lai, JH. et al. Etanercept as a rescue agent in patient with adult onset Still’s disease complicated with congestive heart failure. Rheumatol Int 29, 95–98 (2008). https://doi.org/10.1007/s00296-008-0619-3
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00296-008-0619-3