TY - JOUR T1 - Longstanding Remission of Adult Onset Still’s Disease Under Imatinib Therapy in a Patient with Chronic Myelogenous Leukemia JF - The Journal of Rheumatology JO - J Rheumatol SP - 1349 LP - 1350 DO - 10.3899/jrheum.081159 VL - 36 IS - 6 AU - YOJI NAGASAKI AU - TOSHIHIRO MIYAMOTO AU - HIDEHO HENZAN AU - KOJI NAGAFUJI AU - MINE HARADA AU - KOICHI AKASHI Y1 - 2009/06/01 UR - http://www.jrheum.org/content/36/6/1349.abstract N2 - To the Editor:Imatinib mesylate is a potent and selective inhibitor of tyrosine kinases such as Bcr-Abl, platelet-derived growth factor (PDGF) receptor, and c-Kit, and it is widely used to treat chronic myeloid leukemia (CML) and c-Kit-positive gastrointestinal stromal tumors. Imatinib also exerts potent immunomodulatory effects in vitro and in vivo1. Recent reports described the efficacy of imatinib in treating patients with various autoimmune and inflammatory diseases such as rheumatoid arthritis (RA), systemic scleroderma, pulmonary arterial hypertension, and lupus nephritis2–5.We describe a patient with steroid-resistant adult onset of Stills disease (AOSD), who incidentally developed CML. Treatment with imatinib induced hematological remission of CML as well as improvement of symptoms of AOSD. Immunosuppressants were tapered and discontinued 2 years after the initial imatinib treatment. The patient has maintained remission of AOSD for 5 years under imatinib treatment.In August 2000, a 25-year-old Japanese man was admitted to the Department of GeneralMedicine, Kyushu University Hospital, due to fever of unknown origin. He exhibited symptoms of pharyngitis, arthralgia, typical rash, leukocytosis, high level of C-reactive protein (CRP; 33.0 mg/dl), and hyperferritinemia (19,777 ng/ml). A diagnosis of AOSD was made based on his symptoms and these results. He was treated with prednisolone (PSL; 1 mg/kg daily). … Address reprint requests to Dr. T. Miyamoto, Center for Cellular and Molecular Medicine, Kyushu University Hospital, 3-1-1 Maidashi, Higashi-ku, Fukuoka, Japan 812-8582. E-mail: toshmiya{at}intmed1.med.kyushu-u.ac.jp ER -