578 zyxwvutsrqp ARTHRITIS zyxw & RHEUMATISM Vol. 40, No. zyxwv 3, March 1997, pp 578-582 zy 0 1997, American College of Rheumatology TENOSYNOVIAL NODULOSIS IN A PATIENT INFECTED WITH HUMAN T CELL LYMPHOTROPIC VIRUS I TOMOKO HASUNUMA, TADANOBU MORIMOTO, TRAN THI MINH HOA, ULF MULLER-LADNER, HIROYUKI AONO, RYOKEI OGAWA, STEFFEN GAY, and KUSUKI NISHIOKA We describe a 45-year-old man who presented with multiple nodules along the tendons of the scapular region, the elbows, wrists, forearms, thighs, and ankles. The patient was a carrier of human T cell lymphotropic virus I (HTLV-I), which was probably transmitted from his mother; his mother also had polyarthritis. His- topathologically, the nodules consisted of numerous, small, fibrinoid masses. The synovium adjacent to the tendon sheath was hyperplastic, with fibrinoid necrosis mimicking rheumatoid synovium. However, synovitis was not present inside the adjacent joint. HTLV-I proviral DNA was detected in the cells of the nodule, in tenosynovial cells, and in peripheral blood lymphocytes, but not in skin fibroblasts. In situ reverse transcription assay showed a high quantity of zyxwvutsrq taxlrex messenger RNA in the proliferating lining cells. Based on these features, we classified this case as an atypical manifestation of HTLV-I-associated arthropathy associated with fibrin- oid nodules resulting from chronic tenosynovitis. Results of recent studies suggest that human T cell lymphotropic virus I (HTLV-I) may be a causative agent in myelopathy, arthropathy, Sjogren’s syndrome, uveitis, adult T cell leukemia, and other immunogenic disorders (1-6). Accumulating evidence from in vivo, in vitro, and epidemiologic studies suggest that HTLV-I is involved in arthritis (7-11). Supported by the Ministry of Health and Welfare and the Ministry of Education of Japan. Tomoko Hasunuma, MD, Tran Thi Minh Hoa, MD, Hiroyuki Aono, PhD, Kusuki Nishioka, MD: Institute of Medical Science, St. Marianna University, Kawasaki, Japan; Tadanobu Morimoto, MD, Ryokei Ogawa, MD: Kansai Medical University, Osaka, Japan; Ulf Miiller-Ladner, MD, Steffen Gay, MD: The University of Alabama at Birmingham. Address reprint requests to Kusuki Nishioka, MD, Professor of Medicine, Director, Division of Rheumatology and Immunology, Institute of Medical Science, St. Marianna University, 2-16-1 Sugao, Miyamae-ku, Kawasaki-shi, Kanagawa-ken 216 Japan. Submitted for publication April 15, 1996; accepted in revised form September 3, 1996. We describe here the case of an HTLV-I carrier with subcutaneous nodulosis and tenosynovitis without arthritis. The nodules were composed of fibrinoid ma- terial, probably resulting from tenosynovitis in adjacent areas. Pathologic and molecular examinations were con- ducted to clarify the role of HTLV-I in this case. CASE REPORT The patient, a 45-year-old man, presented in 1990 with systemic lymphadenopathy and multiple subcutane- ous nodules on both thighs, after a 2-week episode of general fatigue, mild fever, and leg edema. A provisional diagnosis of malignant lymphoma was made, and ingui- nal lymph node biopsy was performed. However, histo- logic examination of the tissue showed only nonspecific lymphadenitis and no evidence of lymphoma. The condition was considered to be polymyalgia rheumatica based on clinical symptoms and a progres- sive rise in the erythrocyte sedimentation rate (ESR; 82 mm/hour). Accordingly, the patient was treated with 10 mgJday of prednisolone, which caused regression of the lymphadenopathy and disappearance of the fever. Two years later, subcutaneous nodules appeared on the dorsal aspect of the patient’s right elbow, the flexor tendons of both forearms, the dorsal aspect of the second and third metacarpophalangeal (MCP) joints bilaterally, the dorsal aspect of the wrist joints bilater- ally, and the left scapular region. Because he complained of stiffness and pain in the affected areas, the nodules on the right elbow and both forearms were surgically re- moved on March 5,1993, and those on the dorsal side of the wrists were removed on July 27, 1994. Since synovitis was also detected in the adjacent tendon sheath, treat- ment with 5 mg/day of prednisolone and 100 mg/day of bucillamine (a sulfhydryl compound used as an antirheu- matic drug in Japan) was instituted after the first oper- ation, but treatment failed to inhibit the development of new nodules.