Adult-onset Still's disease: an unusual presentation of rubella infection S.H.K. HUANG, MD; W.E. DECOTEAU, MD Still's disease in the adult is uncom- and tenderness of both ankles and sion, but it rose to 1/5120 1 mon."4 Recently Harth, Thompson several metacarpophalangeal joints. week later and 1/10240 6 weeks and Ralph4 described four adults The leukocyte count was 25.75 x later. Roentgenograms of the joints with this disease. Two of the pa- 109/1 (78% neutrophils), the ery- were normal. A technetium 99m tients had elevated rubella antibody throcyte sedimentation rate (ESR) liver and spleen scan revealed he- titres, and rubella virus was isolated 91 mm/h (Wintrobe) and the C- patosplenomegaly, with an increase from the urine of one of them. In reactive protein value 4+. She was in the splenic span to 20 cm. An an accompanying editorial5 Petty treated with acetylsalicylic acid, 6 echocardiogram, gallium 67 total noted the "provocative and possibly g/d and penicillin G, 500 mg four body scan, abdominal ultrasono- coincidental similarities" between times a day by mouth for, tenta- gram, chest roentgenogram and rubella arthritis and Still's disease, tively, rheumatic fever. electrocardiogram were all normal. and suggested further studies to When there was no improvement The patient's temperature con- clarify the relation between rubella a week later the woman was ad- tinued to spike to 40°C each day infection and Still's disease. There- mitted to University Hospital. By in association with chills and pro- fore, we are reporting the case of this time she had lost 16 kg. She fuse sweating. Typically the rash a 29-year-old woman with typical looked ill, had a temperature of faded in the evening and recurred adult-onset Still's disease who had 39°C and was having chills. A the next day with the fever. a brisk rise in the rubella antibody salmon-pink macular rash was Four days after admission acute titre. noted over the extremities and the monoarthritis of the left wrist was trunk. A grade 3/6 systolic ejec- noted. Aspirated synovial fluid had Case report tion murmur was heard at the left a leukocyte count of 40 x 10'/l A 29-year-old woman presented sternal border. The liver and spleen (93% neutrophils); a routine cul- to her family physician with a sore were palpable, but there was no ture was negative. The inflamma- throat, fever, rash, polyarthralgia synovitis. tion spontaneously subsided in 48 and severe myalgia of the back and The hemoglobin level was 12 hours but was followed 4 days later legs. Her fever was remittent, with g/dl, the, leukocyte count 9.2 x by acute arthritis of the right knee. a daily spike in temperature to 109/l, the ESR 105 mm/h (Win- Shortly thereafter the left fifth prox- 40°C associated with chills and trobe) and the serum albumin level imal interphalangeal joint became profuse sweating. The rash con- 2.9 g/dl. The serum levels of the involved. Therapy with indome- sisted of small, mildly pruritic, sal- following were persistently elev- thacin, 50 mg three times a day, mon-pink macules over the legs ated: alkaline phosphatase, at 110 was begun; almost immediately the and, later, the back and arms. to 234 (normal 30 to 85) IU/l; daily temperature spike was re- Characteristically, the rash disap- lactate dehydrogenase, at 272 to duced and the rash cleared. peared during the night and reap- 386 (normal 100 to 270) IU/l; and The patient was discharged feel- peared the next day with the fever. glutamic oxaloacetic transaminase, ing well following a total stay in The joint pain was migratory, af- at 57 to 109 (normal 10 to 50) hospital of 16 days. Follow-up fecting the wrists, elbows, knees, IU/l. The following gave either visits 1 and 2 months later revealed ankles and, finally, the small joints negative or normal results: tests for no signs or symptoms, and the labo- of the hands and feet. antinuclear antibody, DNA binding ratory findings were all normal ex- At the time of physical examina- and rheumatoid factor; measure- cept for the rubella antibody titre, tion her temperature was 40°C and ment of the serum levels of immu- which had remained at 1/ 10240. the pulse rate 100 beats/min. A 1/6 noglobulins A, G and M and Discussion systolic ejection murmur was de- third component of complement; tected along the left sternal border. multiple blood and urine cultures; The patient we have presented There was swelling of both knees and serologic tests for cytomegalo- had typical clinical features of virus, measles and mumps viruses, adult-onset Still's disease. In addi- Mycoplasma, Salmonella, hepatitis tion, there was strong evidence of From the rheumatic disease unit, recent rubella infection because of department of medicine, University B antigen, Brucel a, Toxoplasma, Hospital, Saskatoon Yersinia, Treponema pallidum, he- a brisk rise in the titre of rubella terophil antibody and antistrepto- hemagglutination-inhibiting anti- Reprint requests to: Dr S.H.K. Huang, Rheumatic disease unit, Department lysin 0. The titre of rubella he- body. of medicine, University Hospital, magglutination-inhibiting antibody The relation between the pa- Saskatoon, Sask. S7N OXO was 1/1280 at the time of admis- tient's Still's disease and rubella in- CMA JOURNAL/JUNE 7, 1980/VOL. 122 1275 fection is unknown, but some of pincott, Philadelphia, 1977, p 579 1157, 1975 the possibilities are as follows. 9. OGRA PL, HERD JK: Serologic asso- 11. COOPER LZ: The rubella hemagglu- ciation of rubella virus infection and tination inhibition test, in Rubella; First, rubella infection can produce juvenile rheumatoid arthritis (abstr). First Annual Symposium of the a variety of musculoskeletal disor- Arthritis Rheum 15: 121, 1972 Eastern Pennsylvania Branch, Amer- ders, including polyarthritis of the 10. OGRA PL, OGRA SS, CHIBA Y, et al: ican Society for Microbiology, small joints, which closely mimics Rubella-virus infection in juvenile FRIEDMAN H, PRIER JE (eds), CC adult-onset rheumatoid arthritis,4 rheumatoid arthritis. Lancet 1: Thomas, Springfield, III, 1973, p 8 an illness that resembles juvenile rheumatoid arthritis,9'0 and, as in our case, a systemic illness that has I BOOKS the typical features of adult-onset Still's disease. Second, it is possible continued from page 1274 that the rubella antibody response in this patient was nonspecific, as HORMONAL BIOLOGY OF ENDOME- PRACTICAL OBSTETRICS PROBLEMS. patients with various inflammatory TRIAL CANCER. A Series of Work- 5th ed. Ian Donald. 1071 pp. lllust. Lloyd- disorders can show nonspecific in- shops on the Biology of Human Cancer. Luke (Medical Books) Ltd., London, Report No. 8. UICC Technical Report 1979. Price not stated. ISBN 0-85324- creases in the titres of various anti- Series - Volume 42. Edited by G.S. 132-5 bodies.11 However, the eightfold Richardson and D.T. MacLaughlin. 187 MED- rise in pp. Illust. International Union Against PROBLEM SOLVING IN CLINICAL the rubella antibody titre Cancer, Geneva, 1978. $13.41, paper- ICINE. From Data to Diagnosis. Paul and the lack of elevation of other bound. ISBN 92-9018-042-0 Cutier. 373 pp. The Williams & Wilkins in Company, Baltimore; the Macmillan antibody titres our patient make A MANUAL OF ADVERSE DRUG INTER- Company of Canada Limited, Toronto, this unlikely. Finally, it is possible, ACTIONS. 2nd ed. J.P. Griffin and P.F. 1979. $24.50. ISBN 0-683-0225-2 though unlikely, that the concomit- D'Arcy. 402 pp. Illust. John Wright & PROSTATIC CANCER. Edited by Gerald ant occurrence of adult-onset Still's Sons Ltd., Bristol; Year Book Medical P. Murphy. 238 pp. lllust. PSG Publish- Publishers Inc., Chicago, 1979. Price ing Company, Inc., Littleton, Massa- disease and rubella infection in our not stated. ISBN 0-8151-4001-0 chusetts; the Macmillan Company of patient was purely coincidental. Canada Limited, Toronto, 1979. $30.95. MEDICAL EXPERIMENTATION. Edited ISBN 0-88416-190-0 Unfortunately, no attempt was by Amnon Carmi. 148 pp. Turtledove made to isolate rubella virus from Publishing, Ramat Gan, Israel; ISBS, PSYCHO-SOCIAL ASPECTS OF A SE- the synovial aspirate, which might Inc., Forest Grove, Oregon, 1979. $12.50. VERE BURN: A Review of the Litera- ISBN 965-20-0002-7 ture. 1979 Supplement to the Interna- have clarified the association be- tional Bibliography on Burns. M.L. tween these two entities. However, MEDICAL ONCOLOGY. An Advanced Bowden, Claudella A. Jones and Irving this report should alert clinicians Course. A Self-Assessment Guide for Feller. 127 pp. lllust. National Institute Subspecialty Board Examinations and for Burn Medicine, Ann Arbor, Michi- to study their patients with adult- Practice. Joseph G. Sinkovics. 679 pp. gan, 1979. Price not stated, paperbound. onset Still's disease for the presence Illust. Marcel Dekker, Inc., New York, ISBN 0-917478-24-7 1979. $49.50. ISBN 0-8247-6863-9 of rubella and other infections. Like PULMONARY EDEMA. Edited by Alfred many other diseases, adult-onset MEMBRANE BIOENERGETICS. Based on P. Fishman and Eugene M. Renkin. Still's disease be a the International Workshop held at 261 pp. Illust. American Physiological may syndrome Cranbrook Schools, Bloomfield Hills, Society, Bethesda, Maryland; the Mac- of many causes. Michigan, July 5-7, 1979, in honor of millan Company of Canada Limited, Efraim Racker. Edited by C.P. Lee, G. Toronto, 1979. $38.95. ISBN 0-683-08245- Schatz and L. Ernster. 609 pp. Illust. 3 References Addison-Wesley Publishing Company, Inc., Reading, Massachusetts, 1979. PULMONARY EMBOLISM. Walter G. 1. BYWATERS EGL: Still's disease in $25.50, paperbound. ISBN 0-201-03999-0 Wolfe and David C.