DOI QR코드

DOI QR Code

A case of encephalitis in a juvenile rheumatoid arthritis patient treated with etanercept

소아기 류마티스모양 관절염 환자에서 etanercept 사용 후 발생한 뇌염 1예

  • Kwon, Ah Reum (Department of Pediatrics, Yonsei University College of Medicine, Severance Children's Hospital) ;
  • Park, Eun Jung (Department of Pediatrics, Yonsei University College of Medicine, Severance Children's Hospital) ;
  • Kim, Ki Hwan (Department of Pediatrics, Yonsei University College of Medicine, Severance Children's Hospital) ;
  • Kim, Dong Soo (Department of Pediatrics, Yonsei University College of Medicine, Severance Children's Hospital)
  • 권아름 (연세대학교 의과대학 소아과학교실) ;
  • ;
  • 김기환 (연세대학교 의과대학 소아과학교실) ;
  • 김동수 (연세대학교 의과대학 소아과학교실)
  • Received : 2009.07.24
  • Accepted : 2009.10.18
  • Published : 2010.02.15

Abstract

Tumor necrosis $factor-{\alpha}$ ($TNF-{\alpha}$) is a major proinflammatory cytokine involved in the pathophysiology of juvenile rheumatoid arthritis. Etanercept is an effective inhibitor of $TNF-{\alpha}$ and has shown a beneficial effect in patients with JRA. However, the most important cause of concern related to etanercept administration is infection. We report a case of encephalitis in a JRA patient receiving long-term treatment with etanercept. The patient was a 4-year-old boy with refractory JRA, and he received etanercept subcutaneously at a dose of $0.4\;mg\;kg^{-1}\;day^{-1}$ twice a week for 14 months, along with non-steroidal anti-inflammatory drugs, methotrexate, oral steroids, and sulfasalazine. The patient presented with sudden fever, headache, vomiting, a generalized tonic seizure, and changes in mental status. We suspected a central nervous system infection, and simultaneously administered antibiotics, an antiviral agent, and steroids. After 2 days of hospitalization, his mental function returned to normal, and he showed no further seizure-like movements. Brain magnetic resonance imaging scan of the patient showed a multifocal cortical lesion on both sides of the temporoparietooccipital lobe, which indicated encephalitis. Although we were unable to identify the causative organism of encephalitis, we think that the encephalitis may be attributed to infection, and the use of etanercept may have increased the risk of severe infection. Therefore, etanercept was discontinued and the patient recovered shortly after. To the best of our knowledge, this is the first case of encephalitis in a juvenile rheumatoid arthritis patient treated with etanercept.

$TNF-{\alpha}$ 는 소아기 류마티스모양 관절염의 병태생리학에 관여하는 주요 cytokine 이다. Etanercept 는 $TNF-{\alpha}$ 억제제 중 하나로 소아기 류마티스모양 관절염에 효과적인 약물로 각광받고 있다. Etanercept의 주요 부작용은 면역력 저하에 의한 감염으로, 대게 중등도의 상기도 감염이 대부분으로 알려져 있으나, 최근 중증의 감염도 보고되고 있다. 저자들은 소아기 류마티스모양 관절염 환자가 etanercept를 14개월간 투약 후 발생한 뇌염 1예를 경험하였기에 보고하는 바이다.

Keywords

References

  1. Ilowite NT. Current treatment of juvenile rheumatoid arthritis. Pediatrics 2002;109:109-15 https://doi.org/10.1542/peds.109.1.109
  2. Huang JL. Methotrexate in the treatment of children with chronic arthritis-long-term observations of efficacy and safety. Br J Clin Pract 1996;50:311-4
  3. Ou LS, See LC, Wu CJ, Kao CC, Lin YL, Huang JL. Association between serum inflammatory cytokines and disease activity in juvenile idiopathic arthritis. Clin Rheumatol 2002;21:52-6 https://doi.org/10.1007/s100670200012
  4. Mohler KM, Torrance DS, Smith CA, Goodwin RG, Stremler KE, Fung VP, et al. Soluble tumor necrosis factor (TNF) receptors are effective therapeutic agents in lethal endotoxemia and function simultaneously as both TNF carriers and TNF antagonists. J Immunol 1993;151:1548-61
  5. Russo RA, Katsicas MM, Zelazko M. Etanercept in systemic juvenile idiopathic arthritis. Clin Exp Rheumatol 2002;20:723-6
  6. Lovell DJ, Giannini EH, Reiff A, Jones OY, Schneider R, Olson JC, et al. Long-term efficacy and safety of etanercept in children with polyarticular-course juvenile rheumatoid arthritis: interim results from an ongoing multicenter, openlabel, extended-treatment trial. Arthritis Rheum 2003;48:218-26 https://doi.org/10.1002/art.10710
  7. Fleischmann R, Iqbal I, Nandeshwar P, Quiceno A. Safety and efficacy of disease-modifying anti-rheumatic agents: focus on the benefits and risks of etanercept. Drug Saf 2002;25:173-97 https://doi.org/10.2165/00002018-200225030-00004
  8. Mori S, Tomita Y, Horikawa T, Cho I, Sugimoto M. Delayed spinal infection after laminectomy in a patient with rheumatoid arthritis interruptedly exposed to anti-tumor necrosis factor alpha agents. Clin Rheumatol 2008;27:937-9 https://doi.org/10.1007/s10067-008-0869-1
  9. Elbek O, Uyar M, Aydin N, Borekci S, Bayram N, Bayram H, et al. Increased risk of tuberculosis in patients treated with antitumor necrosis factor alpha. Clin Rheumatol 2009;28:421-6 https://doi.org/10.1007/s10067-008-1067-x
  10. Levinson JE, Wallace CA. Dismantling the pyramid. J Rheumatol Suppl 1992;33:6-10
  11. Fantini F, Gerloni V, Gattinara M, Cimaz R, Arnoldi C, Lupi E. Remission in juvenile chronic arthritis: a cohort study of 683 consecutive cases with a mean 10 year followup. J Rheumatol 2003;30:579-84
  12. Lovell DJ. Update on treatment of arthritis in children: new treatments, new goals. Bull NYU Hosp Jt Dis 2006;64:72-6
  13. Carrasco R, Smith JA, Lovell D. Biologic agents for the treatment of juvenile rheumatoid arthritis: current status. Paediatr Drugs 2004;6:137-46 https://doi.org/10.2165/00148581-200406030-00001
  14. Lovell DJ, Reiff A, Ilowite NT, Wallace CA, Chon Y, Lin SL, et al. Safety and efficacy of up to eight years of continuous etanercept therapy in patients with juvenile rheumatoid arthritis. Arthritis Rheum 2008;58:1496-504 https://doi.org/10.1002/art.23427
  15. Phillips K, Husni ME, Karlson EW, Coblyn JS. Experience with etanercept in an academic medical center: are infection rates increased? Arthritis Rheum 2002;47:17-21 https://doi.org/10.1002/art1.10243
  16. Steiner I, Budka H, Chaudhuri A, Koskiniemi M, Sainio K, Salonen O, et al. Viral encephalitis: a review of diagnostic methods and guidelines for management. Eur J Neurol 2005;12:331-43 https://doi.org/10.1111/j.1468-1331.2005.01126.x
  17. Tebas P, Nease RF, Storch GA. Use of the polymerase chain reaction in the diagnosis of herpes simplex encephalitis: a decision analysis model. Am J Med 1998;105:287-95 https://doi.org/10.1016/S0002-9343(98)00259-9
  18. Muir P, van Loon AM. Enterovirus infections of the central nervous system. Intervirology 1997;40:153-66 https://doi.org/10.1159/000150542