Clinical Rheumatology

, Volume 31, Issue 3, pp 569–574

Successful treatment of adult-onset Still’s disease with tocilizumab monotherapy: two case reports and literature review

  • Ryota Sakai
  • Hayato Nagasawa
  • Eiko Nishi
  • Ayumi Okuyama
  • Hirofumi Takei
  • Takahiko Kurasawa
  • Tsuneo Kondo
  • Koji Nishimura
  • Yuichiro Shirai
  • Tatsuya Ito
  • Hideto Kameda
  • Tsutomu Takeuchi
  • Koichi Amano
Case Based Review

DOI: 10.1007/s10067-011-1917-9

Cite this article as:
Sakai, R., Nagasawa, H., Nishi, E. et al. Clin Rheumatol (2012) 31: 569. doi:10.1007/s10067-011-1917-9

Abstract

Adult-onset Still’s disease (AOSD) is a systemic inflammatory disease of unknown etiology. Recently, it has been reported that quite a few cases of refractory AOSD were successfully treated with tocilizumab (TCZ) and corticosteroids were withdrawn in some of these patients. We report two AOSD patients who were treated successfully with TCZ monotherapy; thus, avoiding corticosteroid treatment. Because both of the patients refused to take corticosteroids, we planned to treat them with 8 mg/kg of TCZ monotherapy at weeks 0, 2, 6 and subsequently every 4 weeks. The efficacy of TCZ was assessed by patients’ clinical symptoms such as fever, arthralgia, skin eruptions, and laboratory markers such as serum levels of CRP, ferritin, and IL-6. We also reviewed 14 previous case reports including 30 cases who had been treated with TCZ for AOSD. Our patients responded rapidly and have been maintained in clinical remission without corticosteroid treatment. In the literature review, concomitant corticosteroid treatment described in 13 cases was successfully tapered in 7 and discontinued in 6 cases. TCZ monotherapy can be a candidate for the first-line therapy for some AOSD patients.

Keywords

Adult-onset Still’s diseaseInterleukin-6MonotherapyTocilizumab

Copyright information

© Clinical Rheumatology 2011

Authors and Affiliations

  • Ryota Sakai
    • 1
  • Hayato Nagasawa
    • 1
  • Eiko Nishi
    • 1
  • Ayumi Okuyama
    • 1
  • Hirofumi Takei
    • 1
  • Takahiko Kurasawa
    • 1
  • Tsuneo Kondo
    • 1
  • Koji Nishimura
    • 1
  • Yuichiro Shirai
    • 1
  • Tatsuya Ito
    • 3
  • Hideto Kameda
    • 2
  • Tsutomu Takeuchi
    • 1
    • 2
  • Koichi Amano
    • 1
  1. 1.Department of Rheumatology and Clinical Immunology, Saitama Medical CenterSaitama Medical UniversityKawagoeJapan
  2. 2.Division of Rheumatology, Department of Internal Medicine, School of MedicineKeio UniversityTokyoJapan
  3. 3.Department of Internal MedicineSaitama Yorii HospitalYoriiJapan