International Journal of Hematology

, Volume 95, Issue 2, pp 198–203

Interleukin-6-producing dermoid cyst associated with multicentric Castleman’s disease

  • Shigeyuki Ebara
  • Soken-Nakazawa J. Song
  • Hiroyuki Mizuta
  • Yasushi Ito
  • Kenji Hasegawa
  • Tsuneko Kamata
  • Teppei Matsumura-Nishikawa
  • Takafumi Ogawa
  • Jyunichi Soneda
  • Kazuyuki Yoshizaki
Case Report

DOI: 10.1007/s12185-011-0978-5

Cite this article as:
Ebara, S., Song, SN.J., Mizuta, H. et al. Int J Hematol (2012) 95: 198. doi:10.1007/s12185-011-0978-5

Abstract

Dysregulated overproduction of interleukin-6 (IL-6) from activated B cells in affected lymph nodes has been implicated in the pathogenesis of multicentric Castleman’s disease (MCD), a rare lymphoproliferative disorder accompanied by systemic manifestations. We here report the case of a 32-year-old female presenting with MCD associated with a dermoid cyst in the pelvic cavity. The co-occurrence of MCD and dermoid cyst has not been reported before. Immunohistochemical analysis of the tissue sections showed IL-6 production in CD68-positive macrophage cells, which had infiltrated the dermoid cyst. Removal of the cyst resulted in partial improvement in systemic symptoms accompanied by a decrease in serum IL-6, while complete improvement was obtained by treatment with an anti-IL-6 receptor antibody following resection of the dermoid cyst. To the best of our knowledge, this is the first study to provide evidence of IL-6 production by CD68+ cells in a dermoid cyst involved in MCD.

Keywords

Multicentric Castleman’s disease (MCD) Interleukin 6 (IL-6) IL-6 producing dermoid cyst Tocilizumab 

Copyright information

© The Japanese Society of Hematology 2011

Authors and Affiliations

  • Shigeyuki Ebara
    • 1
  • Soken-Nakazawa J. Song
    • 2
  • Hiroyuki Mizuta
    • 1
  • Yasushi Ito
    • 1
  • Kenji Hasegawa
    • 1
  • Tsuneko Kamata
    • 1
  • Teppei Matsumura-Nishikawa
    • 2
  • Takafumi Ogawa
    • 3
  • Jyunichi Soneda
    • 1
  • Kazuyuki Yoshizaki
    • 1
    • 2
  1. 1.Kobe Tokushukai HospitalKobeJapan
  2. 2.Immuno-Medical Science, Division of Applied Chemistry, Graduate School of EngineeringOsaka UniversitySuitaJapan
  3. 3.Kyoudo Byori Inc., Institute of PathologyKobeJapan

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