International Journal of Hematology

, Volume 97, Issue 6, pp 782–785

Spontaneous remission of 2-chlorodeoxyadenosine (2-CdA)-related secondary myelodysplastic syndrome in a patient with refractory Langerhans cell histiocytosis

  • Daisuke Suzuki
  • Ryoji Kobayashi
  • Hirozumi Sano
  • Kenji Kishimoto
  • Kazue Yasuda
  • Masanori Nakanishi
  • Tetsuro Nagashima
  • Kunihiko Kobayashi
Original Article

DOI: 10.1007/s12185-013-1343-7

Cite this article as:
Suzuki, D., Kobayashi, R., Sano, H. et al. Int J Hematol (2013) 97: 782. doi:10.1007/s12185-013-1343-7

Abstract

Langerhans cell histiocytosis (LCH) is sometimes resistant to conventional chemotherapies, and treatment with 2-chlorodeoxyadenosine (2-CdA) is gaining importance as a salvage treatment for refractory or recurrent LCH. Secondary malignancies such as acute myelogenous leukemia and myelodysplastic syndrome (MDS) due to 2-CdA have recently been reported. However, there have been no reports to date of cases of 2-CdA-related secondary MDS in which spontaneous remission was achieved. Here, we report the case of a 1-year-old boy with an occipital tumor who was diagnosed with LCH by biopsy and underwent chemotherapy. However, the disease relapsed and became refractory to chemotherapy. He received 2-CdA treatment, which was effective. However 6 months after the start of treatment, he developed MDS with chromosomal abnormality of 7q−. After 1-year observation without any intervention, his hematological findings had returned to normal, and the chromosomal abnormality had also disappeared. To our knowledge, this is the first report of 2-CdA-related MDS with spontaneous hematological remission.

Keyword

Secondary myelodysplastic syndromeSpontaneous remission2-ChlorodeoxyadenosineLangerhans cell histiocytosis

Copyright information

© The Japanese Society of Hematology 2013

Authors and Affiliations

  • Daisuke Suzuki
    • 1
  • Ryoji Kobayashi
    • 1
  • Hirozumi Sano
    • 1
  • Kenji Kishimoto
    • 1
  • Kazue Yasuda
    • 1
  • Masanori Nakanishi
    • 2
  • Tetsuro Nagashima
    • 2
  • Kunihiko Kobayashi
    • 1
  1. 1.Department of PediatricsSapporo Hokuyu HospitalShiroishikuJapan
  2. 2.Department of PediatricsKushiro Red Cross HospitalKushiroJapan