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International Journal of Hematology

, Volume 108, Issue 4, pp 438–442 | Cite as

Clinical characteristics of pediatric patients with myeloid sarcoma without bone marrow involvement in Japan

  • Takashi Taga
  • Toshihiko Imamura
  • Kentaro Nakashima
  • Naoko Maeda
  • Akihiro Watanabe
  • Yuji Miyajima
  • Sachi Sakaguchi
  • Hitoshi Sano
  • Daiichiro Hasegawa
  • Hirohide Kawasaki
  • Souichi Adachi
  • Masatoshi Takagi
  • Katsuyoshi Koh
  • Atsushi Manabe
  • Tomohiko Taki
  • Yasushi Ishida
Original Article
  • 154 Downloads

Abstract

Myeloid sarcoma (MS) is a rare neoplastic condition that is often described in association with acute myeloid leukemia (AML). MS in childhood has received little attention, particularly in Japan. We carried out a nationwide retrospective analysis of Japanese children diagnosed with MS without bone marrow involvement. Inclusion criteria were diagnosis of MS at younger than 20 years of age between January 1, 2000 and December 31, 2013. There was a predominance of males (8:2), and the median age at MS diagnosis was 4 years. Sites of involvement varied and included skin (n = 3), head and/or neck (n = 2), and multiple sites (n = 2). Karyotypes were evaluated in seven patients, with one individual carrying t(8;21) and t(9;11). Four patients developed bone marrow involvement 2–55 months after diagnosis of MS. All patients received chemotherapy for de novo AML and two individuals received HSCT in first remission. Seven of ten patients survived for 50–152 months (median, 93 months) without disease after initial chemotherapy. This retrospective study confirmed that pediatric MS without bone marrow involvement in Japan is a very rare disease. MS patients responded favorably to therapies for de novo AML, and HSCT in first remission was not indicated for all patients.

Keywords

Myeloid sarcoma Children AML Chemotherapy 

Abbreviations

CR

Complete remission

SCT

Stem cell transplantation

AML99

AML99 protocol2

CCLSG AML 9805

CCLSG AML 9805 protocol3

AML-05

JPLSG AML-05 protocol4

VAC

Vincristine, actinomycin D and cyclophosphamide

ECM

Etoposide, cytarabine and mitoxantrone

HCEI

High-dose cytarabine, etoposide and idarubicin

FLAG-IDA

Fludarabine, high-dose cytarabine, granulocyte-colony stimulating factor and idarubicin

MPO

Myeloperoxidase

ND

Not done

R-BM

Related bone marrow

U-CB

Unrelated cord blood

MS to AML

Duration from onset of MS to AML

OS

Overall survival

Notes

Acknowledgements

We deeply thank all members of the JSPHO who cooperated in the retrospective study of rare leukemia of children, including myeloid sarcoma with bone marrow involvement.

Compliance with ethical standards

Conflict of interest

The authors declare no conflicts of interest in association with this study.

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Copyright information

© The Japanese Society of Hematology 2018

Authors and Affiliations

  • Takashi Taga
    • 1
    • 2
  • Toshihiko Imamura
    • 1
    • 3
  • Kentaro Nakashima
    • 4
  • Naoko Maeda
    • 5
  • Akihiro Watanabe
    • 6
  • Yuji Miyajima
    • 7
  • Sachi Sakaguchi
    • 8
  • Hitoshi Sano
    • 9
  • Daiichiro Hasegawa
    • 10
  • Hirohide Kawasaki
    • 1
    • 11
  • Souichi Adachi
    • 1
    • 12
  • Masatoshi Takagi
    • 1
    • 13
  • Katsuyoshi Koh
    • 1
    • 14
  • Atsushi Manabe
    • 1
    • 15
  • Tomohiko Taki
    • 1
    • 16
  • Yasushi Ishida
    • 1
    • 17
  1. 1.Leukemia/Lymphoma Committee of Japanese Society of Pediatric Hematology and OncologyTokyoJapan
  2. 2.Department of PediatricsShiga University of Medical ScienceOtsuJapan
  3. 3.Department of PediatricsKyoto Prefectural University of MedicineKyotoJapan
  4. 4.Department of Pediatrics, Graduate School of MedicineKyushu UniversityFukuokaJapan
  5. 5.Department of Pediatrics, National Hospital OrganizationNagoya Medical CenterNagoyaJapan
  6. 6.Department of PediatricsNiigata Cancer Center HospitalNiigataJapan
  7. 7.Department of PediatricsAnjo Kosei HospitalAnjoJapan
  8. 8.Department of PediatricsJuntendo University HospitalTokyoJapan
  9. 9.Department of PediatricsTottori University HospitalYonagoJapan
  10. 10.Department of Hematology/OncologyHyogo Children’s Medical CenterKobeJapan
  11. 11.Department of PediatricsKansai Medical UniversityHirakataJapan
  12. 12.Department of Human Health Sciences, Graduate School of MedicineKyoto UniversityKyotoJapan
  13. 13.Department of Pediatrics and Developmental BiologyTokyo Medical and Dental University Graduate School of Medical and Dental SciencesTokyoJapan
  14. 14.Department of Hematology/OncologySaitama Children’s Medical CenterSaitamaJapan
  15. 15.Department of PediatricsSt. Luke’s International HospitalTokyoJapan
  16. 16.Department of Medical TechnologyKyorin University Faculty of Health SciencesMitakaJapan
  17. 17.Department of PediatricsEhime Prefectural Central HospitalMatsuyamaJapan

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