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Successful treatment of JMML with related bone marrow transplantation after reduced-intensity conditioning

Bone Marrow Transplantation volume 36, pages 453–454 (2005)Cite this article

Juvenile myelomonocytic leukemia (JMML) is a rare hematopoietic malignancy that occurs during early childhood, representing about 2% of all childhood hematologic malignancies.1 Allogeneic hematopoietic stem cell transplantation (HSCT) is presently the only curative treatment for JMML, because the disease usually progresses rapidly. Leukemia recurrence represents the main cause of treatment failure in children with JMML given HSCT, with the relapse rate being as high as 50%.2 Relapse occurs early, generally within the first year after the allograft.3, 4 On the other hand, HSCT is associated with high transplantation-related mortality (TRM) because the patients received myeloablative conditioning regimen that contained total body irradiation (TBI) or busulfan.2, 5, 6

The recent paper by Korthof et al6 represented that intensive chemotherapy given before the start of the conditioning for HSCT had a significant adverse effect on the relapse rate after HSCT. However, they noted that this finding was not in agreement with the findings of other authors and needed further study. In a recent multicenter overview from the European Working Group on Childhood MDS and the European Blood and Marrow Transplantation Group, no significant differences in terms of event-free survival, cumulative incidence of relapse, and TRM were observed between children given acute myeloid leukemia (AML)-type chemotherapy or less intensive treatment.7 They also described that neither splenectomy prior to HSCT nor spleen size at the time of transplantation influenced the outcome. However, it is a retrospective and multi-institutional study. Moreover, they reported that 47% had achieved a second sustained hematologic remission thanks to a second transplantation. They hypothesized that less intensive graft-versus-host-disease (GVHD) prophylaxis could have contributed to the sustained remission after the second allograft in these patients by better preserving graft-versus-leukemia effect. The remaining controversial issues are the role of pretransplant therapies. Although cytoreductive therapy before HSCT remains unproven, consensus clinical opinion recommends the use of AML-like chemotherapy to reduce the tumor burden before HSCT.

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Authors and Affiliations

  1. Department of Pediatrics, Osaka Medical Center and Research Institute for Maternal and Child Health, Osaka, Japan

    M Koyama, Y Takeshita, A Sakata, A Sawada, M Yasui, T Okamura, M Inoue & K Kawa

  2. Department of Pediatrics, Kansai Medical University Hospital, Osaka, Japan

    T Nakano

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  1. M Koyama
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Koyama, M., Nakano, T., Takeshita, Y. et al. Successful treatment of JMML with related bone marrow transplantation after reduced-intensity conditioning. Bone Marrow Transplant 36, 453–454 (2005). https://doi.org/10.1038/sj.bmt.1705047

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