Skip to main content

Thank you for visiting nature.com. You are using a browser version with limited support for CSS. To obtain the best experience, we recommend you use a more up to date browser (or turn off compatibility mode in Internet Explorer). In the meantime, to ensure continued support, we are displaying the site without styles and JavaScript.

  • Letter to the Editor
  • Published:

Successful double umbilical cord blood transplantation for relapsed juvenile myelomonocytic leukemia

Leukemia volume 27, pages 988–989 (2013)Cite this article

Subjects

Juvenile myelomonocytic leukemia (JMML) is an aggressive myeloproliferative neoplasm of early childhood for which allogeneic hematopoietic stem cell transplantation (HSCT) is the mainstay of treatment.1 HSCT for JMML is burdened with a high probability of relapse (5-year cumulative incidence at 35%), despite high-intensity conditioning regimens and rapid tapering of immunosuppressants so as to maximize graft-versus-leukemia effects.1, 2 In relapsed cases, donor lymphocyte infusions fail to show consistent therapeutic efficacy, and it is generally felt that the risk-benefit ratio of this modality does not warrant universal recommendation in relapsed JMML. Data compiled by the European Working Group of Myelodysplastic Syndromes in Childhood indicate that approximately half of relapsed cases can be rescued with a second HSCT.3

Assuming inferior engraftment capability and weaker graft-versus-leukemia properties, umbilical cord blood (UCB) is not considered as the stem cell source of choice for children with JMML undergoing HSCT. However, it remains a potential alternative option for patients who lack a suitable related or unrelated donor. A total of 19 UCB transplantations for children with JMML have been reported in the literature with a failure rate of 8/19 or 42% (relapse 6/19; non-engraftment and subsequent relapse 1/19; other transplantation-related mortality 1/19).1, 4, 5, 6, 7 A large retrospective evaluation of 110 UCB transplantations for JMML was presented by Crotta et al.8 at the 2011 annual meeting of the European Group for Blood and Marrow Transplantation; the 4-year cumulative incidence of relapse was 37% and 4-year transplant-related mortality was 20%.

This is a preview of subscription content, access via your institution

Access options

Buy this article

  • Purchase on Springer Link
  • Instant access to full article PDF
Buy now

Prices may be subject to local taxes which are calculated during checkout

References

  1. Locatelli F, Nöllke P, Zecca M, Korthof E, Lanino E, Peters C et al. Hematopoietic stem cell transplantation (HSCT) in children with juvenile myelomonocytic leukemia (JMML): results of the EWOG-MDS/EBMT trial. Blood 2005; 105: 410–419.

    Article  CAS  Google Scholar 

  2. Yoshimi A, Niemeyer CM, Bohmer V, Duffner U, Strahm B, Kreyenberg H et al. Chimaerism analyses and subsequent immunological intervention after stem cell transplantation in patients with juvenile myelomonocytic leukaemia. Br J Haematol 2005; 129: 542–549.

    Article  Google Scholar 

  3. Yoshimi A, Mohamed M, Bierings M, Urban C, Korthof E, Zecca M et al. Second allogeneic hematopoietic stem cell transplantation (HSCT) results in outcome similar to that of first HSCT for patients with juvenile myelomonocytic leukemia. Leukemia 2007; 21: 556–560.

    Article  CAS  Google Scholar 

  4. MacMillan ML, Davies SM, Orchard PJ, Ramsay NK, Wagner JE . Haemopoietic cell transplantation in children with juvenile myelomonocytic leukaemia. Br J Haematol 1998; 103: 552–558.

    Article  CAS  Google Scholar 

  5. Tanoshima R, Goto H, Yanagimachi M, Kajiwara R, Kuroki F, Yokota S . Graft versus leukemia effect against juvenile myelomonocytic leukemia after unrelated cord blood transplantation. Pediatr Blood Cancer 2008; 50: 665–667.

    Article  Google Scholar 

  6. Yabe M, Sako M, Yabe H, Osugi Y, Kurosawa H, Nara T et al. A conditioning regimen of busulfan, fludarabine, and melphalan for allogeneic stem cell transplantation in children with juvenile myelomonocytic leukemia. Pediatr Transplant 2008; 12: 862–867.

    Article  CAS  Google Scholar 

  7. de Vries AC, Bredius RG, Lankester AC, Bierings M, Trebo M, Sedlacek P et al. HLA-identical umbilical cord blood transplantation from a sibling donor in juvenile myelomonocytic leukemia. Haematologica 2009; 94: 302–304.

    Article  Google Scholar 

  8. Crotta A, Rocha V, Eapen M, Wagner JE, MacMillan ML, Zecca M et al. Analysis of risk factors influencing outcomes after unrelated cord blood transplantation in children with juvenile myelomonocytic leukaemia. An Eurocord, EBMT, EWOG-MDS, CIBMTR Study. Bone Marrow Transplant 2011; 46: S15.

    Article  Google Scholar 

  9. Brunstein CG, Gutman JA, Weisdorf DJ, Woolfrey AE, DeFor TE, Gooley TA et al. Allogeneic hematopoietic cell transplantation for hematologic malignancy: relative risks and benefits of double umbilical cord blood. Blood 2010; 116: 4693–4699.

    Article  CAS  Google Scholar 

  10. Verneris MR, Brunstein CG, Barker J, MacMillan ML, DeFor T, McKenna DH et al. Relapse risk after umbilical cord blood transplantation: enhanced graft-versus-leukemia effect in recipients of 2 units. Blood 2009; 114: 4293–4299.

    Article  CAS  Google Scholar 

  11. Baumann I, Bennett JM, Niemeyer CM, Thiele J, Shannon K . Juvenile myelomonocytic leukaemia. In: Swerdlow SH, Campo E, Harris NL, Jaffe ES, Pileri SA, Stein H, et al. (eds). WHO Classification of Tumours of Haematopoietic and Lymphoid Tissues. IARC: Lyon, 2008, 82–84.

    Google Scholar 

  12. Barker JN, Weisdorf DJ, DeFor TE, Blazar BR, McGlave PB, Miller JS et al. Transplantation of 2 partially HLA-matched umbilical cord blood units to enhance engraftment in adults with hematologic malignancy. Blood 2005; 105: 1343–1347.

    Article  CAS  Google Scholar 

  13. Ramirez P, Wagner JE, DeFor TE, Blazar BR, Verneris MR, Miller JS et al. Factors predicting single-unit predominance after double umbilical cord blood transplantation. Bone Marrow Transplant 2012; 47: 799–803.

    Article  CAS  Google Scholar 

  14. Gutman JA, Turtle CJ, Manley TJ, Heimfeld S, Bernstein ID, Riddell SR et al. Single-unit dominance after double-unit umbilical cord blood transplantation coincides with a specific CD8+ T-cell response against the nonengrafted unit. Blood 2010; 115: 757–765.

    Article  CAS  Google Scholar 

  15. Orchard PJ, Miller JS, McGlennen R, Davies SM, Ramsay NK . Graft-versus-leukemia is sufficient to induce remission in juvenile myelomonocytic leukemia. Bone Marrow Transplant 1998; 22: 201–203.

    Article  CAS  Google Scholar 

Download references

Acknowledgements

Grant support: Deutsche Forschungsgemeinschaft FL 345/4–1 to CF and CMN.

Author information

Authors and Affiliations

  1. Division of Pediatric Hematology/Oncology, Department of Pediatric and Adolescent Medicine, University of Freiburg, Freiburg, Germany

    C Flotho, T Vraetz, C M Niemeyer & B Strahm

  2. Department of Pediatric Hematology/Oncology, University Children’s Hospital, Tübingen, Tübingen, Germany

    P Lang

Authors
  1. C Flotho
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. T Vraetz
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. P Lang
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. C M Niemeyer
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. B Strahm
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to C Flotho.

Ethics declarations

Competing interests

The authors declare no conflict of interest.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Flotho, C., Vraetz, T., Lang, P. et al. Successful double umbilical cord blood transplantation for relapsed juvenile myelomonocytic leukemia. Leukemia 27, 988–989 (2013). https://doi.org/10.1038/leu.2012.273

Download citation

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1038/leu.2012.273

Search

Advanced search

Quick links