Skip to main content

Advertisement

Log in

Effective Treatment of a Childhood Blastic Plasmacytoid Dendritic Cell Neoplasm with a Cutaneous Tumor Alone by Stem Cell Transplantation with Reduced Intensity Conditioning

  • Case Report
  • Published:
Indian Journal of Hematology and Blood Transfusion Aims and scope Submit manuscript

Abstract

Pediatric blastic plasmacytoid dendritic cell neoplasm (BPDCN) is a rare hematological malignancy that has an extremely poor prognosis despite the use of intensive chemotherapy. Recently, treatment of BPDCN with bone marrow transplantation (BMT) using myeloablative conditioning has been reported to increase survival in adults. We report a 9-year-old girl with cutaneous BPDCN who was successfully treated with combination chemotherapy followed by BMT using reduced intensity conditioning (RIC), without any adverse complications. The success of this treatment regimen suggests that BMT with RIC may be a feasible option for treating children with cutaneous BPDCN.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3

References

  1. Vardiman JW, Thiele J, Arber DA et al (2009) The 2008 revision of the World Health prganization (WHO) classification of myeloid neoplasms and acute leukemia: rationale and important changes. Blood 114:937–951

    Article  CAS  PubMed  Google Scholar 

  2. Petrella T, Bagot M, Willemze R et al (2005) Blastic NK cell lymphomas (agranular CD4+ CD56+ hematodermic neoplasms). Am J Clin Pathol 123:662–675

    Article  PubMed  Google Scholar 

  3. Roos-Weil D, Dietrich S, Boumendil A et al (2013) Stem cell transplantation can provide durable disease control in blastic plasmacytoid dendritic cell neoplasm (BPDCN). Blood 121:440–446

  4. Yamaguchi M, Maekawa M, Nakamura Y et al (2005) Long-term remission of blastic natural killer-cell lymphoma after autologous peripheral blood stem-cell transplantation. Am J Hematol 80:124–127

    Article  PubMed  Google Scholar 

  5. Margo CM, Porcu P, Schaefer J et al (2010) Cutaneous CD4+CD56+hematologic malignancies. J Am Acad Dermatol 63:292–308

    Article  Google Scholar 

  6. Dalle S, Beylot-Barry M, Bagot M et al (2010) Blastic plasmacytoid dendritic cell neoplasm: is transplantation the treatment of choice? Br J Dermatol 162:74–79

    Article  CAS  PubMed  Google Scholar 

  7. Chicha L, Jarrossay D, Manz MG (2004) Clonal type I interferon-producing and dendritic cell precursors are contained in both human lymphoid and myeloid progenitor populations. J Exp Med 200:1519–1524

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  8. Herling M, Jones D (2007) CD4+/CD56+ hematodermic tumor: the features of an evolving entity and its relationship to dendritic cells. Am J Clin Pathol 127:687–700

    Article  PubMed  Google Scholar 

  9. Cota C, Vale E, Viana I et al (2010) Cutaneous manifestations of blastic plasmacytoid dendritic cell neoplasm-morphologic and phenotypic variability in a series of 33 patients. Am J Surg Pathol 34:75–87

    Article  PubMed  Google Scholar 

  10. Rossi JG, Felice MS, Bernasconi AR et al (2006) Acute leukemia of dendritic cell lineage in childhood: incidence, biological characteristics and outcome. Leuk Lymphoma 47:715–725

    Article  PubMed  Google Scholar 

  11. Borchiellini D, Ghibaudo N, Mounier N et al (2013) Blastic plasmacytoid dendritic cell neoplasm: a report of four cases and review of the literature. J Eur Acad Dermatol Venereol 27:1176–1181

    Article  CAS  PubMed  Google Scholar 

  12. Murashige N, Kami M, Kishi Y et al (2005) Allogeneic haematopoietic stem cell transplantation as a promising treatment for natural killer-cell neoplasms. Br J Haematol 130:561–567

    Article  PubMed  Google Scholar 

  13. Suzuki R, Suzumiya J, Nakamura S et al (2006) Hematopoietic stem cell transplantation for natural killer-cell lineage neoplasms. Bone Marrow Transplant 37:425–431

    Article  CAS  PubMed  Google Scholar 

  14. Jegalian AG, Buxbaum NP, Facchetti F et al (2010) Blastic plasmacytoid dendritic cell neoplasm in children: diagnostic features and clinical implications. Haematologica 95:1873–1879

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  15. Sakashita K, Saito S, Yanagisawa R et al (2013) Usefulness of Allogeneic hematopoietic stem cell transplantation in first complete remission for pediatric blastic plasmacytoid dendritic cell neoplasm with skin involvement: a case report and review of literature. Pediatr Blood Cancer 60:140–142

    Article  Google Scholar 

  16. Rauh MJ, Rahman F, Good D et al (2012) Blastic plasmacytoid dendritic cell neoplasm with leukemic presentation, lacking cutaneous involvement: case series and literature. Leuk Res 36:81–86

    Article  CAS  PubMed  Google Scholar 

  17. Hashikawa K, Niino D, Yasumoto S et al (2012) Clinicopathological features and prognostic significance of CXCL12 in blastic plasmacytoid dendritic cell neoplasm. J Am Acad Dermatol 66:278–291

  18. Karube K, Ohshima K, Tsuchiya T et al (2003) Non-B, non-T neoplasms with lymphoblast morphology: further clarification and classification. Am J Surg Pathol 27:1366–1374

  19. Gambichler T, Pantelaki I, Stücker M (2013) Childhood blastic plasmacytoid dendritic cell neoplasm treated with allogenic stem cell transplantation. Pediatr Dermatol 30:142–144

  20. Dijkman R, van Doorn R, Szuhai K et al (2007) Gene-expression profiling and array-based CGH classify CD4+ CD56+ hematodermic neoplasm and cutaneous myelomonocytic leukemia as distinct disease entities. Blood 109:1720–1727

  21. Julia F, Petrella T, Beylot-Barry M et al (2013) Blastic plasmacytoid dendritic cell neoplasm: clinical features in 90 patients. Br J Dermatol 169:579–586

  22. Slavin S, Nagler A, Naparstek E et al (1998) Nonmyeloablative stem cell transplantation and cell therapy as an alternative to conventional bone marrow transplantation with lethal cytoreduction for the treatment of malignant and nonmalignant hematologic diseases. Blood 91:756–763

  23. Bitan M, He W, Zhang MJ et al (2014) Transplantation for children with acute myeloid leukemia: a comparison of outcomes with reduced intensity and myeloablative regimens. Blood 123:1615–1620

  24. Sorror ML, Sandmaier BM, Storer BE et al (2007) Comorbidity and disease status based risk stratification of outcomes among patients with acute myeloid leukemia or myelodysplasia receiving allogeneic hematopoietic cell transplantation. J Clin Oncol 25:4246–4254

  25. Pulsipher MA, Boucher KM, Wall D et al (2009) Reduced-intensity allogeneic transplantation in pediatric patients ineligible for myeloablative therapy: results of the Pediatric Blood and Marrow Transplant Consortium Study ONC0313. Blood 114:1429–1436

Download references

Acknowledgments

We thank Atsuko Nakagawa, MD, Department of pathology, National Center for Child Health and Development, Tokyo, Japan, for the histological examination and the immunohistochemical staining.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Maiko Shimomura.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Shimomura, M., Asano, T., Furue, A. et al. Effective Treatment of a Childhood Blastic Plasmacytoid Dendritic Cell Neoplasm with a Cutaneous Tumor Alone by Stem Cell Transplantation with Reduced Intensity Conditioning. Indian J Hematol Blood Transfus 32 (Suppl 1), 26–31 (2016). https://doi.org/10.1007/s12288-015-0543-0

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12288-015-0543-0

Keywords

Navigation