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.
References
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
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
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
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
Margo CM, Porcu P, Schaefer J et al (2010) Cutaneous CD4+CD56+hematologic malignancies. J Am Acad Dermatol 63:292–308
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
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
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
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
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
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
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
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
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
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
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
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
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
Gambichler T, Pantelaki I, Stücker M (2013) Childhood blastic plasmacytoid dendritic cell neoplasm treated with allogenic stem cell transplantation. Pediatr Dermatol 30:142–144
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
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
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
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
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
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
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
Corresponding author
Rights and permissions
About this article
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
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12288-015-0543-0