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STEM CELL TRANSPLANTATION

Retrospective analysis of hematopoietic cell transplantation for blastic plasmacytoid dendritic cell neoplasm: conditioning intensity matters

Leukemia volume 37, pages 465–472 (2023)Cite this article

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Abstract

Blastic plasmacytoid dendritic cell neoplasia (BPDCN) is a rare myeloid malignancy with a generally poor prognosis. Although preliminary evidence suggests that hematopoietic cell transplantation (HCT) could improve outcome in patients with BPDCN, the individual contributions of conditioning and graft-versus-tumor (GVT) effects to HCT success are undefined. We present a retrospective study of 162 adult patients who underwent a first HCT (allogeneic 146, autologous 16) between 2009 and 2017, and were registered with the EBMT. Median age was 57 (range 20–73) years, and disease status at HCT was first complete remission (CR1) in 78%. Among patients receiving allogeneic HCT (alloHCT), myeloablative conditioning (MAC), reduced intensity conditioning (RIC) and in-vivo T-cell depletion (TCD) were used in 54%, 46%, and 59% respectively. Total body irradiation (TBI) was the conditioning backbone in 61% of MAC and 26% of RIC transplants. One-year overall survival (OS) and progression-free survival (PFS) rates were comparable after alloHCT and autologous HCT (autoHCT). Among alloHCT recipients, MAC with TBI significantly improved OS and PFS, independently of CR1, age, Karnofsky index and TCD. Accordingly, MAC (ideally based on TBI) should be preferred for alloHCT recipients with BPDCN. In patients who are not elegible for MAC alloHCT, autoHCT could be considered.

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Fig. 1: Outcome of allogeneic and autologous HCT.
Fig. 2: Type of conditioning and outcome of alloHCT.

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Data availability

The dataset analysed during the current study is available from the EBMT ProMISe databank on reasonable request to protect the privacy of the patients.

References

  1. Herling M, Teitell MA, Shen RR, Medeiros LJ, Jones D. TCL1 expression in plasmacytoid dendritic cells (DC2s) and the related CD4+ CD56+ blastic tumors of skin. Blood. 2003;101:5007–9.

    Article  CAS  Google Scholar 

  2. Laribi K, Baugier de Materre A, Sobh M, Cerroni L, Valentini CG, Aoki T, et al. Blastic plasmacytoid dendritic cell neoplasms: results of an international survey on 398 adult patients. Blood Adv. 2020;4:4838–48.

    Article  Google Scholar 

  3. Garnache-Ottou F, Vidal C, Biichle S, Renosi F, Poret E, Pagadoy M, et al. How should we diagnose and treat blastic plasmacytoid dendritic cell neoplasm patients? Blood Adv. 2019;3:4238–51.

    Article  CAS  Google Scholar 

  4. Martin-Martin L, Almeida J, Pomares H, Gonzalez-Barca E, Bravo P, Gimenez T, et al. Blastic plasmacytoid dendritic cell neoplasm frequently shows occult central nervous system involvement at diagnosis and benefits from intrathecal therapy. Oncotarget. 2016;7:10174–81.

    Article  Google Scholar 

  5. Taylor J, Haddadin M, Upadhyay VA, Grussie E, Mehta-Shah N, Brunner AM, et al. Multicenter analysis of outcomes in blastic plasmacytoid dendritic cell neoplasm offers a pretargeted therapy benchmark. Blood 2019;134:678–87.

    Article  CAS  Google Scholar 

  6. Reimer P, Rudiger T, Kraemer D, Kunzmann V, Weissinger F, Zettl A, et al. What is CD4+CD56+ malignancy and how should it be treated? Bone Marrow Transplant. 2003;32:637–46.

    Article  CAS  Google Scholar 

  7. Dietrich S, Andrulis M, Hegenbart U, Schmitt T, Bellos F, Martens UM, et al. Blastic plasmacytoid dendritic cell neoplasia (BPDC) in elderly patients: results of a treatment algorithm employing allogeneic stem cell transplantation with moderately reduced conditioning intensity. Biol Blood Marrow Transplant. 2011;17:1250–4.

    Article  Google Scholar 

  8. Roos-Weil D, Dietrich S, Boumendil A, Polge E, Bron D, Carreras E, et al. Stem cell transplantation can provide durable disease control in blastic plasmacytoid dendritic cell neoplasm: a retrospective study from the European Group for Blood and Marrow Transplantation. Blood. 2013;121:440–6.

    Article  CAS  Google Scholar 

  9. Aoki T, Suzuki R, Kuwatsuka Y, Kako S, Fujimoto K, Taguchi J, et al. Long-term survival following autologous and allogeneic stem cell transplantation for blastic plasmacytoid dendritic cell neoplasm. Blood. 2015;125:3559–62.

    Article  CAS  Google Scholar 

  10. Kharfan-Dabaja MA, Reljic T, Murthy HS, Ayala E, Kumar A. Allogeneic hematopoietic cell transplantation is an effective treatment for blastic plasmacytoid dendritic cell neoplasm in first complete remission: systematic review and meta-analysis. Clin Lymphoma Myeloma Leuk. 2018;18:703–9.e1

    Article  Google Scholar 

  11. Kharfan-Dabaja MA, Al Malki MM, Deotare U, Raj RV, El-Jurdi N, Majhail N, et al. Haematopoietic cell transplantation for blastic plasmacytoid dendritic cell neoplasm: a North American multicentre collaborative study. Br J Haematol. 2017;179:781–9.

    Article  CAS  Google Scholar 

  12. Yun S, Chan O, Kerr D, Vincelette ND, Idrees A, Mo Q, et al. Survival outcomes in blastic plasmacytoid dendritic cell neoplasm by first-line treatment and stem cell transplant. Blood Adv. 2020;4:3435–42.

    Article  Google Scholar 

  13. Bacigalupo A, Ballen K, Rizzo D, Giralt S, Lazarus H, Ho V, et al. Defining the intensity of conditioning regimens: working definitions. Biol Blood Marrow Transplant. 2009;15:1628–33.

    Article  Google Scholar 

  14. Pemmaraju N, Konopleva M. Approval of tagraxofusp-erzs for blastic plasmacytoid dendritic cell neoplasm. Blood Adv. 2020;4:4020–7.

    Article  CAS  Google Scholar 

  15. Leclerc M, Peffault de Latour R, Michallet M, Blaise D, Chevallier P, Rohrlich PS, et al. Can a reduced-intensity conditioning regimen cure blastic plasmacytoid dendritic cell neoplasm? Blood. 2017;129:1227–30.

    Article  CAS  Google Scholar 

  16. Kaloyannidis P, Zomas A, Paterakis G, Vadikoliou C, Mallouri D, Sakkas L, et al. GVL effect in plasmacytoid DC leukemia/lymphoma. Bone marrow Transplant. 2010;45:961–2.

    Article  CAS  Google Scholar 

  17. Ben Amor R, Hicheri Y, Pautas C, Jouault H, Kuentz M, Cordonnier C, et al. Successful non-myeloablative allogeneic HLA-identical stem cell transplantation for CD4/CD56 positive acute leukemia. Transplantation. 2007;84:1066–7.

    Article  Google Scholar 

  18. D’Souza A, Fretham C, Lee SJ, Arora M, Brunner J, Chhabra S, et al. Current use of and trends in hematopoietic cell transplantation in the United States. Biol Blood Marrow Transplant. 2020;26:e177–e82.

    Article  Google Scholar 

  19. Pemmaraju N, Lane AA, Sweet KL, Stein AS, Vasu S, Blum W, et al. Tagraxofusp in blastic plasmacytoid dendritic-cell neoplasm. N Engl J Med. 2019;380:1628–37.

    Article  CAS  Google Scholar 

  20. Montero J, Stephansky J, Cai T, Griffin GK, Cabal-Hierro L, Togami K, et al. Blastic plasmacytoid dendritic cell neoplasm is dependent on BCL2 and sensitive to venetoclax. Cancer Discov. 2017;7:156–64.

    Article  CAS  Google Scholar 

  21. Hourigan CS, Dillon LW, Gui G, Logan BR, Fei M, Ghannam J, et al. Impact of conditioning intensity of allogeneic transplantation for acute myeloid leukemia with genomic evidence of residual disease. J Clin Oncol. 2020;38:1273–83.

    Article  CAS  Google Scholar 

  22. Wang W, Khoury JD, Miranda RN, Jorgensen JL, Xu J, Loghavi S, et al. Immunophenotypic characterization of reactive and neoplastic plasmacytoid dendritic cells permits establishment of a 10-color flow cytometric panel for initial workup and residual disease evaluation of blastic plasmacytoid dendritic cell neoplasm. Haematologica. 2021;106:1047–55.

    Article  CAS  Google Scholar 

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Funding

This study is supported by EBMT.

Author information

Authors and Affiliations

  1. Department of Hematology, Oncology, and Clinical Immunology, Düsseldorf University Hospital, Düsseldorf, Germany

    Peter-Martin Bruch & Sascha Dietrich

  2. Department Medicine V, University of Heidelberg, Heidelberg, Germany

    Peter-Martin Bruch, Sascha Dietrich & Peter Dreger

  3. European Society for Blood and Marrow Transplantation (EBMT), Paris, France

    Sascha Dietrich, Herve Finel & Ariane Boumendil

  4. Medical University of Graz, Division of Hematology, Graz, Austria

    Hildegard Greinix

  5. University Hospital Magdeburg, Magdeburg, Germany

    Thomas Heinicke

  6. University Hospital Tübingen, Tübingen, Germany

    Wolfgang Bethge

  7. University Hospital Essen, Essen, Germany

    Dietrich Beelen

  8. University Hospital Augsburg, Augsburg, Germany

    Christoph Schmid

  9. University Hospital Frankfurt, Frankfurt am Main, Germany

    Hans Martin

  10. Ospedale Villa Sofia Cervello, BMT unit, Palermo, Italy

    Luca Castagna

  11. University of Cologne, Cologne, Germany

    Christof Scheid

  12. Klinikum Nürnberg, Paracelsus Medizinische Privatuniversität, Nürnberg, Germany

    Kerstin Schäfer-Eckart

  13. University Hospital Saarland, Homburg, Germany

    Jörg Bittenbring

  14. University Hospital Freiburg, Freiburg, Germany

    Jürgen Finke

  15. Rigshospitalet, Copenhagen, Denmark

    Henrik Sengeloev

  16. Hôpital Saint Antoine, Paris, France

    Mael Heiblig

  17. Erasmus MC Cancer Institute, Rotterdam, Netherlands

    Jan Cornelissen

  18. CHU, Nantes, France

    Patrice Chevallier

  19. Sorbonne University, Saint-Antoine Hospital, INSERM UMRs 938, Paris, France

    Mohamad Mohty

  20. University Hospital of Bristol, Bristol, UK

    Stephen Robinson

  21. St Bartholomew’s Hospital, London, UK

    Silvia Montoto

Authors
  1. Peter-Martin Bruch
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  2. Sascha Dietrich
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  18. Jan Cornelissen
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  22. Silvia Montoto
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  23. Peter Dreger
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Contributions

Conception and design: SD, PD. Collection and assembly of data: HF, AB. Data analysis: SD, P-MB, AB. Interpretation: All authors. Manuscript writing: First draft prepared by SD and PD. All authors helped with further developing the manuscript. Final approval of manuscript: All authors.

Corresponding author

Correspondence to Sascha Dietrich.

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Competing interests

PD: consultancy for AbbVie, AstraZeneca, Gilead, Janssen, Novartis, Riemser, Roche; speakers bureau for AbbVie, Gilead, Novartis, Riemser, Roche; research support from Neovii and Riemser. SD: consultancy for AbbVie, AstraZeneca, Gilead, Janssen, Novartis, Riemser, Roche; speakers bureau for AbbVie, Gilead, Novartis, Riemser, Roche; research support from Gilead and Janssen. P-MB: speakers bureau for AstraZeneca. TH: consultancy Eurocept, travel grants from Jazz, Gilead, Eurocept and Daichii. All other authors declare no conflict of interest.

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Bruch, PM., Dietrich, S., Finel, H. et al. Retrospective analysis of hematopoietic cell transplantation for blastic plasmacytoid dendritic cell neoplasm: conditioning intensity matters. Leukemia 37, 465–472 (2023). https://doi.org/10.1038/s41375-022-01782-z

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