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EBMT prospective observational study on allogeneic hematopoietic stem cell transplantation in T-prolymphocytic leukemia (T-PLL)

Bone Marrow Transplantation volume 54, pages 1391–1398 (2019)Cite this article

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Abstract

Preliminary data suggest that allogeneic stem cell transplantation (allo-SCT) may be effective in T-prolymphocytic leukemia (T-PLL). The purpose of the present observational study was to assess the outcome of allo-SCT in patients aged 65 years or younger with a centrally confirmed diagnosis of T-PLL. Patients were consecutively registered with the EBMT at the time of transplantation and followed by routine EBMT monitoring but with an extended dataset. Between 2007 and 2012, 37 evaluable patients (median age 56 years) were accrued. Pre-treatment contained alemtuzumab in 95% of patients. Sixty-two percent were in complete remission (CR) at the time of allo-SCT. Conditioning contained total body irradiation with 6 Gy or more (TBI6) in 30% of patients. With a median follow-up of 50 months, the 4-year non-relapse mortality, relapse incidence, progression-free (PFS) and overall survival were 32, 38, 30 and 42%, respectively. By univariate analysis, TBI6 in the conditioning was the only significant predictor for a low relapse risk, and an interval between diagnosis and allo-SCT of more than 12 months was associated with a lower NRM. This study confirms for the first time prospectively that allo-SCT can provide long-term disease control in a sizable albeit limited proportion of patients with T-PLL.

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References

  1. Dearden CE, Matutes E, Cazin B, Tjonnfjord GE, Parreira A, Nomdedeu B. et al. High remission rate in T-cell prolymphocytic leukemia with CAMPATH-1H. Blood. 2001;98:1721–6.

    Article  CAS  Google Scholar 

  2. Dearden CE, Khot A, Else M, Hamblin M, Grand E, Roy A, et al. Alemtuzumab therapy in T-cell prolymphocytic leukaemia: comparing efficacy in a series treated intravenously and a study piloting the subcutaneous route. Blood. 2011;118:5799–802.

    Article  CAS  Google Scholar 

  3. Hopfinger G, Busch R, Pflug N, Weit N, Westermann A, Fink AM, et al. Sequential chemoimmunotherapy of fludarabine, mitoxantrone, and cyclophosphamide induction followed by alemtuzumab consolidation is effective in T-cell prolymphocytic leukemia. Cancer. 2013;119:2258–67.

    Article  CAS  Google Scholar 

  4. Krishnan B, Else M, Tjonnfjord GE, Cazin B, Carney D, Carter J, et al. Stem cell transplantation after alemtuzumab in T-cell prolymphocytic leukaemia results in longer survival than after alemtuzumab alone: a multicentre retrospective study. Br J Haematol. 2010;149:907–10.

    Article  Google Scholar 

  5. Kalaycio ME, Kukreja M, Woolfrey AE, Szer J, Cortes J, Maziarz RT, et al. Allogeneic hematopoietic cell transplant for prolymphocytic leukemia. Biol Blood Marrow Transplant. 2009;16:543–7.

    Article  Google Scholar 

  6. Wiktor-Jedrzejczak W, Dearden C, de WL, van BA, Brinch L, Leblond V, et al. Hematopoietic stem cell transplantation in T-prolymphocytic leukemia: a retrospective study from the European Group for Blood and Marrow Transplantation and the Royal Marsden Consortium. Leukemia. 2012;26:972–6.

    Article  CAS  Google Scholar 

  7. Guillaume T, Beguin Y, Tabrizi R, Nguyen S, Blaise D, Deconinck E, et al. Allogeneic hematopoietic stem cell transplantation for T-prolymphocytic leukemia: a report from the French society for stem cell transplantation (SFGM-TC). Eur J Haematol. 2015;94:265–9.

    Article  Google Scholar 

  8. Dearden C. Management of prolymphocytic leukemia. Hematol Am Soc Hematol Educ Program. 2015;2015:361–7.

    Article  Google Scholar 

  9. 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 

  10. Boidol B, Kornauth C, van der Kouwe E, Prutsch N, Kazianka L, Gültekin S, et al. First-in-human response of BCL-2 inhibitor venetoclax in T-cell prolymphocytic leukemia. Blood. 2017;130:2499–503.

    Article  CAS  Google Scholar 

  11. Kiel MJ, Velusamy T, Rolland D, Sahasrabuddhe AA, Chung F, Bailey NG, et al. Integrated genomic sequencing reveals mutational landscape of T-cell prolymphocytic leukemia. Blood. 2014;124:1460–72.

    Article  CAS  Google Scholar 

  12. Weston VJ, Oldreive CE, Skowronska A, Oscier DG, Pratt G, Dyer MJ, et al. The PARP inhibitor olaparib induces significant killing of ATM-deficient lymphoid tumor cells in vitro and in vivo. Blood. 2010;116:4578–87.

    Article  CAS  Google Scholar 

  13. Stelljes M, Bornhauser M, Kroger M, Beyer J, Sauerland MC, Heinecke A, et al. Conditioning with 8-Gy total body irradiation and fludarabine for allogeneic hematopoietic stem cell transplantation in acute myeloid leukemia. Blood. 2005;106:3314–21.

    Article  CAS  Google Scholar 

  14. Hahn M, Böttcher S, Dietrich S, Hegenbart U, Rieger M, Stadtherr P, et al. Allogeneic hematopoietic stem cell transplantation for poor-risk chronic lymphocytic leukemia: Dissecting immune-modulating strategies for disease eradication and treatment of relapse. Bone Marrow Transplant. 2015;50:1279–85.

    Article  CAS  Google Scholar 

  15. Sellner L, Brüggemann M, Schlitt M, Knecht H, Herrmann D, Reigl T, et al. Graft-versus-leukemia effects in T-prolymphocytic leukemia: evidence from MRD kinetics and TCR repertoire analyses. Bone Marrow Transplant. 2017;52:544–51.

    Article  CAS  Google Scholar 

  16. Jain P, Aoki E, Keating M, Wierda WG, O'Brien S, Gonzalez GN, et al. Characteristics, outcomes, prognostic factors and treatment of patients with T-cell prolymphocytic leukemia (T-PLL). Ann Oncol. 2017;28:1554–9.

    Article  CAS  Google Scholar 

  17. Andersson EI, Pützer S, Yadav B, Dufva O, Khan S, He L, et al. Discovery of novel drug sensitivities in T-PLL by high-throughput ex vivo drug testing and mutation profiling. Leukemia. 2018;32:774–87.

    Article  CAS  Google Scholar 

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Acknowledgements

We acknowledge all collaborating EBMT Investigators and Institutions that contributed cases to this study as listed in the Appendix.

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Authors and Affiliations

  1. Medical University of Warsaw, Warsaw, Poland

    W. Wiktor-Jedrzejczak & J. Drozd-Sokolowska

  2. EBMT Statisticians, Leiden, The Netherlands

    D. J. Eikema

  3. Data Office Leiden, Leiden, The Netherlands

    J. Hoek & A. van Biezen

  4. Royal Marsden Hospital, London, UK

    M. Potter

  5. Universitätsklinikum Göttingen, Göttingen, Germany

    G. Wulf

  6. University of Heidelberg, Heidelberg, Germany

    L. Sellner & P. Dreger

  7. Karolinska University Hospital, Karolinska Institutet, Stockholm, Sweden

    P. Ljungman

  8. CHU Nantes, Nantes, France

    P. Chevallier

  9. HUCH Comprehensive Cancer Center, Helsinki, Finland

    L. Volin

  10. Medical Park Hospitals, Antalya, Turkey

    Y. Koc

  11. Robert-Bosch-Krankenhaus, Stuttgart, Germany

    S. Martin

  12. Universitätsklinikum Ulm, Ulm, Germany

    D. Bunjes

  13. Hospital Clinic, Barcelona, Spain

    M. Rovira

  14. Turku University Hospital, Turku, Finland

    M. Itälä-Remes

  15. Univ. ‘La Sapienza’, Rome, Italy

    R. Foá

  16. Hopital Jean Minjoz, Besancon, France

    E. Deconinck

  17. Oslo University Hospital, Rikshospitalet, Oslo, Norway

    T. Gedde-Dahl

  18. Erasmus MC Cancer Institute, Rotterdam, Netherlands

    J. Cornelissen

  19. Freeman Hospital, Newcastle, UK

    M. Collin

  20. Deutsche Klinik für Diagnostik, KMT Zentrum, Wiesbaden, Germany

    A. Brecht

  21. Clatterbridge Cancer centre, Liverpool, UK

    A. Patel

  22. University Medical Center Groningen, Groningen, Netherlands

    M. de Groot

  23. St. Istvan & St. Laszlo Hospital, Budapest, Hungary

    P. Reményi

  24. Chaim Sheba Medical Center, Tel-Hashomer, Israel

    A. Nagler

  25. University of Freiburg, Freiburg, Germany

    J. Finke

  26. CHRU Limoges, Limoges, France

    P. Turlure

  27. Tor Vergata University, Rome, Italy

    S. Iacobelli

  28. Universitätsklinikum Dresden, Dresden, Germany

    J. Schetelig

  29. University Hospital Eppendorf, Hamburg, Germany

    N. Kröger

Authors
  1. W. Wiktor-Jedrzejczak
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  2. J. Drozd-Sokolowska
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  29. A. van Biezen
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  30. J. Schetelig
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  31. N. Kröger
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  32. P. Dreger
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Correspondence to W. Wiktor-Jedrzejczak.

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Supplementary information

41409_2019_448_MOESM1_ESM.doc

Study Protocol. Allogeneic stem cell transplantation for the treatment of T cell prolymphocytic leukaemia: A prospective observational EBMT study

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Wiktor-Jedrzejczak, W., Drozd-Sokolowska, J., Eikema, D.J. et al. EBMT prospective observational study on allogeneic hematopoietic stem cell transplantation in T-prolymphocytic leukemia (T-PLL). Bone Marrow Transplant 54, 1391–1398 (2019). https://doi.org/10.1038/s41409-019-0448-x

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