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Complete response following toxic epidermal necrolysis in relapsed adult T cell leukemia/lymphoma after haploidentical stem cell transplantation

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Abstract

Allogeneic hematopoietic cell transplantation (allo-HSCT) is considered the curative treatment option in patients with aggressive adult T cell leukemia/lymphoma (ATLL), but the treatment of relapse after allo-HSCT remains a major challenge. We report a case of ATLL that was treated with sequential mogamulizumab (MOG) and lenalidomide (LEN) for early relapse after allo-HSCT. A 73-year-old Japanese male with acute-type ATLL underwent haploidentical-HSCT with post-transplant cyclophosphamide. He attained a complete response. However, ATLL relapse was diagnosed by biopsy of skin lesions that appeared on day 67. Discontinuation of immunosuppressant therapy alone did not result in improvement of ATLL, however, the skin lesions disappeared after an immune response was induced by sequential MOG and LEN. Following MOG and LEN, very serious toxic epidermal necrolysis (TEN) developed requiring high-dose intravenous immunoglobulin and methylprednisolone pulse therapy. Although graft-versus-host disease exacerbated and progressed to TEN, a complete response was achieved after successful treatment of TEN. These agents may thus enhance anti-ATLL activity by immune modulation. Further investigation is necessary to determine the optimal use of MOG and LEN in relapsed ATLL after allo-HSCT.

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References

  1. Miyoshi H, Ohshima K. Epidemiology of malignant lymphoma and recent progress in research on adult T-cell leukemia/lymphoma in Japan. Int J Hematol. 2018;107:420–7.

    Article  CAS  PubMed  Google Scholar 

  2. Katsuya H, Ishitsuka K, Utsunomiya A, Hanada S, Eto T, Moriuchi Y, et al. Treatment and survival among 1594 patients with ATL. Blood. 2015;126:2570–7.

    Article  CAS  PubMed  Google Scholar 

  3. Kanda J, Hishizawa M, Utsunomiya A, Taniguchi S, Eto T, Moriuchi Y, et al. Impact of graft-versus-host disease on outcomes after allogeneic hematopoietic cell transplantation for adult T-cell leukemia: a retrospective cohort study. Blood. 2012;119:2141–8.

    Article  CAS  PubMed  Google Scholar 

  4. Ishida T, Joh T, Uike N, Yamamoto K, Utsunomiya A, Yoshida S, et al. Defucosylated anti-CCR4 monoclonal antibody (KW-0761) for relapsed adult T-cell leukemia-lymphoma: a multicenter phase II study. J Clin Oncol. 2012;30:837–42.

    Article  CAS  PubMed  Google Scholar 

  5. Fuji S, Inoue Y, Utsunomiya A, Moriuchi Y, Uchimaru K, Choi I, et al. Pretransplantation anti-CCR5 antibody mogamulizumab against adult T-cell leukemia/lymphoma is associated with significantly increased risks of severe and corticosteroid-refractory graft-versus-host disease, nonrelapse mortality, and overall mortality. J Clin Oncol. 2016;34:3426–33.

    Article  PubMed  Google Scholar 

  6. Sugio T, Kato K, Aoki T, Ohta T, Saito N, Yoshida S, et al. Mogamulizumab treatment prior to allogeneic hematopoietic stem cell transplantation induces severe acute graft-versus-host disease. Biol Blood Marrow Transplant. 2016;22:1608–14.

    Article  CAS  PubMed  Google Scholar 

  7. Ito Y, Miyamoto T, Chong Y, Aoki T, Kato K, Akashi K, et al. Successful treatment with anti-CC chemokine receptor 4 MoAb of relapsed adult T-cell leukemia/lymphoma after umbilical cord blood transplantation. Bone Marrow Transplant. 2013;48:998–9.

    Article  CAS  PubMed  Google Scholar 

  8. Ando T, Kojima K, Sano H, Kidoguchi K, Kusaba K, Yoshimura M, et al. Successful treatment of post-transplant relapsed adult T cell leukemia after cord blood transplantation with low-dose, short-term lenalidomide. Leuk Lymphoma. 2018;59:2474–7.

    Article  CAS  PubMed  Google Scholar 

  9. Shimoyama M. Diagnostic criteria and classification of clinical subtypes of adult T-cell leukaemia-lymphoma. A report from the Lymphoma Study Group (1984-87). Br J Haematol. 1991;79:428–37.

    Article  CAS  PubMed  Google Scholar 

  10. Ichikawa A, Miyoshi H, Yamauchi T, Arakawa F, Kawano R, Muta H, et al. Composite lymphoma of peripheral T-cell lymphoma and Hodgkin lymphoma, mixed cellularity type; pathological and molecular analysis. Pathol Int. 2017;67:194–201.

    Article  CAS  PubMed  Google Scholar 

  11. Fukushima T, Miyazaki Y, Honda S, Kawano F, Moriuchi Y, Masuda M, et al. Allogeneic hematopoietic stem cell transplantation provides sustained long-term survival for patients with adult T-cell leukemia/lymphoma. Leukemia. 2005;19:829–34.

    Article  CAS  PubMed  Google Scholar 

  12. Ishida T, Hishizawa M, Kato K, Tanosaki R, Fukuda T, Taniguchi S, et al. Allogeneic hematopoietic stem cell transplantation for adult T-cell leukemia-lymphoma with special emphasis on preconditioning regimen: a nationwide retrospective study. Blood. 2012;120:1734–41.

    Article  CAS  PubMed  Google Scholar 

  13. Kato K, Uike N, Wake A, Yoshimitsu M, Tobai T, Sawayama Y, et al. The outcome and characteristics of patients with relapsed adult T cell leukemia/lymphoma after allogeneic hematopoietic stem cell transplantation. Hematol Oncol. 2019;37:54–61.

    Article  CAS  PubMed  Google Scholar 

  14. Umino A, Nakagawa M, Utsunomiya A, Tsukasaki K, Taira N, Katayama N, et al. Clonal evolution of adult T-cell leukemia/lymphoma takes place in the lymph nodes. Blood. 2011;117:5473–8.

    Article  CAS  PubMed  Google Scholar 

  15. Aoki S, Firouzi S, Lopez Y, Yamochi T, Nakano K, Uchimaru K, et al. Transition of adult T-cell leukemia/lymphoma clones during clinical progression. Int J Hematol. 2016;104:330–7.

    Article  CAS  PubMed  Google Scholar 

  16. Shiratori S, Ohhigashi H, Ito S, Kudo K, Adachi M, Minamimoto T, et al. Late onset toxic epidermal necrolysis induced by mogamulizumab, an anti-CC chemokine receptor 4 antibody for the treatment of adult T-cell leukaemia/lymphoma. Hematol Oncol. 2017;35:138–40.

    Article  PubMed  Google Scholar 

  17. Ishida T, Ito A, Sato F, Kusumoto S, Iida S, Inagaki H, et al. Stevens-Johnson Syndrome associated with mogamulizumab treatment of adult T-cell leukemia/lymphoma. Cancer Sci. 2013;104:647–50.

    Article  CAS  PubMed  Google Scholar 

  18. Honda T, Hishizawa M, Kataoka TR, Ohmori K, Takaori-Kondo A, Miyachi Y, et al. Stevens-Johnson syndrome associated with mogamulizumab-induced deficiency of regulatory T cells in an adult T-cell leukaemia patient. Acta Derm Venereol. 2015;95:606–7.

    Article  PubMed  Google Scholar 

  19. Takeda H, Mitsuhashi Y, Kondo S, Kato Y, Tajima K. Toxic epidermal necrolysis possibly linked to hyperacute graft-versus-host disease after allogeneic bone marrow transplantation. J Dermatol. 1997;24:635–41.

    Article  CAS  PubMed  Google Scholar 

  20. Naik H, Lockwood S, Saavedra A. A pilot study comparing histological and immunophenotypic patterns in stage 4 skin graft vs host disease from toxic epidermal necrolysis. J Cutan Pathol. 2017;44:857–60.

    Article  PubMed  Google Scholar 

  21. Castaneda CP, Brandenburg NA, Bwire R, Burton GH, Zeldis JB. Erythema multiforme/Stevens-Johnson syndrome/toxic epidermal necrolysis in lenalidomide-treated patients. J Clin Oncol. 2009;27:156–7.

    Article  PubMed  Google Scholar 

  22. Kneppers E, van der Holt B, Kersten MJ, Zweegman S, Meijer E, Huls G, et al. Lenalidomide maintenance after nonmyeloablative allogeneic stem cell transplantation in multiple myeloma is not feasible: results of the HOVON 76 Trial. Blood. 2011;118:2413–9.

    Article  CAS  PubMed  Google Scholar 

  23. Alsina M, Becker PS, Zhong X, Adams A, Hari P, Rowley S, et al. Lenalidomide maintenance for high-risk multiple myeloma after allogeneic hematopoietic cell transplantation. Biol Blood Marrow Transplant. 2014;20:1183–9.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  24. Wolschke C, Stubig T, Hegenbart U, Schonland S, Heinzelmann M, Hildebrandt Y, et al. Postallograft lenalidomide induces strong NK cell-mediated antimyeloma activity and risk for T cell-mediated GvHD: Results from a phase I/II dose-finding study. Exp Hematol. 2013;41(134–42):e3.

    Google Scholar 

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Correspondence to Koji Nagafuji.

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Morishige, S., Nishi, M., Saruta, H. et al. Complete response following toxic epidermal necrolysis in relapsed adult T cell leukemia/lymphoma after haploidentical stem cell transplantation. Int J Hematol 110, 506–511 (2019). https://doi.org/10.1007/s12185-019-02675-1

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  • DOI: https://doi.org/10.1007/s12185-019-02675-1

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