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

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 m...

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Bibliographic Details
Published in:International journal of hematology 2019-10, Vol.110 (4), p.506-511
Main Authors: Morishige, Satoshi, Nishi, Marina, Saruta, Hiroshi, Arakawa, Fumiko, Yamasaki, Yoshitaka, Oya, Shuki, Nakamura, Takayuki, Seki, Ritsuko, Yamaguchi, Maki, Aoyama, Kazutoshi, Mouri, Fumihiko, Osaki, Koichi, Ohshima, Koichi, Nagafuji, Koji
Format: Article
Language:English
Subjects:
Adult T cell leukemia
Aged
Allografts
Antibodies, Monoclonal, Humanized - administration & dosage
Antibodies, Monoclonal, Humanized - adverse effects
Antineoplastic Agents - administration & dosage
Antineoplastic Agents - adverse effects
Biopsy
Case Report
Case reports
Combined Modality Therapy
Cyclophosphamide
Cyclophosphamide - administration & dosage
Graft-versus-host reaction
Hematology
Hematopoietic Stem Cell Transplantation
Humans
Immune response
Immune system
Immunoglobulins, Intravenous - administration & dosage
Immunomodulation
Immunotherapy
Intravenous administration
Lenalidomide - administration & dosage
Lenalidomide - adverse effects
Lesions
Leukemia
Leukemia-Lymphoma, Adult T-Cell - therapy
Lymphocytes
Lymphocytes T
Lymphoma
Male
Medicine
Medicine & Public Health
Methylprednisolone
Methylprednisolone - administration & dosage
Oligodendrocyte-myelin glycoprotein
Oncology
Pulse Therapy, Drug
Recurrence
Skin
Skin diseases
Stem cell transplantation
Stem cells
Stevens-Johnson Syndrome - drug therapy
Stevens-Johnson Syndrome - etiology
Targeted cancer therapy
Therapy
Toxic epidermal necrolysis
Transplantation
Transplantation, Haploidentical
Transplants & implants
Treatment Outcome
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Summary: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.
ISSN:0925-5710
1865-3774
DOI:10.1007/s12185-019-02675-1