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Allogeneic hematopoietic stem cell transplantation for adult T-cell leukemia/lymphoma with HTLV-1-associated myelopathy

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Abstract

Allogeneic hematopoietic stem cell transplantation (allo-HSCT) can be curative for adult T-cell leukemia/lymphoma (ATL), but comorbidities increase transplant-related mortality. Here we report the outcome of allo-HSCT in a patient with ATL with human T-cell leukemia virus type I (HTLV-1)-associated myelopathy–tropical spastic paraparesis (HAM/TSP). A 48-year-old man was diagnosed with HAM/TSP and started prednisolone therapy. Ten years later, he developed lymphoma-type ATL. At the diagnosis of ATL, Osame’s Motor Disability Score (OMDS) was 4. When prednisolone was gradually tapered and stopped following chemotherapy for ATL, HAM/TSP symptoms recurred (OMDS 7). Bone marrow transplantation from a human leukocyte antigen allele 8/8 matched unrelated donor was performed while ATL was in partial remission. Neutrophil engraftment with complete donor chimerism was achieved on day 19 after allo-HSCT. Mild gait improvement (OMDS 5) was observed on day 30. Although ATL relapsed on day 275, progression of HAM/TSP symptoms was not observed. Furthermore, there was no clear progression of HAM/TSP symptoms after donor lymphocyte infusions. The outcome of this case suggests that ATL patients with HAM/TSP tolerate allo-HSCT and donor lymphocyte infusions.

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Acknowledgements

We appreciate the contribution of Naoko Yagishita, Ph.D. (Department of Rare Diseases Research, Institute of Medical Science, St. Marianna University School of Medicine, Kawasaki, Japan.) to the examination and analysis of samples.

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Authors and Affiliations

  1. Department of Hematology, Atomic Bomb Disease and Hibakusha Medicine Unit, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan

    Hikaru Sakamoto, Masahiko Chiwata, Machiko Fujioka & Yasushi Miyazaki

  2. Department of Hematology, Nagasaki University Hospital, Nagasaki, Japan

    Hikaru Sakamoto, Yasushi Sawayama, Azusa Kojima, Machiko Fujioka, Makiko Horai, Yoshitaka Imaizumi & Yasushi Miyazaki

  3. Department of Hematology, Atomic Bomb Disease and Hibakusha Medicine Unit, Atomic Bomb Disease Institute, Nagasaki University, 1-12-4 Sakamoto, Nagasaki, 852-8523, Japan

    Hidehiro Itonaga, Tomoko Hata & Yasushi Miyazaki

  4. Department of Neurology and Strokology, Nagasaki University Hospital, Nagasaki, Japan

    Hiroko Kitanosono, Teiichiro Miyazaki & Hirokazu Shiraishi

  5. Department of Hematology, National Hospital Organization Nagasaki Medical Center, Omura, Japan

    Shinichiro Yoshida

  6. Division of Neurology, Department of Internal Medicine, St. Marianna University School of Medicine, Kawasaki, Japan

    Yoshihisa Yamano

  7. Department of Rare Diseases Research, Institute of Medical Science, St. Marianna University School of Medicine, Kawasaki, Japan

    Yoshihisa Yamano

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  1. Hikaru Sakamoto
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  2. Hidehiro Itonaga
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Contributions

HS (Sakamoto), HI, and YM designed this project and wrote the manuscript: HS (Sakamoto), HI, YS, AK, MC, MF, HK, MH, TM, HS (Shiraishi), YI, SY, TH, and YM performed clinical management; YY analyzed the samples; all authors interpreted the results, and reviewed and approved the manuscript.

Corresponding author

Correspondence to Hidehiro Itonaga.

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Conflicts of interest

Y. Miyazaki has received honoraria from Otsuka Pharma, Novartis Pharma, Nippon-Shinyaku Co, Sumitomo Dainippon Pharma, and Kyowa Hakko Kirin Co and research funding from Chugai Pharma. S. Yoshida has received grants from Chugai Pharma and Kyowa-Kirin.

Ethical approval

This study was approved by the Ethics Committees of Nagasaki University Hospital (approval no. 18061816 and 18061804) and St. Marianna University School of Medicine (approval no. 1646) at which this study was organized.

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Sakamoto, H., Itonaga, H., Sawayama, Y. et al. Allogeneic hematopoietic stem cell transplantation for adult T-cell leukemia/lymphoma with HTLV-1-associated myelopathy. Int J Hematol 113, 765–769 (2021). https://doi.org/10.1007/s12185-020-03075-6

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  • DOI: https://doi.org/10.1007/s12185-020-03075-6

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