Sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation: a case report

Background: Progressive multifocal leukoencephalopathy (PML) is characterized by demyelination in the central nervous system. It is caused by infection with human polyomavirus 2 and has a poor prognosis. Therapeutic strategies involve restoring immune function and/or discontinuing immunosuppressive...

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Main Authors: Xuelian Jin, Xushu Zhong, Qinyu Liu, Xinchuan Chen
Format: Article
Language:English
Published: Taylor & Francis Group 2025-12-01
Series:Hematology
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Online Access:https://www.tandfonline.com/doi/10.1080/16078454.2025.2458932
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author Xuelian Jin
Xushu Zhong
Qinyu Liu
Xinchuan Chen
author_facet Xuelian Jin
Xushu Zhong
Qinyu Liu
Xinchuan Chen
author_sort Xuelian Jin
collection DOAJ
description Background: Progressive multifocal leukoencephalopathy (PML) is characterized by demyelination in the central nervous system. It is caused by infection with human polyomavirus 2 and has a poor prognosis. Therapeutic strategies involve restoring immune function and/or discontinuing immunosuppressive treatment. Immune checkpoint inhibitors such as those targeting programmed death receptor-1 (PD-1) can alleviate PML by restoring T cell function. There are no case reports on the use of the PD-1 inhibitor, Sintilimab, for treating PML. Here, we report a case of successful treatment of PML with sintilimab following allogeneic hematopoietic stem cell transplantation.Case presentation: A 35-year-old woman with high-risk acute myeloid leukemia underwent allogeneic hematopoietic stem cell transplantation after induced remission and developed PML 12 months after transplantation. She received five courses of 100 mg every 4 weeks with monitoring by magnetic resonance imaging (MRI) and viral load in the cerebrospinal fluid, showing clinical improvement, resolution of neurological symptoms, and reduced viral load. MRI showed initial exacerbation of lesions but significant improvement after five courses of treatment. No graft-versus-host disease occurred, but manageable immune reconstitution inflammatory syndrome was observed.Conclusion: Sintilimab, a PD-1 inhibitor, might be used to treat PML in patients with hematologic malignancies undergoing allo-HSCT, which needs further investigation.
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series Hematology
spelling doaj-art-0b2464bd35a844fcb68282ce93510e232025-02-03T15:26:49ZengTaylor & Francis GroupHematology1607-84542025-12-0130110.1080/16078454.2025.2458932Sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation: a case reportXuelian Jin0Xushu Zhong1Qinyu Liu2Xinchuan Chen3Department of Hematology, West China Hospital, Sichuan University, Chengdu, the People’s Republic of ChinaDepartment of Hematology, West China Hospital, Sichuan University, Chengdu, the People’s Republic of ChinaDepartment of Hematology, West China Hospital, Sichuan University, Chengdu, the People’s Republic of ChinaDepartment of Hematology, West China Hospital, Sichuan University, Chengdu, the People’s Republic of ChinaBackground: Progressive multifocal leukoencephalopathy (PML) is characterized by demyelination in the central nervous system. It is caused by infection with human polyomavirus 2 and has a poor prognosis. Therapeutic strategies involve restoring immune function and/or discontinuing immunosuppressive treatment. Immune checkpoint inhibitors such as those targeting programmed death receptor-1 (PD-1) can alleviate PML by restoring T cell function. There are no case reports on the use of the PD-1 inhibitor, Sintilimab, for treating PML. Here, we report a case of successful treatment of PML with sintilimab following allogeneic hematopoietic stem cell transplantation.Case presentation: A 35-year-old woman with high-risk acute myeloid leukemia underwent allogeneic hematopoietic stem cell transplantation after induced remission and developed PML 12 months after transplantation. She received five courses of 100 mg every 4 weeks with monitoring by magnetic resonance imaging (MRI) and viral load in the cerebrospinal fluid, showing clinical improvement, resolution of neurological symptoms, and reduced viral load. MRI showed initial exacerbation of lesions but significant improvement after five courses of treatment. No graft-versus-host disease occurred, but manageable immune reconstitution inflammatory syndrome was observed.Conclusion: Sintilimab, a PD-1 inhibitor, might be used to treat PML in patients with hematologic malignancies undergoing allo-HSCT, which needs further investigation.https://www.tandfonline.com/doi/10.1080/16078454.2025.2458932Progressive multifocal leukoencephalopathyhuman polyomavirus 2allogeneic hematopoietic stem cell transplantationprogrammed death receptor 1 inhibitortreatment
spellingShingle Xuelian Jin
Xushu Zhong
Qinyu Liu
Xinchuan Chen
Sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation: a case report
Hematology
Progressive multifocal leukoencephalopathy
human polyomavirus 2
allogeneic hematopoietic stem cell transplantation
programmed death receptor 1 inhibitor
treatment
title Sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation: a case report
title_full Sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation: a case report
title_fullStr Sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation: a case report
title_full_unstemmed Sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation: a case report
title_short Sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation: a case report
title_sort sintilimab for treating progressive multifocal leukoencephalopathy caused by human polyomavirus 2 virus infection following allogeneic hematopoietic cell transplantation a case report
topic Progressive multifocal leukoencephalopathy
human polyomavirus 2
allogeneic hematopoietic stem cell transplantation
programmed death receptor 1 inhibitor
treatment
url https://www.tandfonline.com/doi/10.1080/16078454.2025.2458932
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