Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2025 Dec;30(1):2458932.
doi: 10.1080/16078454.2025.2458932. Epub 2025 Feb 3.

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

Xuelian Jin  1 , Xushu Zhong  1 , Qinyu Liu  1 , Xinchuan Chen  1
Affiliations
Free article
Case Reports

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

Xuelian Jin et al. Hematology. 2025 Dec.
Free article

Abstract

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.

Keywords: Progressive multifocal leukoencephalopathy; allogeneic hematopoietic stem cell transplantation; human polyomavirus 2; programmed death receptor 1 inhibitor; treatment.

Plain language summary

This report describes a 35-year-old woman with acute myeloid leukemia, a high-risk malignancy, who underwent stem cell transplantation. A year later, she developed progressive multifocal leukoencephalopathy in the brain caused by infection with human polyomavirus 2. She was treated with an immune checkpoint inhibitor, sintilimab, that enhances the immune response against the virus. The patient received five treatments, with monitoring of her symptoms, brain scans, and measurement of virus levels in her spinal fluid. This showed improvement in symptoms, decreased virus levels, and reduced disease activity in the brain. Despite some side effects, such as an initial increase in brain lesions, these were manageable and did not lead to severe complications. This suggests that sintilimab may be a potential treatment for PML in patients following stem cell transplantation.

PubMed Disclaimer

Publication types

MeSH terms

Substances

LinkOut - more resources