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Efficacy of filgrastim therapy for patients with progressive multifocal leukoencephalopathy: Two case reports
Hanako Aoki1, Ryunosuke Matsumura1, Yoichiro Nishida1
1Department of Neurology and Neurological Science, Institute of Science Tokyo hospital, Tokyo, Japan.
Abstract:
Progressive multifocal leukoencephalopathy (PML) is a life-threatening central nervous system condition caused by reactivation of JC virus in individuals with immunosuppression. Although PML typically develops in patients with human immunodeficiency virus (HIV), cases in individuals without HIV have been increasing in recent years. Non-HIV-associated PML has a particularly poor prognosis, high mortality rates, and few therapeutic options. Here, we present the cases of two patients with non-HIV-, nondrug-associated PML who achieved good clinical outcomes after treatment with filgrastim (granulocyte colony-stimulating factor [G-CSF]). One patient had a diagnosis of Good syndrome, a rare primary immunodeficiency disease, whereas the other presented with lymphopenia and a mild decrease in CD4 T cells. After G-CSF administration, both patients showed decreases in CSF JC virus loads and improvements on magnetic resonance images. Given these findings, we propose that, by stimulating innate immunity and activating anti-JC viral immune surveillance, G-CSF may be useful as an immune-enhancing therapy for patients with non-HIV-associated PML.
Insights
Two patients with rare, non-HIV-associated Progressive Multifocal Leukoencephalopathy (PML) improved after treatment with granulocyte colony-stimulating factor (G-CSF). This suggests G-CSF may be a viable immune-enhancing therapy for PML.
Area of Science:
- Neuroimmunology
- Virology
- Immunology
Background:
- Progressive Multifocal Leukoencephalopathy (PML) is a severe demyelinating disease of the central nervous system caused by JC virus reactivation.
- While often associated with Human Immunodeficiency Virus (HIV), non-HIV-associated PML is increasing and carries a poor prognosis.
- Limited therapeutic options exist for non-HIV-associated PML, necessitating novel treatment strategies.
Observation:
- Two patients with non-HIV-, non-drug-associated PML, one with Good syndrome and another with lymphopenia, were treated with filgrastim (granulocyte colony-stimulating factor [G-CSF]).
- Both patients exhibited clinical improvement, decreased JC virus loads in cerebrospinal fluid, and enhanced magnetic resonance imaging findings post-G-CSF administration.
Findings:
- Treatment with G-CSF led to positive clinical outcomes in two patients with severe PML.
- JC virus loads decreased, and neurological improvements were observed following G-CSF therapy.
Implications:
- Granulocyte colony-stimulating factor (G-CSF) shows potential as an immune-enhancing therapy for non-HIV-associated PML.
- G-CSF may stimulate innate immunity and bolster anti-JC viral immune surveillance, offering a new therapeutic avenue.
- Further research into G-CSF for PML management in immunocompromised individuals is warranted.
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