ArticleAnnals of neurology2023
Progressive Multifocal Leukoencephalopathy Treated by Immune Checkpoint Inhibitors.
Article in Annals of neurology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 34 papers.
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Who cites it
34 citing papers in PubMed, 57 citations in OpenAlex.
- Safety, Immunovirologic, and Central Nervous System Effects of PD-1 Inhibition in People With HIV Without Malignancy.The Journal of infectious diseases · 2026Article
- Bispecific antibody therapy in multiple myeloma and risk of progressive multifocal leukoencephalopathy.Blood advances · 2026Article
- Pembrolizumab in people with HIV-associated progressive multifocal leukoencephalopathy: a retrospective case series.Infection · 2026Article
- Overcoming resistance to immune checkpoint inhibitors in cancer: translational mechanisms, dynamic biomarkers, clinically actionable combination strategies, and comparative checkpoint biology.Immunologic research · 2026Review
- Characteristics of progressive multifocal leukoencephalopathy in hematological malignancies, With reference to HIV-infected patients.International journal of hematology · 2026Article
- Emerging evidence for anti-PD-1 and IFN-γ as adjunctive immunotherapy in invasive mold infections.mBio · 2026Article
- Pembrolizumab Experience in a Patient with Progressive Multifocal Leukoencephalopathy Following Multiple Myeloma Treatment: A Case Report and Literature Review.Noro psikiyatri arsivi · 2026Article
- Current infectious disease management challenges in inborn errors of immunity.Annals of clinical microbiology and antimicrobials · 2025Review
- Discovery of novel disulfide-containing PD-1/PD-L1 inhibitor with in vivo influenza therapeutic efficacy.Scientific reports · 2025Article
- JC Polyomavirus Infection: A Narrative Review.Infectious diseases and therapy · 2025Review
- Article
- What would it take to prove that a chronic infection is a causal agent in Alzheimer's disease?Trends in neurosciences · 2025Review
- Therapeutic advances in multiple sclerosis: Novel therapies (immune checkpoint inhibitors, CAR-T, Anti-CD40L).Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2025Review
- CRISPR antiviral inhibits neurotrophic JC polyomavirus in 2D and 3D culture models through dual-gRNA excision by SaCas9.Molecular therapy. Nucleic acids · 2025Article
- Review
- Bridging the gaps between JCV infection models and human disease.Brain : a journal of neurology · 2024Article
- Directly Isolated Allogeneic Virus-Specific T Cells in Progressive Multifocal Leukoencephalopathy.JAMA neurology · 2024Article
- Exploring JC Polyomavirus Sequences and Human Gene Expression in Brain Tissue of Patients With Progressive Multifocal Leukoencephalopathy.The Journal of infectious diseases · 2024Article
- Biomarkers for progressive multifocal leukoencephalopathy: emerging data for use of JC virus DNA copy number in clinical trials.The Lancet. Neurology · 2024Review
- Treatment of Progressive Multifocal Leukoencephalopathy with IL-2 and Mirtazapine.Journal of clinical immunology · 2024Article
Corrections and comments
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Authors and funding
22 authors at 17 institutions in 8 countries.
Funding
Abstract
objectiveOur aim was to assess the real-world effectiveness of immune checkpoint inhibitors for treatment of patients with progressive multifocal leukoencephalopathy (PML).
methodsWe conducted a multicenter survey compiling retrospective data from 79 PML patients, including 38 published cases and 41 unpublished cases, who received immune checkpoint inhibitors as add-on to standard of care. One-year follow-up data were analyzed to determine clinical outcomes and safety profile. Logistic regression was used to identify variables associated with 1-year survival.
resultsPredisposing conditions included hematological malignancy (n = 38, 48.1%), primary immunodeficiency (n = 14, 17.7%), human immunodeficiency virus/acquired immunodeficiency syndrome (n = 12, 15.2%), inflammatory disease (n = 8, 10.1%), neoplasm (n = 5, 6.3%), and transplantation (n = 2, 2.5%). Pembrolizumab was most commonly used (n = 53, 67.1%). One-year survival was 51.9% (41/79). PML-immune reconstitution inflammatory syndrome (IRIS) was reported in 15 of 79 patients (19%). Pretreatment expression of programmed cell death-1 on circulating T cells did not differ between survivors and nonsurvivors. Development of contrast enhancement on follow-up magnetic resonance imaging at least once during follow-up (OR = 3.16, 95% confidence interval = 1.20-8.72, p = 0.02) was associated with 1-year survival. Cerebrospinal fluid JC polyomavirus DNA load decreased significantly by 1-month follow-up in survivors compared to nonsurvivors (p < 0.0001). Thirty-two adverse events occurred among 24 of 79 patients (30.4%), and led to treatment discontinuation in 7 of 24 patients (29.1%).
interpretationIn this noncontrolled retrospective study of patients with PML who were treated with immune checkpoint inhibitors, mortality remains high. Development of inflammatory features or overt PML-IRIS was commonly observed. This study highlights that use of immune checkpoint inhibitors should be strictly personalized toward characteristics of the individual PML patient. ANN NEUROL 2023;93:257-270.
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