ArticleNeurology2014
JCV GCN in a natalizumab-treated MS patient is associated with mutations of the VP1 capsid gene.
Article in Neurology, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.
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Who cites it
22 citing papers in PubMed, 37 citations in OpenAlex.
- Hot cross bun sign in JC-Virus Granule cell neuronopathy in HIV infected patient - a case report.BMC neurology · 2024Article
- Progressive Multifocal Leukoencephalopathy: Pathogenesis, Diagnostic Tools, and Potential Biomarkers of Response to Therapy.Neurology · 2023Review
- Progressive multifocal leukoencephalopathy and granule cell neuronopathy with novel mutation flanking VP1 C-terminus in natalizumab-extended interval dosing.Neurology(R) neuroimmunology & neuroinflammation · 2020Article
- Treatment of natalizumab-associated PML with filgrastim.Annals of clinical and translational neurology · 2019Article
- Cerebellar syndrome in a man treated with natalizumab: From the National Multiple Sclerosis Society Case Conference Proceedings.Neurology(R) neuroimmunology & neuroinflammation · 2019Article
- JC Virus Granule Cell Neuronopathy as AIDS-Presenting Illness.The Canadian journal of neurological sciences. Le journal canadien des sciences neurologiques · 2018Article
- JC Virus Granule Cell Neuronopathy and Lymphoma.Open forum infectious diseases · 2018Article
- Microorganisms' Footprint in Neurodegenerative Diseases.Frontiers in cellular neuroscience · 2018Review
- Progressive multifocal leukoencephalopathy and hematologic malignancies: a single cancer center retrospective review.Blood advances · 2017Article
- Natalizumab granule cell neuronopathy: FDG-PET in diagnosis and immune reconstitution with G-CSF.Neurology(R) neuroimmunology & neuroinflammation · 2017Article
- JC virus granule cell neuronopathy in the setting of chronic lymphopenia treated with recombinant interleukin-7.Journal of neurovirology · 2017Article
- Progressive Multifocal Leukoencephalopathy in HIV-Uninfected Individuals.Current infectious disease reports · 2016Review
- JC Virus Infects Neurons and Glial Cells in the Hippocampus.Journal of neuropathology and experimental neurology · 2016Article
- Opportunistic DNA Recombination With Epstein-Barr Virus at Sites of Control Region Rearrangements Mediating JC Virus Neurovirulence.The Journal of infectious diseases · 2016Article
- Concomitant granule cell neuronopathy in patients with natalizumab-associated PML.Journal of neurology · 2016Article
- Stratification and monitoring of natalizumab-associated progressive multifocal leukoencephalopathy risk: recommendations from an expert group.Journal of neurology, neurosurgery, and psychiatry · 2016Review
- Low frequencies of central memory CD4 T cells in progressive multifocal leukoencephalopathy.Neurology(R) neuroimmunology & neuroinflammation · 2015Article
- Risk Mitigation Strategies for Adverse Reactions Associated with the Disease-Modifying Drugs in Multiple Sclerosis.CNS drugs · 2015Review
- Lack of Major Histocompatibility Complex Class I Upregulation and Restrictive Infection by JC Virus Hamper Detection of Neurons by T Lymphocytes in the Central Nervous System.Journal of neuropathology and experimental neurology · 2015Article
- Novel syndromes associated with JC virus infection of neurons and meningeal cells: no longer a gray area.Current opinion in neurology · 2015Review
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Authors and funding
7 authors at 1 institution in 1 country.
Funding
Abstract
objectiveTo describe the clinical, neuroimaging, immunologic, and virologic characteristics of JC virus-associated granule cell neuronopathy (JCV GCN) in a natalizumab-treated patient with multiple sclerosis (MS) who developed immune reconstitution inflammatory syndrome (IRIS) after natalizumab withdrawal.
methodsWe obtained longitudinal clinical data as well as MRI and proton magnetic resonance spectroscopy from this patient with MS. We measured JCV-specific cellular immune response in his peripheral blood by intracellular cytokine staining and sequenced a fragment of JCV VP1 capsid gene detected in his CSF. We contrast our findings with the first recently reported case.
resultsThis patient presented with worsening cerebellar symptoms and progressive cerebellar atrophy without new MS lesions on MRI after 63 months of natalizumab monotherapy. JCV DNA was detected in his CSF by PCR and harbored novel GCN-type mutations in the VP1 gene. He developed IRIS upon discontinuation of natalizumab and plasma exchange, which manifested itself by a worsening of clinical symptoms and contrast enhancement in the cerebellum on MRI. Treatment with corticosteroids resulted in resolution of IRIS, as demonstrated by proton magnetic resonance spectroscopy. The patient had a strong JCV-specific T-cell response in his peripheral blood and remains alive after 15 months from onset of symptoms, although with significant disability. He did not have MS relapse on glatiramer acetate.
conclusionsJCV GCN should be considered in patients on natalizumab presenting with progressive cerebellar symptoms and cerebellar atrophy, and is associated with mutations in the JCV VP1 gene. Natalizumab withdrawal may be complicated by JCV GCN IRIS, and require treatment with corticosteroids.
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