4.1 Article

Compassionate-use pocapavir and immunoglobulin therapy for treatment of rituximab-associated enterovirus meningoencephalitis

Journal

JOURNAL OF NEUROVIROLOGY
Volume 28, Issue 2, Pages 329-334

Publisher

SPRINGER
DOI: 10.1007/s13365-021-01038-z

Keywords

Enterovirus; Meningoencephalitis; Antiviral therapy; Immunotherapy

Funding

  1. National Institute of Neurological Disorders and Stroke of the National Institutes of Health [K23NS117310]
  2. National Institute of Health [K23 NS105935]

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This case report describes a patient with rheumatoid arthritis who developed progressive neurologic symptoms and persistent hypogammaglobulinemia after receiving rituximab treatment. Enterovirus infection was identified in the cerebrospinal fluid using PCR and high-throughput sequencing, leading to a diagnosis of pan-Enterovirus. Treatment with pocapavir resulted in clinical stabilization, but the patient eventually died from bacterial pneumonia.
A 71-year-old woman previously on rituximab treatment for rheumatoid arthritis presented with 2 years of progressive neurologic symptoms. She was found to have persistent hypogammaglobulinemia and B cell depletion despite rituximab discontinuation a year prior. MRI revealed diffuse meningeal enhancement along the entire neuroaxis. LP showed a CSF lymphocytic pleocytosis, elevated protein, and presence of enterovirus by PCR. The patient was hospitalized several times for progressive clinical and radiologic decline, though she had transient improvements following treatment with immunoglobulin therapy. Her CSF remained positive for enterovirus PCR for at least 12 months. Though two brain biopsies were non-diagnostic, pan-Enterovirus was ultimately identified using a high-throughput next-generation sequencing technique. She was treated with compassionate-use pocapavir with clinical stabilization at 4-month follow-up; however, she expired 8 months later from a bacterial pneumonia.

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