Case report: Novel treatment regimen for enterovirus encephalitis in SCID

Front Immunol. 2022 Sep 13:13:930031. doi: 10.3389/fimmu.2022.930031. eCollection 2022.

Abstract

Most non-polio enterovirus infections in immunocompetent individuals are acute and self-limiting in nature; however, infection can be severe, chronic and have devastating outcomes in immunocompromised hosts. Therapeutic strategies have predominantly involved supportive care, with the lack of approved antiviral treatments proving challenging for management. We report a case of an 8-month-old child who presented with severe enterovirus encephalitis following gene therapy for X-linked severe combined immunodeficiency (X-SCID) and who demonstrated clinical and microbiological improvement after a novel regimen of favipiravir, fluoxetine, and high-dose intravenous immunoglobulin (IVIg). The patient presented 6 weeks post-gene therapy with rapid neurological deterioration in the context of incomplete immune reconstitution, with microbiological and radiological evidence confirming enterovirus encephalitis. His neurologic examination stabilised 8 weeks after treatment, and he subsequently demonstrated excellent immune recovery. This is the first case report of combined therapy with favipiravir, fluoxetine, and high-dose IVIg in the context of severe enterovirus encephalitis in an immunocompromised host. This case highlights the importance of considering enterovirus encephalitis in immunocompromised patients presenting with both acute and chronic neurological signs, as well as developmental regression. The demonstrated treatment success and the associated low risk of toxicity warrant further investigation of this therapeutic regimen.

Keywords: antiviral agents; case report; enterovirus; gene therapy (GT); infectious encephalitis; primary immunodeficiency diseases; severe combined immunodeficiency.

Publication types

  • Case Reports

MeSH terms

  • Amides
  • Antiviral Agents / therapeutic use
  • Encephalitis, Viral* / diagnosis
  • Encephalitis, Viral* / drug therapy
  • Enterovirus Infections* / diagnosis
  • Enterovirus Infections* / drug therapy
  • Enterovirus*
  • Fluoxetine
  • Humans
  • Immunoglobulins, Intravenous / therapeutic use
  • Infant
  • Male
  • Pyrazines

Substances

  • Amides
  • Antiviral Agents
  • Immunoglobulins, Intravenous
  • Pyrazines
  • Fluoxetine
  • favipiravir