Frontiers in Immunology (Sep 2022)

Case report: Novel treatment regimen for enterovirus encephalitis in SCID

  • Kritika Chetty,
  • Kritika Chetty,
  • Iek Cheng,
  • Iek Cheng,
  • Marios Kaliakatsos,
  • Luis Ignacio Gonzalez-Granado,
  • Luis Ignacio Gonzalez-Granado,
  • Dimitra Klapsa,
  • Javier Martin,
  • Alasdair Bamford,
  • Alasdair Bamford,
  • Judith Breuer,
  • Judith Breuer,
  • Claire Booth,
  • Claire Booth

DOI
https://doi.org/10.3389/fimmu.2022.930031
Journal volume & issue
Vol. 13

Abstract

Read online

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