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Case report: Novel treatment regimen for enterovirus encephalitis in SCID

Chetty, Kritika; Cheng, Iek; Kaliakatsos, Marios; Gonzalez-Granado, Luis Ignacio; Klapsa, Dimitra; Martin, Javier; Bamford, Alasdair; ... Booth, Claire; + view all (2022) Case report: Novel treatment regimen for enterovirus encephalitis in SCID. Frontiers in Immunology , 13 , Article 930031. 10.3389/fimmu.2022.930031. Green open access

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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.

Type: Article
Title: Case report: Novel treatment regimen for enterovirus encephalitis in SCID
Location: Switzerland
Open access status: An open access version is available from UCL Discovery
DOI: 10.3389/fimmu.2022.930031
Publisher version: https://doi.org/10.3389/fimmu.2022.930031
Language: English
Additional information: © 2022 Chetty, Cheng, Kaliakatsos, Gonzalez-Granado, Klapsa, Martin, Bamford, Breuer and Booth. This is an open-access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/).
Keywords: case report, primary immunodeficiency diseases, enterovirus, infectious encephalitis, antiviral agents, severe combined immunodeficiency, gene therapy (GT)
UCL classification: UCL
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health > Infection, Immunity and Inflammation Dept
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences
URI: https://discovery.ucl.ac.uk/id/eprint/10157398
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