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Case report: Novel treatment regimen for enterovirus encephalitis in SCID

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 approve...

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Autores principales: Chetty, Kritika, Cheng, Iek, Kaliakatsos, Marios, Gonzalez-Granado, Luis Ignacio, Klapsa, Dimitra, Martin, Javier, Bamford, Alasdair, Breuer, Judith, Booth, Claire
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9513597/
https://www.ncbi.nlm.nih.gov/pubmed/36177038
http://dx.doi.org/10.3389/fimmu.2022.930031
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author Chetty, Kritika
Cheng, Iek
Kaliakatsos, Marios
Gonzalez-Granado, Luis Ignacio
Klapsa, Dimitra
Martin, Javier
Bamford, Alasdair
Breuer, Judith
Booth, Claire
author_facet Chetty, Kritika
Cheng, Iek
Kaliakatsos, Marios
Gonzalez-Granado, Luis Ignacio
Klapsa, Dimitra
Martin, Javier
Bamford, Alasdair
Breuer, Judith
Booth, Claire
author_sort Chetty, Kritika
collection PubMed
description 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.
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spelling pubmed-95135972022-09-28 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 Breuer, Judith Booth, Claire Front Immunol Immunology 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. Frontiers Media S.A. 2022-09-13 /pmc/articles/PMC9513597/ /pubmed/36177038 http://dx.doi.org/10.3389/fimmu.2022.930031 Text en Copyright © 2022 Chetty, Cheng, Kaliakatsos, Gonzalez-Granado, Klapsa, Martin, Bamford, Breuer and Booth https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Immunology
Chetty, Kritika
Cheng, Iek
Kaliakatsos, Marios
Gonzalez-Granado, Luis Ignacio
Klapsa, Dimitra
Martin, Javier
Bamford, Alasdair
Breuer, Judith
Booth, Claire
Case report: Novel treatment regimen for enterovirus encephalitis in SCID
title Case report: Novel treatment regimen for enterovirus encephalitis in SCID
title_full Case report: Novel treatment regimen for enterovirus encephalitis in SCID
title_fullStr Case report: Novel treatment regimen for enterovirus encephalitis in SCID
title_full_unstemmed Case report: Novel treatment regimen for enterovirus encephalitis in SCID
title_short Case report: Novel treatment regimen for enterovirus encephalitis in SCID
title_sort case report: novel treatment regimen for enterovirus encephalitis in scid
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9513597/
https://www.ncbi.nlm.nih.gov/pubmed/36177038
http://dx.doi.org/10.3389/fimmu.2022.930031
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