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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...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Frontiers Media S.A.
2022
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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. |
format | Online Article Text |
id | pubmed-9513597 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
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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