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Salvage Therapy for Alveolar Echinococcosis—A Case Series

Benzimidazoles are the only approved drugs for the treatment of inoperable human alveolar echinococcosis but may be limited due to intolerance or, rarely, ineffectiveness. A medical second-line or salvage therapy is not available, though it is urgently needed. We report long-term follow-up data from...

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Autores principales: Burkert, Sanne, Peters, Lynn, Bloehdorn, Johannes, Grüner, Beate
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8949663/
https://www.ncbi.nlm.nih.gov/pubmed/35335657
http://dx.doi.org/10.3390/pathogens11030333
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author Burkert, Sanne
Peters, Lynn
Bloehdorn, Johannes
Grüner, Beate
author_facet Burkert, Sanne
Peters, Lynn
Bloehdorn, Johannes
Grüner, Beate
author_sort Burkert, Sanne
collection PubMed
description Benzimidazoles are the only approved drugs for the treatment of inoperable human alveolar echinococcosis but may be limited due to intolerance or, rarely, ineffectiveness. A medical second-line or salvage therapy is not available, though it is urgently needed. We report long-term follow-up data from 14 patients who underwent salvage therapy with repurposed drugs with cumulatively 53.25 patient-years. Treatment response was evaluated by both clinical outcome and image studies, preferably PET/CT. Eleven patients received amphotericin B, and 70% of evaluable cases showed some positive treatment response, but side effects often limited therapy. Five patients received nitazoxanide, of which two showed clear progression but one achieved a lasting stable disease. One patient was treated with mefloquine combination therapy in advanced disease, and overall, a positive treatment response could not be assessed. Furthermore, we report on one patient receiving pembrolizumab for a concomitant malignancy, which did not result in a reduction of echinococcal manifestation. In summary, current options of salvage therapy can sometimes induce persistent disease control, although with potentially significant side effects and high treatment costs, and mortality remains high. No clear recommendation for a salvage therapy can be given; treatment remains highly experimental, and non-pharmaceutical interventions have to be considered.
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spelling pubmed-89496632022-03-26 Salvage Therapy for Alveolar Echinococcosis—A Case Series Burkert, Sanne Peters, Lynn Bloehdorn, Johannes Grüner, Beate Pathogens Article Benzimidazoles are the only approved drugs for the treatment of inoperable human alveolar echinococcosis but may be limited due to intolerance or, rarely, ineffectiveness. A medical second-line or salvage therapy is not available, though it is urgently needed. We report long-term follow-up data from 14 patients who underwent salvage therapy with repurposed drugs with cumulatively 53.25 patient-years. Treatment response was evaluated by both clinical outcome and image studies, preferably PET/CT. Eleven patients received amphotericin B, and 70% of evaluable cases showed some positive treatment response, but side effects often limited therapy. Five patients received nitazoxanide, of which two showed clear progression but one achieved a lasting stable disease. One patient was treated with mefloquine combination therapy in advanced disease, and overall, a positive treatment response could not be assessed. Furthermore, we report on one patient receiving pembrolizumab for a concomitant malignancy, which did not result in a reduction of echinococcal manifestation. In summary, current options of salvage therapy can sometimes induce persistent disease control, although with potentially significant side effects and high treatment costs, and mortality remains high. No clear recommendation for a salvage therapy can be given; treatment remains highly experimental, and non-pharmaceutical interventions have to be considered. MDPI 2022-03-09 /pmc/articles/PMC8949663/ /pubmed/35335657 http://dx.doi.org/10.3390/pathogens11030333 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Burkert, Sanne
Peters, Lynn
Bloehdorn, Johannes
Grüner, Beate
Salvage Therapy for Alveolar Echinococcosis—A Case Series
title Salvage Therapy for Alveolar Echinococcosis—A Case Series
title_full Salvage Therapy for Alveolar Echinococcosis—A Case Series
title_fullStr Salvage Therapy for Alveolar Echinococcosis—A Case Series
title_full_unstemmed Salvage Therapy for Alveolar Echinococcosis—A Case Series
title_short Salvage Therapy for Alveolar Echinococcosis—A Case Series
title_sort salvage therapy for alveolar echinococcosis—a case series
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8949663/
https://www.ncbi.nlm.nih.gov/pubmed/35335657
http://dx.doi.org/10.3390/pathogens11030333
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