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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...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
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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. |
format | Online Article Text |
id | pubmed-8949663 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
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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