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Successful Treatment of Patient With Ewing Sarcoma in the Setting of Inherited Cholestatic Liver Disease
Progressive familial intrahepatic cholestasis type 1 (PFIC1) is an inherited, progressive cholestatic liver disease. Here, we present an approach to the treatment of Ewing sarcoma in a patient with PFIC1. The diagnosis of PFIC1 presents a unique challenge in the treatment of Ewing sarcoma, as the st...
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Lippincott Williams & Wilkins
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10284126/ https://www.ncbi.nlm.nih.gov/pubmed/36728269 http://dx.doi.org/10.1097/MPH.0000000000002623 |
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author | Daley, Jessica Halligan, Katharine Howrie, Denise Salgado, Claudia M. Superdock, Alexandra Friehling, Erika Bailey, Kelly M. |
author_facet | Daley, Jessica Halligan, Katharine Howrie, Denise Salgado, Claudia M. Superdock, Alexandra Friehling, Erika Bailey, Kelly M. |
author_sort | Daley, Jessica |
collection | PubMed |
description | Progressive familial intrahepatic cholestasis type 1 (PFIC1) is an inherited, progressive cholestatic liver disease. Here, we present an approach to the treatment of Ewing sarcoma in a patient with PFIC1. The diagnosis of PFIC1 presents a unique challenge in the treatment of Ewing sarcoma, as the standard-of-care vincristine, doxorubicin, cyclophosphamide/ifosfamide and etoposide chemotherapy backbone for Ewing sarcoma therapy treatment relies heavily on intact hepatic metabolism. In addition, we report prolonged lymphopenia and severe infectious complications in this patient, both of which may be attributed to more severe immunosuppression in setting of poor hepatic metabolism of chemotherapeutic agents. |
format | Online Article Text |
id | pubmed-10284126 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Lippincott Williams & Wilkins |
record_format | MEDLINE/PubMed |
spelling | pubmed-102841262023-06-22 Successful Treatment of Patient With Ewing Sarcoma in the Setting of Inherited Cholestatic Liver Disease Daley, Jessica Halligan, Katharine Howrie, Denise Salgado, Claudia M. Superdock, Alexandra Friehling, Erika Bailey, Kelly M. J Pediatr Hematol Oncol Online Articles: Clinical and Laboratory Observations Progressive familial intrahepatic cholestasis type 1 (PFIC1) is an inherited, progressive cholestatic liver disease. Here, we present an approach to the treatment of Ewing sarcoma in a patient with PFIC1. The diagnosis of PFIC1 presents a unique challenge in the treatment of Ewing sarcoma, as the standard-of-care vincristine, doxorubicin, cyclophosphamide/ifosfamide and etoposide chemotherapy backbone for Ewing sarcoma therapy treatment relies heavily on intact hepatic metabolism. In addition, we report prolonged lymphopenia and severe infectious complications in this patient, both of which may be attributed to more severe immunosuppression in setting of poor hepatic metabolism of chemotherapeutic agents. Lippincott Williams & Wilkins 2023-07 2023-01-12 /pmc/articles/PMC10284126/ /pubmed/36728269 http://dx.doi.org/10.1097/MPH.0000000000002623 Text en Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (https://creativecommons.org/licenses/by-nc-nd/4.0/) (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) |
spellingShingle | Online Articles: Clinical and Laboratory Observations Daley, Jessica Halligan, Katharine Howrie, Denise Salgado, Claudia M. Superdock, Alexandra Friehling, Erika Bailey, Kelly M. Successful Treatment of Patient With Ewing Sarcoma in the Setting of Inherited Cholestatic Liver Disease |
title | Successful Treatment of Patient With Ewing Sarcoma in the Setting of Inherited Cholestatic Liver Disease |
title_full | Successful Treatment of Patient With Ewing Sarcoma in the Setting of Inherited Cholestatic Liver Disease |
title_fullStr | Successful Treatment of Patient With Ewing Sarcoma in the Setting of Inherited Cholestatic Liver Disease |
title_full_unstemmed | Successful Treatment of Patient With Ewing Sarcoma in the Setting of Inherited Cholestatic Liver Disease |
title_short | Successful Treatment of Patient With Ewing Sarcoma in the Setting of Inherited Cholestatic Liver Disease |
title_sort | successful treatment of patient with ewing sarcoma in the setting of inherited cholestatic liver disease |
topic | Online Articles: Clinical and Laboratory Observations |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10284126/ https://www.ncbi.nlm.nih.gov/pubmed/36728269 http://dx.doi.org/10.1097/MPH.0000000000002623 |
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