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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...

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Autores principales: Daley, Jessica, Halligan, Katharine, Howrie, Denise, Salgado, Claudia M., Superdock, Alexandra, Friehling, Erika, Bailey, Kelly M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2023
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.
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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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