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Management of erythropoietic protoporphyria with cholestatic liver disease: A case report

Erythropoietic protoporphyria (EPP) is a rare metabolic disease of the heme biosynthetic pathway where an enzymatic dysfunction results in protoporphyrin IX (PPIX) accumulation in erythroid cells. The porphyrins are photo-reactive and are responsible for severe photosensitivity in patients, thus dra...

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Autores principales: Poli, Antoine, Frieri, Camilla, Lefebvre, Thibaud, Delforge, Juliette, Mirmiran, Arienne, Talbi, Neila, Moulouel, Boualem, Six, Marion, Paradis, Valérie, Parquet, Nathalie, Puy, Hervé, Schmitt, Caroline, Aslangul, Elisabeth, de Fontbrune, Flore Sicre, Gouya, Laurent
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10694760/
http://dx.doi.org/10.1016/j.ymgmr.2023.101018
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author Poli, Antoine
Frieri, Camilla
Lefebvre, Thibaud
Delforge, Juliette
Mirmiran, Arienne
Talbi, Neila
Moulouel, Boualem
Six, Marion
Paradis, Valérie
Parquet, Nathalie
Puy, Hervé
Schmitt, Caroline
Aslangul, Elisabeth
de Fontbrune, Flore Sicre
Gouya, Laurent
author_facet Poli, Antoine
Frieri, Camilla
Lefebvre, Thibaud
Delforge, Juliette
Mirmiran, Arienne
Talbi, Neila
Moulouel, Boualem
Six, Marion
Paradis, Valérie
Parquet, Nathalie
Puy, Hervé
Schmitt, Caroline
Aslangul, Elisabeth
de Fontbrune, Flore Sicre
Gouya, Laurent
author_sort Poli, Antoine
collection PubMed
description Erythropoietic protoporphyria (EPP) is a rare metabolic disease of the heme biosynthetic pathway where an enzymatic dysfunction results in protoporphyrin IX (PPIX) accumulation in erythroid cells. The porphyrins are photo-reactive and are responsible for severe photosensitivity in patients, thus drastically decreasing their quality of life. The liver eliminates PPIX and as such, the main and rare complication of EPP is progressive cholestatic liver disease, which can lead to liver failure. The management of this complication is challenging, as it often requires a combination of approaches to promote PPIX elimination and suppress the patient's erythropoiesis. Here we described a 3-year follow-up of an EPP patient, with three episodes of liver involvement, aggravated by the coexistence of a factor VII deficiency. It covers all the different types of intervention available for the management of liver disease, right through to successful allogeneic hematopoietic stem cell transplantation.
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spelling pubmed-106947602023-12-05 Management of erythropoietic protoporphyria with cholestatic liver disease: A case report Poli, Antoine Frieri, Camilla Lefebvre, Thibaud Delforge, Juliette Mirmiran, Arienne Talbi, Neila Moulouel, Boualem Six, Marion Paradis, Valérie Parquet, Nathalie Puy, Hervé Schmitt, Caroline Aslangul, Elisabeth de Fontbrune, Flore Sicre Gouya, Laurent Mol Genet Metab Rep Case Report Erythropoietic protoporphyria (EPP) is a rare metabolic disease of the heme biosynthetic pathway where an enzymatic dysfunction results in protoporphyrin IX (PPIX) accumulation in erythroid cells. The porphyrins are photo-reactive and are responsible for severe photosensitivity in patients, thus drastically decreasing their quality of life. The liver eliminates PPIX and as such, the main and rare complication of EPP is progressive cholestatic liver disease, which can lead to liver failure. The management of this complication is challenging, as it often requires a combination of approaches to promote PPIX elimination and suppress the patient's erythropoiesis. Here we described a 3-year follow-up of an EPP patient, with three episodes of liver involvement, aggravated by the coexistence of a factor VII deficiency. It covers all the different types of intervention available for the management of liver disease, right through to successful allogeneic hematopoietic stem cell transplantation. Elsevier 2023-10-31 /pmc/articles/PMC10694760/ http://dx.doi.org/10.1016/j.ymgmr.2023.101018 Text en © 2023 The Authors https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Poli, Antoine
Frieri, Camilla
Lefebvre, Thibaud
Delforge, Juliette
Mirmiran, Arienne
Talbi, Neila
Moulouel, Boualem
Six, Marion
Paradis, Valérie
Parquet, Nathalie
Puy, Hervé
Schmitt, Caroline
Aslangul, Elisabeth
de Fontbrune, Flore Sicre
Gouya, Laurent
Management of erythropoietic protoporphyria with cholestatic liver disease: A case report
title Management of erythropoietic protoporphyria with cholestatic liver disease: A case report
title_full Management of erythropoietic protoporphyria with cholestatic liver disease: A case report
title_fullStr Management of erythropoietic protoporphyria with cholestatic liver disease: A case report
title_full_unstemmed Management of erythropoietic protoporphyria with cholestatic liver disease: A case report
title_short Management of erythropoietic protoporphyria with cholestatic liver disease: A case report
title_sort management of erythropoietic protoporphyria with cholestatic liver disease: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10694760/
http://dx.doi.org/10.1016/j.ymgmr.2023.101018
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