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

被引:4
|
作者
Poli, Antoine [1 ,2 ,3 ,4 ,10 ]
Frieri, Camilla [5 ,6 ]
Lefebvre, Thibaud [3 ]
Delforge, Juliette [7 ]
Mirmiran, Arienne [3 ]
Talbi, Neila [3 ]
Moulouel, Boualem [3 ]
Six, Marion
Paradis, Valerie [8 ]
Parquet, Nathalie [9 ]
Puy, Herve [1 ,2 ,3 ,4 ]
Schmitt, Caroline [1 ,2 ,3 ,4 ]
Aslangul, Elisabeth [7 ]
de Fontbrune, Flore Sicre [5 ]
Gouya, Laurent [1 ,2 ,3 ,4 ]
机构
[1] INSERM, Ctr Rech Inflammat, U1149, Paris, France
[2] Univ Paris Cite, Paris, France
[3] Hop Louis Mourier, AP HP, Ctr Francais Porphyries, Colombes, France
[4] Lab Excellence Gr Ex, Paris, France
[5] Hop St Louis, AP HP, Serv Hematol Greffe, Paris, France
[6] AORN San Giuseppe Moscati Hosp, Avellino, Italy
[7] Hop Louis Mourier, AP HP, Serv Radiol, F-92700 Colombes, France
[8] Hop Beaujon, AP HP, Dept Pathol, Clichy, France
[9] Hop St Louis, AP HP, Unite Apherese Therapeut, Paris, France
[10] Louis Mourier Hosp, Ctr Francais Porphyries, 178 Rue Renouillers, F-92700 Colombes, France
关键词
Protoporphyria; Erythropoietic; Protoporphyrin IX; Cholestasis; Intrahepatic; Hematopoietic stem cell transplantation; Iron;
D O I
10.1016/j.ymgmr.2023.101018
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
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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页数:4
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