Eosinophilic fasciitis with paroxysmal nocturnal hemoglobinuria
Copyright © 2012 Société française de rhumatologie. Published by Elsevier SAS. All rights reserved..
Eosinophilic fasciitis is a rare connective tissue disorder, which can be associated with hematological complications in 10% of cases, such as aplastic anemia or acquired amegakaryocytic thrombocytopenia. Paroxysmal nocturnal hemoglobinuria had never been described in a patient suffering from eosinophilic fasciitis. We report an original case of a 59-year-old patient who developed a moderate aplastic pancytopenia while he was treated for a biopsy-proven eosinophilic fasciitis. A complete set of investigations was carried out and was found to be negative, including a first research of paroxysmal nocturnal hemoglobinuria. Two years after disease onset, while pancytopenia remained stable, occurrence of morning dark urine led to found a paroxysmal nocturnal hemoglobinuria clone. We discuss a potential link between the two conditions and hypothesize that paroxysmal nocturnal hemoglobinuria blood cells may pre-exist for a long time and take a survival advantage in the setting of marrow injury, as observed in eosinophilic fasciitis with hematological complications. We finally suggest that paroxysmal nocturnal hemoglobinuria should be included as a hematological complication of eosinophilic fasciitis.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2013 |
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| Erschienen: |
2013 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:80 |
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| Enthalten in: |
Joint bone spine - 80(2013), 2 vom: 10. März, Seite 208-10 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
de Boysson, Hubert [VerfasserIn] |
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| Anmerkungen: |
Date Completed 16.09.2013 Date Revised 05.02.2020 published: Print-Electronic Citation Status MEDLINE |
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| doi: |
10.1016/j.jbspin.2012.07.008 |
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| Förderinstitution / Projekttitel: |
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| 520 | |a Copyright © 2012 Société française de rhumatologie. Published by Elsevier SAS. All rights reserved. | ||
| 520 | |a Eosinophilic fasciitis is a rare connective tissue disorder, which can be associated with hematological complications in 10% of cases, such as aplastic anemia or acquired amegakaryocytic thrombocytopenia. Paroxysmal nocturnal hemoglobinuria had never been described in a patient suffering from eosinophilic fasciitis. We report an original case of a 59-year-old patient who developed a moderate aplastic pancytopenia while he was treated for a biopsy-proven eosinophilic fasciitis. A complete set of investigations was carried out and was found to be negative, including a first research of paroxysmal nocturnal hemoglobinuria. Two years after disease onset, while pancytopenia remained stable, occurrence of morning dark urine led to found a paroxysmal nocturnal hemoglobinuria clone. We discuss a potential link between the two conditions and hypothesize that paroxysmal nocturnal hemoglobinuria blood cells may pre-exist for a long time and take a survival advantage in the setting of marrow injury, as observed in eosinophilic fasciitis with hematological complications. We finally suggest that paroxysmal nocturnal hemoglobinuria should be included as a hematological complication of eosinophilic fasciitis | ||
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| 700 | 1 | |a Chapon, Françoise |e verfasserin |4 aut | |
| 700 | 1 | |a Le Mauff, Brigitte |e verfasserin |4 aut | |
| 700 | 1 | |a Auzary, Christophe |e verfasserin |4 aut | |
| 700 | 1 | |a Geffray, Loïk |e verfasserin |4 aut | |
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