Case Report: Evolution of a Severe Vascular Refractory Form of ECD Requiring Liver Transplantation Correlated With the Change in the Monocyte Subset Analysis
Erdheim–Chester disease is a rare histiocytosis characterized by iconic features associated with compatible histology. Most patients have somatic mutations in the MAP-kinase pathway gene, and the mutations occur in CD14+ monocytes. Differentiation of the myeloid lineage plays a central role in the p...
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Frontiers Media S.A.
2021
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oai:doaj.org-article:4ee5df62bbbe43378a91e086829755362021-11-12T06:56:28ZCase Report: Evolution of a Severe Vascular Refractory Form of ECD Requiring Liver Transplantation Correlated With the Change in the Monocyte Subset Analysis1664-322410.3389/fimmu.2021.755846https://doaj.org/article/4ee5df62bbbe43378a91e086829755362021-11-01T00:00:00Zhttps://www.frontiersin.org/articles/10.3389/fimmu.2021.755846/fullhttps://doaj.org/toc/1664-3224Erdheim–Chester disease is a rare histiocytosis characterized by iconic features associated with compatible histology. Most patients have somatic mutations in the MAP-kinase pathway gene, and the mutations occur in CD14+ monocytes. Differentiation of the myeloid lineage plays a central role in the pathogenesis of histiocytosis. Monocytes are myeloid-derived white blood cells, divided into three subsets, but only the CD14++CD16− “classical monocyte” can differentiate into dendritic cells and tissue macrophages. Since most mutations occur in CD14+ cells and since ECD patients have a particular monocytic phenotype resembling CMML, we studied the correlation between disease activity and monocytic subset distribution during the course of a severe vascular form of ECD requiring liver transplantation. During early follow-up, increased CD14++CD16− “classical monocyte” associated with decreased CD14lowCD16++ “non-classical monocyte” correlated with disease activity. Further studies are needed to confirm the use of monocyte as a marker of disease activity in patients with ECD.Jérôme RazanamaheryAnne RoggyJean-François EmileAlexandre MalakhiaZaher LakkisFrancine Garnache-OttouThibaud SoumagneFleur Cohen-AubartJulien HarocheBernard BonnotteFrontiers Media S.A.articlemonocytehistiocytosisErdheim–Chester diseasetransplantationvascular diagnosisImmunologic diseases. AllergyRC581-607ENFrontiers in Immunology, Vol 12 (2021) |
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DOAJ |
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EN |
| topic |
monocyte histiocytosis Erdheim–Chester disease transplantation vascular diagnosis Immunologic diseases. Allergy RC581-607 |
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monocyte histiocytosis Erdheim–Chester disease transplantation vascular diagnosis Immunologic diseases. Allergy RC581-607 Jérôme Razanamahery Anne Roggy Jean-François Emile Alexandre Malakhia Zaher Lakkis Francine Garnache-Ottou Thibaud Soumagne Fleur Cohen-Aubart Julien Haroche Bernard Bonnotte Case Report: Evolution of a Severe Vascular Refractory Form of ECD Requiring Liver Transplantation Correlated With the Change in the Monocyte Subset Analysis |
| description |
Erdheim–Chester disease is a rare histiocytosis characterized by iconic features associated with compatible histology. Most patients have somatic mutations in the MAP-kinase pathway gene, and the mutations occur in CD14+ monocytes. Differentiation of the myeloid lineage plays a central role in the pathogenesis of histiocytosis. Monocytes are myeloid-derived white blood cells, divided into three subsets, but only the CD14++CD16− “classical monocyte” can differentiate into dendritic cells and tissue macrophages. Since most mutations occur in CD14+ cells and since ECD patients have a particular monocytic phenotype resembling CMML, we studied the correlation between disease activity and monocytic subset distribution during the course of a severe vascular form of ECD requiring liver transplantation. During early follow-up, increased CD14++CD16− “classical monocyte” associated with decreased CD14lowCD16++ “non-classical monocyte” correlated with disease activity. Further studies are needed to confirm the use of monocyte as a marker of disease activity in patients with ECD. |
| format |
article |
| author |
Jérôme Razanamahery Anne Roggy Jean-François Emile Alexandre Malakhia Zaher Lakkis Francine Garnache-Ottou Thibaud Soumagne Fleur Cohen-Aubart Julien Haroche Bernard Bonnotte |
| author_facet |
Jérôme Razanamahery Anne Roggy Jean-François Emile Alexandre Malakhia Zaher Lakkis Francine Garnache-Ottou Thibaud Soumagne Fleur Cohen-Aubart Julien Haroche Bernard Bonnotte |
| author_sort |
Jérôme Razanamahery |
| title |
Case Report: Evolution of a Severe Vascular Refractory Form of ECD Requiring Liver Transplantation Correlated With the Change in the Monocyte Subset Analysis |
| title_short |
Case Report: Evolution of a Severe Vascular Refractory Form of ECD Requiring Liver Transplantation Correlated With the Change in the Monocyte Subset Analysis |
| title_full |
Case Report: Evolution of a Severe Vascular Refractory Form of ECD Requiring Liver Transplantation Correlated With the Change in the Monocyte Subset Analysis |
| title_fullStr |
Case Report: Evolution of a Severe Vascular Refractory Form of ECD Requiring Liver Transplantation Correlated With the Change in the Monocyte Subset Analysis |
| title_full_unstemmed |
Case Report: Evolution of a Severe Vascular Refractory Form of ECD Requiring Liver Transplantation Correlated With the Change in the Monocyte Subset Analysis |
| title_sort |
case report: evolution of a severe vascular refractory form of ecd requiring liver transplantation correlated with the change in the monocyte subset analysis |
| publisher |
Frontiers Media S.A. |
| publishDate |
2021 |
| url |
https://doaj.org/article/4ee5df62bbbe43378a91e08682975536 |
| work_keys_str_mv |
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