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- W2510192800 abstract "In 1974 Shulman described a disease characterized by fibrosis and inflammation of the muscle fascia and scleroderma-like skin changes, associated with hypergammaglobulinemia and blood eosinophilia. Eosinophilic fasciitis (EF) or Shulman syndrome is a rare disease of so far largely unknown etiology and pathophysiology. We could show a strong immunoreactivity for the mannose receptor CD206 expressed by macrophages dominating the inflammatory infiltrate in the peri- and epimysium of EF patients. Conversely, a predominant Th1 phenotype of the immune response with strong IFN-γ expression in the muscle and fascia tissue was observed at molecular level. Furthermore, the majority of EF patients exhibited a perifascicular pathology with upregulation of MHC class II in addition to upregulation of MHC class I, accompanied by a moderate perifascicular atrophy in a number of cases. Matching the rather low numbers of eosinophils seen in the tissue, there was no upregulation of IL-3, IL-5, IL-8, CCL5, CCL11, CCL24, and CCL26, cytokines and chemokines involved in activation and chemoattraction of eosinophils. Capillary loss and hypoxia are assumed to be essential pathomechanisms leading to perifascicular pathology in dermatomyositis (DM), but we could not demonstrate signs of hypoxia in EF by immunohistochemical staining for HIF1α, or show a reduction in capillary density. There was also no upregulation of type I interferon-associated genes (such as ISG15) in EF patients, which are associated with perifascicular atrophy in adult DM. Therefore, we suggest a specific IFN-γ-driven mechanism of perifascicular pathology in Shulman syndrome, associated with MHC class II expression, distinct from the mechanisms involved in other forms of myositis with perifascicular pathology." @default.
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- W2510192800 date "2015-10-01" @default.
- W2510192800 modified "2023-10-16" @default.
- W2510192800 title "Perifascicular pathology in eosinophilic fasciitis with muscle involvement" @default.
- W2510192800 doi "https://doi.org/10.1016/j.nmd.2015.06.440" @default.
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