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- W2005442011 abstract "Palmitoyl-protein thioesterase-1 (PPT1) deficiency causes infantile neuronal ceroid lipofuscinosis (INCL), a devastating childhood neurodegenerative storage disorder. We previously reported that neuronal apoptosis in INCL is mediated by endoplasmic reticulum-stress. ER-stress disrupts Ca(2+)-homeostasis and stimulates the expression of Ca(2+)-binding proteins. We report here that in the PPT1-deficient human and mouse brain the levels of S100B, a Ca(2+)-binding protein, and its receptor, RAGE (receptor for advanced glycation end-products) are elevated. We further demonstrate that activation of RAGE signaling in astroglial cells mediates pro-inflammatory cytokine production, which is inhibited by SiRNA-mediated suppression of RAGE expression. We propose that RAGE signaling contributes to neuroinflammation in INCL." @default.
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- W2005442011 date "2008-10-21" @default.
- W2005442011 modified "2023-09-26" @default.
- W2005442011 title "RAGE signaling contributes to neuroinflammation in infantile neuronal ceroid lipofuscinosis" @default.
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- W2005442011 doi "https://doi.org/10.1016/j.febslet.2008.10.015" @default.
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