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Chronic hypoxia leads to cognitive impairment by promoting HIF-2α-mediated ceramide catabolism and alpha-synuclein hyperphosphorylation

Gaifen Li, Jia Liu, Mengyuan Guo, Yakun Gu, Yuying Guan, Qianqian Shao, Wei Ma, Xunming Ji

2022Cell Death Discovery41 citationsDOIOpen Access PDF

Abstract

Chronic hypoxia leads to irreversible cognitive impairment, primarily due to hippocampal neurodegeneration, for which the underlying mechanism remains poorly understood. We administered hypoxia (13%) to C57BL mice for 1-14 days in this study. Chronic hypoxia for 7 or 14 d, but not 1 or 3 d, resulted in alpha-synuclein hyperphosphorylation at serine129 (α-Syn p-S129) and protein aggregation, hippocampal neurodegeneration, and cognitive deficits, whereas the latter could be prevented by alpha-synuclein knockdown or an administered short peptide competing at α-Syn S129. These results suggest that α-Syn p-S129 mediates hippocampal degeneration and cognitive impairment following chronic hypoxia. Furthermore, we found that chronic hypoxia enhanced ceramide catabolism by inducing hypoxia-inducible factor (HIF)-2α and HIF-2α-dependent transcriptional activation of alkaline ceramidase 2 (Acer2). Thus, the enzymatic activity of protein phosphatase 2A (PP2A), a specific phosphatase for α-syn, is inhibited, leading to the sustained induction of α-Syn p-S129. Finally, we found that intermittent hypoxic preconditioning protected against subsequent chronic hypoxia-induced hippocampal neurodegeneration and cognitive impairment by preventing α-Syn p-S129. These results proved the critical role of α-syn pathology in chronic hypoxia-afforded cognitive impairment and revealed a novel mechanism underlying α-syn hyperphosphorylation during chronic hypoxia. The findings bear implications in developing novel therapeutic interventions for chronic hypoxia-related brain disorders.

Topics & Concepts

HyperphosphorylationCeramideHypoxia (environmental)CatabolismAlpha-synucleinCell biologyCognitive impairmentNeuroscienceBiologyCognitionMedicinePsychologyCancer researchEndocrinologyInternal medicineChemistryPhosphorylationParkinson's diseaseBiochemistryDiseaseMetabolismApoptosisOxygenOrganic chemistryHigh Altitude and HypoxiaNeuroscience of respiration and sleepCancer, Hypoxia, and Metabolism
Chronic hypoxia leads to cognitive impairment by promoting HIF-2α-mediated ceramide catabolism and alpha-synuclein hyperphosphorylation | Litcius