Source abstract
Abstract
Fabry disease (FD) is an X-linked hereditary disorder that results in the malfunction of α-galactosidase A (αGLA), leading to the accumulation of globotriaosylceramide (GB3) in cells and causing organ damage. This condition induces several pathological intracellular signaling pathways, with the dysfunction in autophagy being a crucial component. Phospholipid–polyethylene glycol-capped Ceria–Zirconia antioxidant nanoparticles (PEG–CZNPs) have been reported to enhance autophagy flux. This study aims to assess the mechanisms of action of PEG–CZNPs in autophagy regulation and examine their effects on chronic kidney injury in cellular and animal models of FD. A stable cellular model of FD was successfully created through the shRNA transfection of αGLA. PEG–CZNPs were found to enhance autophagy flux by translocating Transcription factor EB (TFEB) to the nucleus. To demonstrate TFEB’s importance in autophagy flux by PEG–CZNPs, HK-2 cells were transfected with siTFEB. Autophagy flux significantly decreased after the knockdown of TFEB, despite PEG–CZNPs treatment. We next assessed the upper signaling pathway of TFEB by PEG–CZNPs. TFEB dephosphorylation was significantly influenced by the Akt/GSK3ß signaling pathway in response to PEG–CZNPs. PEG–CZNPs successfully reduced intracellular GB3 accumulation and decreased fibrous markers such as α-smooth muscle actin (αSMA), collagen type IV (ColIV), and matrix metallopeptidase 9 (MMP9) expression in the cellular model of FD. To evaluate the impact of PEG–CZNPs on kidney injury in a mouse model of FD, saline or PEG–CZNPs (10 mg/kg/day) were administered intraperitoneally twice per week for 24 or 48 weeks, starting at the age of 4 weeks. PEG–CZNPs significantly reduced both GB3 accumulation and αSMA expression in the kidneys. In conclusion, these results suggest that PEG–CZNPs promote autophagy flux through the Akt/GSK3β-TFEB signaling pathway and demonstrate a beneficial effect on kidney fibrosis and intracellular GB3 reduction in cellular and animal models of FD. These results provide valuable insights into potential therapeutics for FD.
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Affiliations
Division of Nephrology and Department of Internal Medicine, Myunggok Medical Research Institute, College of Medicine, Konyang University, Daejeon 35365, Republic of Korea
Department of Chemistry, Hannam University, Daejeon 34054, Republic of Korea
ERSTEQ Inc, Daejeon 34013, Republic of Korea
Myunggok Medical Research Institute, College of Medicine, Konyang University, Daejeon 35365, Republic of Korea
Department of Pharmacology, College of Medicine, Konyang University, Daejeon 35365, Republic of Korea
Department of Obstetrics and Gynecology, College of Medicine, Konyang University, Daejeon 35365, Republic of Korea
Personalized Genomic Medicine Research Center, Korea Research Institute of Bioscience and Biotechnology (KRIBB), Daejeon 34101, Republic of Korea
Department of Pathology, College of Medicine, Konyang University, Daejeon 35365, Republic of Korea
Funding
National Research Foundation of Korea