Fabry disease (FD) is a lysosomal storage disorder caused by mutations in the gene for α-galactosidase A, inducing a progressive accumulation of globotriaosylceramide (GB3) and its metabolites in different organs and tissues. GB3 deposition does not fully explain the clinical manifestations of FD, and other pathogenetic mechanisms have been proposed, requiring the identification of new biomarkers for monitoring FD patients. Emerging evidence suggests the involvement of mitochondrial alterations in FD. Here, we propose mitochondrial-related microRNAs (miRs) as potential biomarkers of mitochondrial involvement in FD. Indeed, we demonstate that miRs regulating different aspects of mitochondrial homeostasis including expression and assembly of respiratory chain, mitogenesis, antioxidant capacity, and apoptosis are consistently dysregulated in FD patients. Our data unveil a novel noncoding RNA signature of FD patients, indicating mitochondrial-related miRs as new potential pathogenic players and biomarkers in FD. SIGNIFICANCE STATEMENT: This study demonstrates for the first time that a specific signature of circulating mitochondrial miRs (mitomiRs) is dysregulated in FD patients. MitomiRs regulating fundamental aspects of mitochondrial homeostasis and fitness, including expression and assembly of the respiratory chain, mitogenesis, antioxidant capacity, and apoptosis are significantly dysregulated in FD patients. Taken together, these new findings introduce mitomiRs as unprecedented biomarkers of FD and point at mitochondrial dysfunction as a novel potential mechanistic target for therapeutic approaches.
Mitochondrial microRNAs Are Dysregulated in Patients with Fabry Disease.
法布里病患者线粒体microRNA表达失调
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作者:Gambardella Jessica, Fiordelisi Antonella, Sorriento Daniela, Cerasuolo Federica, Buonaiuto Antonietta, Avvisato Roberta, Pisani Antonio, Varzideh Fahimeh, Riccio Eleonora, Santulli Gaetano, Iaccarino Guido
| 期刊: | Journal of Pharmacology and Experimental Therapeutics | 影响因子: | 3.800 |
| 时间: | 2023 | 起止号: | 2023 Jan;384(1):72-78 |
| doi: | 10.1124/jpet.122.001250 | 研究方向: | 其它 |
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