Aug 2026· Journal of Molecular and Cellular Cardiology· 0 citations· 47 references
Medicine
TL;DR
A cardiac-specific Miro1 knockout mouse model is generated to investigate how cardiomyocyte-Miro1 deficiency affects cardiac and mitochondrial structure-function and identifies Miro1 as a regulator of mitochondrial morphology.
Abstract
Cardiac mitochondrial remodelling is a hallmark of type 2 diabetes-linked heart failure (T2DM-HF). We previously reported that mitochondrial morphological changes occur in early-stage disease and identified down-regulation of the mitochondrial protein Miro1 (Rhot1). Neuronal Miro1 regulates mitochondrial movement but the role of cardiac Miro1 remains poorly understood. Therefore, we generated a cardiac-specific Miro1 knockout (Miro1cko) mouse model to investigate how cardiomyocyte-Miro1 deficiency affects cardiac and mitochondrial structure-function. Miro1cko mice compared to controls develop mild diastolic and systolic dysfunction and electrical abnormalities, cellular hypertrophy and fibrosis. Miro1cko leads to aberrant mitochondrial respiration and elevated H₂O₂ production, consistent with electron microscopy showing disrupted cristae morphology, with putative links to Myosin19 down-regulation. Three-dimensional electron microscopy identified mitochondrial remodelling with interfibrillar mitochondria (IFM) ~50% smaller with an increased surface complexity. Since fusion-fission protein expression was unchanged these data identify Miro1 as a regulator of mitochondrial morphology. Mitochondrial density increases (34% Miro1cko; 30% control), with abnormal IFM clustering, which we suggest is associated with impaired mitophagy since PINK1 and Parkin are down-regulated (~80% and ~ 60% respectively) and imaging flow cytometry of isolated primary cardiomyocytes identified an ~2-fold reduction to mitochondrial clearance indicative of blunted mitophagy. Heterozygous knockout mice, which display a milder cardiac phenotype, rapidly developed HF symptoms when given a high fat diet with L-NAME. In conclusion, loss of Miro1 drives multiple aberrant mitochondrial remodelling events culminating in cardiac dysfunction and predisposes towards accelerated metabolic-HF development. Loss of Miro1 may represent a critical mechanistic link in T2DM-HF pathogenesis and therefore a potential therapeutic target.
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BACKGROUND
Pathogenic variants in RBM20 cause severe dilated cardiomyopathy. Loss-of-function variants disrupt splicing; neomorphic gain-of-function (GoF) variants also mislocalize RBM20 to cytoplasmic ribonucleoprotein granules and are associated with more aggressive disease. The mechanism by which RBM20 mislocalizati...
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Abstract Metabolic syndrome (MetS) is a major contributor to cardiovascular disease and is characterized by impaired Ca2+ handling and mitochondrial dysfunction in cardiomyocytes. However, the upstream mechanisms linking metabolic stress to these alterations remain incompletely defined. Here, we investigated whether si...
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