Key result
CRISPRa targeting of PGC-1α and NRF1 yields ~2-fold gains in iPSC-CM mitochondrial maturation.
Systematic Review
Review highlights interventions boosting mitochondrial development in iPSC-CMs, suggesting pathways for improved functionality.
Human iPSC-derived cardiomyocytes (iPSC-CMs) exhibit fetal-like mitochondrial networks and limited oxidative metabolism, constraining their translational utility. The key bottleneck is mitochondrial immaturity, resulting from blunted PGC-1α–NRF1/2–TFAM axis activation and insufficient nuclear–mitochondrial coordination, rather than sarcomeric or electrophysiological immaturity alone. This review synthesizes genome-guided interventions (CRISPRa and mtDNA editing) and complementary environmental strategies—including metabolic substrate switching, electromechanical stimulation, and extracellular vesicle (EV)-mediated mitochondrial transfer—to drive mitochondrial biogenesis and maturation in iPSC-CMs. We systematically reviewed studies (2005–2025) targeting (1) key regulators of mitochondrial biogenesis (PGC-1α, NRF1/2, TFAM), (2) CRISPR-based transcriptional activators/repressors and mtDNA editors (DdCBE, mitoTALENs), and (3) maturation approaches such as metabolic conditioning, electromechanical stimulation, 3D tissue culture, and EV-mediated mitochondrial transfer. CRISPRa-mediated activation of PGC-1α, NRF1, and GATA4, combined with mtDNA base editors, enhances mitochondrial mass and OXPHOS function, while integration with environmental maturation strategies further promotes adult-like phenotypes. Integrative approaches that combine genome-guided interventions (CRISPRa, mtDNA editing) with environmental maturation cues yield the most adult-like iPSC-CM phenotypes reported to date. CRISPR-guided mitochondrial biogenesis thus represents a frontier for producing metabolically competent, structurally mature iPSC-CMs for disease modeling and therapy. Remaining translational challenges include efficient mitochondrial delivery, metabolic homeostasis, and multi-omics validation. We propose standardized workflows to couple nuclear and mitochondrial editing with maturation strategies.
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Shahannaz et al. (2026) conducted a systematic review in Mitochondrial immaturity in human iPSC-derived cardiomyocytes (iPSC-CMs). CRISPR-guided nuclear activation (CRISPRa of PGC-1α, NRF1/2, TFAM) and mtDNA editing (DdCBE, mitoTALENs) integrated with environmental maturation cues vs. Isolated metabolic conditioning, electromechanical stimulation, or unedited control iPSC-CMs was evaluated on Mitochondrial biogenesis and functional maturation (mtDNA copy number, cristae density, maximal oxygen consumption rate [OCR], ATP production, and calcium handling). CRISPRa activation of PGC-1α and NRF1 yields ~2-fold increases in mtDNA content and maximal OCR in iPSC-CMs, and combining genome editing with environmental cues produces the most adult-like phenotypes.
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