PTPMT1 Is Required for Embryonic Cardiac Cardiolipin Biosynthesis to Regulate Mitochondrial Morphogenesis and Heart Development

Chen, Zee; Zhu, Siting; Wang, Hong; Wang, Li; Zhang, Jianlin; Gu, Yusu; Tan, Changming; Dhanani, Mehul; Wever, Eric; Wang, Xinru; Xie, Boyu; Wang, Shijia; Huang, Lei; Kampen, Antoine H. C. van; Liu, Jie; Han, Zhen; Patel, Hemal H.; Vaz, Frédéric M.; Fang, Xi; Chen, Ju; Ouyang, Kunfu

doi:10.1161/circulationaha.121.054768

Cited by 20 publications

(15 citation statements)

References 5 publications

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“…Abnormal CL biosynthesis and remodeling have been associated with altered cristae morphology and disrupted mitochondrial-shaping proteins, affecting the number, length, and organizing pattern of the cristae, and ultimately the shape of mitochondria and the progress of OXPHOS in both human patients and animal models [ 13 , 14 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 , 58 ]. Mitochondrial ATP synthase is a polymerase complex consisting of two functional domains: F0, located in the IMM, and F1, located in the mitochondrial matrix.…”

Section: CL and Mitochondrial Cristae Formationmentioning

confidence: 99%

“…ATP synthase has been shown to form higher oligomeric assemblies consisting of rows of dimers that are critical for cristae formation and stabilization [ 59 ]. CL appears to be critical for oligomerization and ordering in these ATP synthase assemblies, and an abnormal CL content has been shown to be associated with reduced oligomerization and ordering of ATP synthase [ 13 , 14 , 60 , 61 , 62 ]. On the other hand, the MICOS is the other machinery that has been identified to play a crucial role in maintaining the characteristic architecture of the IMM in addition to ATP synthase [ 63 ].…”

Section: CL and Mitochondrial Cristae Formationmentioning

confidence: 99%

“…Evidently, normal CL biosynthesis and remodeling are required for normal cardiac development and physiology. Our recent study using a cardiac-specific PTPMT1 mouse model demonstrated that deficiency in CL biosynthesis induced by PTPMT1 deletion in mouse cardiomyocytes decreased embryonic cardiac cell proliferation, resulting in decreased thickening of ventricular walls, arrestment of heart development and growth, and eventually embryonic lethality [ 13 ]. Furthermore, various cardiac defects, including dilated cardiomyopathy, hypertrophic cardiomyopathy, left ventricular myocardial noncompaction, and ventricular arrhythmia, have been observed in patients with Barth syndrome, a rare X-linked genetic disorder caused by mutations in TAZ [ 17 , 96 , 133 , 134 ].…”

Section: CL and Cardiomyopathymentioning

confidence: 99%

“…The accumulation of oxidized CL in the OMM leads to the opening of the mitochondrial permeability transition pore (mPTP) and the release of cytochrome c (Cyt C) from mitochondria into the cytoplasm [ 10 ], ultimately inducing apoptosis [ 11 ]. Thus, CL can play multiple roles, including the formation and stability of the mitochondrial cristae respiration supercomplex [ 12 , 13 , 14 ], mitochondrial quality control and dynamics [ 15 ], as well as mitochondrial biogenesis and protein import [ 16 ]. Notably, the roles of CL in regulating mitochondrial structure and functioning have been discussed in many excellent reviews [ 12 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 ].…”

Section: Introductionmentioning

confidence: 99%

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Cardiolipin Regulates Mitochondrial Ultrastructure and Function in Mammalian Cells

Jiang

Shen

Huynh

et al. 2022

Genes

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Cardiolipin (CL) is a unique, tetra-acylated diphosphatidylglycerol lipid that mainly localizes in the inner mitochondria membrane (IMM) in mammalian cells and plays a central role in regulating mitochondrial architecture and functioning. A deficiency of CL biosynthesis and remodeling perturbs mitochondrial functioning and ultrastructure. Clinical and experimental studies on human patients and animal models have also provided compelling evidence that an abnormal CL content, acyl chain composition, localization, and level of oxidation may be directly linked to multiple diseases, including cardiomyopathy, neuronal dysfunction, immune cell defects, and metabolic disorders. The central role of CL in regulating the pathogenesis and progression of these diseases has attracted increasing attention in recent years. In this review, we focus on the advances in our understanding of the physiological roles of CL biosynthesis and remodeling from human patients and mouse models, and we provide an overview of the potential mechanism by which CL regulates the mitochondrial architecture and functioning.

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Section: CL and Mitochondrial Cristae Formationmentioning

confidence: 99%

Section: CL and Mitochondrial Cristae Formationmentioning

confidence: 99%

Section: CL and Cardiomyopathymentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Cardiolipin Regulates Mitochondrial Ultrastructure and Function in Mammalian Cells

Jiang

Shen

Huynh

et al. 2022

Genes

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“…Total RNA was extracted using TRIzol reagent (Invitrogen) or RNeasy Mini Kit (Qiagen), and cDNA was synthesized using TransScript One-Step gDNA Removal and cDNA Synthesis SuperMix Kit (TransGen Biotech) as previously described ( Chen et al., 2021b ). Quantitative real-time PCR (RT-PCR) was performed using TransStart Tip Green qPCR SuperMix (TransGen Biotech) according to the manufacturer’s instructions.…”

Section: Methodsmentioning

confidence: 99%

IP3R-mediated Ca2+ signaling controls B cell proliferation through metabolic reprogramming

Tang

Wang

et al. 2022

iScience

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Extrusion‐Based 3D Bioprinting of Adhesive Tissue Engineering Scaffolds Using Hybrid Functionalized Hydrogel Bioinks

et al. 2023

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Adhesive tissue engineering scaffolds (ATESs) have emerged as an innovative alternative means, replacing sutures and bioglues, to secure the implants onto target tissues. Relying on their intrinsic tissue adhesion characteristics, ATES systems enable minimally invasive delivery of various scaffolds. This study investigates development of the first class of 3D bioprinted ATES constructs using functionalized hydrogel bioinks. Two ATES delivery strategies, in situ printing onto the adherend versus printing and then transferring to the target surface, are tested using two bioprinting methods, embedded versus air printing. Dopamine-modified methacrylated hyaluronic acid (HAMA-Dopa) and gelatin methacrylate (GelMA) are used as the main bioink components, enabling fabrication of scaffolds with enhanced adhesion and crosslinking properties. Results demonstrate that dopamine modification improved adhesive properties of the HAMA-Dopa/GelMA constructs under various loading conditions, while maintaining their structural fidelity, stability, mechanical properties, and biocompatibility. While directly printing onto the adherend yields superior adhesive strength, embedded printing followed by transfer to the target tissue demonstrates greater potential for translational applications. Together, these results demonstrate the potential of bioprinted ATESs as off-the-shelf medical devices for diverse biomedical applications.

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PTPMT1 Is Required for Embryonic Cardiac Cardiolipin Biosynthesis to Regulate Mitochondrial Morphogenesis and Heart Development

Cited by 20 publications

References 5 publications

Cardiolipin Regulates Mitochondrial Ultrastructure and Function in Mammalian Cells

Cardiolipin Regulates Mitochondrial Ultrastructure and Function in Mammalian Cells

IP3R-mediated Ca2+ signaling controls B cell proliferation through metabolic reprogramming

Extrusion‐Based 3D Bioprinting of Adhesive Tissue Engineering Scaffolds Using Hybrid Functionalized Hydrogel Bioinks

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