ArticleHuman molecular genetics2018
Conditional ablation and conditional rescue models for Casq2 elucidate the role of development and of cell-type specific expression of Casq2 in the CPVT2 phenotype.
Article in Human molecular genetics, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.
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Who cites it
8 citing papers in PubMed, 10 citations in OpenAlex.
- Electrophysiological Characteristics and Ablation Outcomes in Patients With Catecholaminergic Polymorphic Ventricular Tachycardia.Journal of the American Heart Association · 2023Article
- Animal Models to Study Cardiac Arrhythmias.Circulation research · 2022Review
- The Purkinje-myocardial junction is the anatomic origin of ventricular arrhythmia in CPVT.JCI insight · 2022Article
- Calsequestrin, a key protein in striated muscle health and disease.Journal of muscle research and cell motility · 2021Review
- Association Study of Genetic Variants in Calcium Signaling-Related Genes With Cardiovascular Diseases.Frontiers in cell and developmental biology · 2021Article
- Bi-FoRe: an efficient bidirectional knockin strategy to generate pairwise conditional alleles with fluorescent indicators.Protein & cell · 2021Article
- Molecular and tissue mechanisms of catecholaminergic polymorphic ventricular tachycardia.The Journal of physiology · 2020Review
- Insights Into the Pathogenesis of Catecholaminergic Polymorphic Ventricular Tachycardia From Engineered Human Heart Tissue.Circulation · 2019Article
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Authors and funding
14 authors at 6 institutions in 2 countries.
Funding
Abstract
Cardiac calsequestrin (Casq2) associates with the ryanodine receptor 2 channel in the junctional sarcoplasmic reticulum to regulate Ca2+ release into the cytoplasm. Patients carrying mutations in CASQ2 display low resting heart rates under basal conditions and stress-induced polymorphic ventricular tachycardia (CPVT). In this study, we generate and characterize novel conditional deletion and conditional rescue mouse models to test the influence of developmental programs on the heart rate and CPVT phenotypes. We also compare the requirements for Casq2 function in the cardiac conduction system (CCS) and in working cardiomyocytes. Our study shows that the CPVT phenotype is dependent upon concurrent loss of Casq2 function in both the CCS and in working cardiomyocytes. Accordingly, restoration of Casq2 in only the CCS prevents CPVT. In addition, occurrence of CPVT is independent of the developmental history of Casq2-deficiency. In contrast, resting heart rate depends upon Casq2 gene activity only in the CCS and upon developmental history. Finally, our data support a model where low basal heart rate is a significant risk factor for CPVT.
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