ArticleJCI insight2021
Inhibition of mTOR or MAPK ameliorates vmhcl/myh7 cardiomyopathy in zebrafish.
Article in JCI insight, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 40 papers, 1 of them a synthesis that pooled it.
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The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
40 citing papers in PubMed, 1 synthesis or guideline pooled it, 37 citations in OpenAlex.
- Phenotypic Spectrum of Subclinical Sarcomere-Related Hypertrophic Cardiomyopathy and Transition to Overt Disease.Circulation. Genomic and precision medicine · 2024Pooled it
- Proteomic Analysis of Valsartan for Attenuating Disease Evolution in Early Sarcomeric Hypertrophic Cardiomyopathy (VANISH) Clinical Trial.Circulation. Heart failure · 2025Trial
- CD47-mediated efferocytosis in diseases: A comprehensive review.Molecular biology reports · 2026Review
- The role of miR-3917/HLA-DRB1 in Th17 cell differentiation affects chronic periodontitis.Odontology · 2026Article
- A comprehensive review of thermal ablation technologies for transdermal drug delivery: mechanisms, commercial products, and future smart systems.Drug delivery and translational research · 2026Review
- AMPKγ2 Regulates Cardiac Hypertrophy and Arrhythmias via Interacting With Myosin.Circulation research · 2026Article
- Molecular Regulation of Pyroptosis in Alzheimer's Disease: Linking Neuroinflammation, Cell Death, and Therapeutic Targeting.Molecular neurobiology · 2026Review
- Identification of potential key efferocytosis-related genes in rheumatoid arthritis using machine learning.Clinical rheumatology · 2026Article
- Targeting NLRP3 inflammasome to mitigate chemotherapy-induced cardiotoxicity: advances in selective inhibitors and natural modulators.Molecular biology reports · 2026Review
- SGLT2 Inhibitors in Hypertrophic Cardiomyopathy: Emerging Evidence and Putative Mechanisms.Biomolecules · 2026Review
- The Role of MYH6 Variants in atrial cardiomyopathy: insights from clinical observations and zebrafish models.NPJ genomic medicine · 2026Article
- Kaempferol inhibited pyroptosis in preeclampsia by modulating the NLRP3 inflammasome through the STAT3-associated mitophagy pathway.Naunyn-Schmiedeberg's archives of pharmacology · 2026Article
- Pharmacovigilance assessment of drug-induced hypokalemia: from systemic risk screening to multi-dataset bioinformatics analysis focused on cisplatin.Naunyn-Schmiedeberg's archives of pharmacology · 2026Article
- Ruminococcaceae promotes the NLRP3/ASC/Caspase-1 axis-mediated pyroptotic signaling in papillary thyroid carcinoma by upregulating RBM15 to increase the N6-methyladenosine methylation modulation of NLRP3.Naunyn-Schmiedeberg's archives of pharmacology · 2026Article
- Salidroside Alleviates Fluoride-Induced Pyroptosis and Developmental Neurotoxicity Through P2X7R/NF-κB/NLRP3 Pathway.Biological trace element research · 2026Article
- ift140 -Deficient Zebrafish as a Model for Kidney Cystogenesis and an F0-Based Screen for Genetic Modifiers of Kidney Cysts.Journal of the American Society of Nephrology : JASN · 2026Article
- Nonmuscle α-Actinin-4 Couples Sarcomere Function to Cardiac Remodeling.Circulation research · 2026Article
- Increased Asialoglycoprotein Receptor 1 Level in Granulosa Cell as a Potential Biomarker for Polycystic Ovary Syndrome.Reproductive sciences (Thousand Oaks, Calif.) · 2026Article
- CD47 signaling in aging and age-related diseases: mechanisms, challenges, and therapeutic opportunities.Biogerontology · 2025Review
- Melt-Extruded Microparticles Based On Chitosan-pectin Complex for Delayed Dissolution of Benznidazole.AAPS PharmSciTech · 2025Article
Corrections and comments
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Authors and funding
9 authors at 2 institutions in 2 countries.
Funding
Abstract
Myosin heavy chain 7 (MYH7) is a major causative gene for hypertrophic cardiomyopathy, but the affected signaling pathways and therapeutics remain elusive. In this research, we identified ventricle myosin heavy chain like (vmhcl) as a zebrafish homolog of human MYH7, and we generated vmhcl frameshift mutants. We noted vmhcl-based embryonic cardiac dysfunction (VEC) in the vmhcl homozygous mutants and vmhcl-based adult cardiomyopathy (VAC) phenotypes in the vmhcl heterozygous mutants. Using the VEC model, we assessed 7 known cardiomyopathy signaling pathways pharmacologically and 11 candidate genes genetically via CRISPR/Cas9 genome editing technology based on microhomology-mediated end joining (MMEJ). Both studies converged on therapeutic benefits of mTOR or mitogen-activated protein kinase (MAPK) inhibition of VEC. While mTOR inhibition rescued the enlarged nuclear size of cardiomyocytes, MAPK inhibition restored the prolonged cell shape in the VEC model. The therapeutic effects of mTOR and MAPK inhibition were later validated in the VAC model. Together, vmhcl/myh7 loss of function is sufficient to induce cardiomyopathy in zebrafish. The VEC and VAC models in zebrafish are amenable to both efficient genetic and chemical genetic tools, offering a rapid in vivo platform for discovering candidate signaling pathways of MYH7 cardiomyopathy.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.