ArticleOrphanet journal of rare diseases2019
Triglyceride deposit cardiomyovasculopathy: a rare cardiovascular disorder.
Article in Orphanet journal of rare diseases, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 34 papers.
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34 citing papers in PubMed, 49 citations in OpenAlex.
- A prospective randomized study comparing effects of empagliflozin to sitagliptin on cardiac fat accumulation, cardiac function, and cardiac metabolism in patients with early-stage type 2 diabetes: the ASSET study.Cardiovascular diabetology · 2021Trial
- Triglyceride deposit cardiomyovasculopathy as a predictor of vascular failure after coronary intervention in patients with hemodialysis: a preliminary analysis.Renal failure · 2026Article
- Reexamining Fat: Exploring Diversity, Plasticity, Development, Functional Implication, and Therapeutic Options.International journal of molecular sciences · 2026Review
- Delineation of ventricular tachycardia isthmus using magnetic resonance imaging in a patient with idiopathic triglyceride deposit cardiomyovasculopathy.HeartRhythm case reports · 2025Article
- AAV9-mediated gene supplementation therapy prevents and rescues arrhythmogenic cardiomyopathy in Pnpla2-mutated mice.Molecular therapy : the journal of the American Society of Gene Therapy · 2025Article
- Genetic deletion or pharmacological inhibition of acetyl-CoA carboxylase 2 enhances fatty acid oxidation and improves cardiac function and survival in the murine ATGL knockout model of severe heart failure.Journal of molecular and cellular cardiology plus · 2025Article
- Chronic kidney disease is a major risk factor for mortality in triglyceride deposit cardiomyovasculopathy patients.Clinical and experimental nephrology · 2025Article
- Long-term survival and durable recovery of heart failure in patients with triglyceride deposit cardiomyovasculopathy treated with tricaprin.Nature cardiovascular research · 2025Article
- Distinct myocardial triglyceride lipolysis pathways in primary and idiopathic triglyceride deposit cardiomyovasculopathy.ESC heart failure · 2024Article
- Dynamic metabolism of endothelial triglycerides protects against atherosclerosis in mice.The Journal of clinical investigation · 2024Article
- Lipotoxicity as a therapeutic target in obesity and diabetic cardiomyopathy.Journal of pharmacy & pharmaceutical sciences : a publication of the Canadian Society for Pharmaceutical Sciences, Societe canadienne des sciences pharmaceutiques · 2024Review
- Carnitine Administration andAnnals of nuclear cardiology · 2024Article
- Practice recommendation for measuring washout rates inAnnals of nuclear medicine · 2024Article
- 1H-MRS to evaluate improved triglyceride accumulation in idiopathic triglyceride deposit cardiomyovasculopathy after CNT-01 (tricaprin/trisdecanion) administration.Endocrinology, diabetes & metabolism case reports · 2023Article
- A novel homozygous missense mutation inMolecular genetics and metabolism reports · 2023Article
- Practice Recommendation for Measuring Washout Rates inAnnals of nuclear cardiology · 2023Review
- Modified Algorithm Using Total Count for Calculating Myocardial Washout Rate in Single-Photon Emission Computerized Tomography.Annals of nuclear cardiology · 2023Article
- Methods of calculatingAnnals of nuclear medicine · 2022Article
- Clinical significance ofEuropean journal of nuclear medicine and molecular imaging · 2022Article
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Authors and funding
39 authors at 17 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Triglyceride deposit cardiomyovasculopathy (TGCV) is a phenotype primarily reported in patients carrying genetic mutations in PNPLA2 encoding adipose triglyceride lipase (ATGL) which releases long chain fatty acid (LCFA) as a major energy source by the intracellular TG hydrolysis. These patients suffered from intractable heart failure requiring cardiac transplantation. Moreover, we identified TGCV patients without PNPLA2 mutations based on pathological and clinical studies. We provided the diagnostic criteria, in which TGCV with and without PNPLA2 mutations were designated as primary TGCV (P-TGCV) and idiopathic TGCV (I-TGCV), respectively. We hereby report clinical profiles of TGCV patients. Between 2014 and 2018, 7 P-TGCV and 18 I-TGCV Japanese patients have been registered in the International Registry. Patients with I-TGCV, of which etiologies and causes are not known yet, suffered from adult-onset severe heart disease, including heart failure and coronary artery disease, associated with a marked reduction in ATGL activity and myocardial washout rate of LCFA tracer, as similar to those with P-TGCV. The present first registry-based study showed that TGCV is an intractable, at least at the moment, and heterogeneous cardiovascular disorder.
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