ArticleDiabetes, metabolic syndrome and obesity : targets and therapy2020
Early Left Ventricular Systolic Dysfunction Detected by Two-Dimensional Speckle-Tracking Echocardiography in Young Patients with Congenital Generalized Lipodystrophy.
Article in Diabetes, metabolic syndrome and obesity : targets and therapy, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.
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7 citing papers in PubMed, 9 citations in OpenAlex.
- The insulin resistance-systemic vascular resistance-isolated diastolic hypertension axis: a metabolic framework for an overlooked hypertension phenotype.Frontiers in cardiovascular medicine · 2026Review
- Comprehensive analysis of morbidity and mortality patterns in familial partial lipodystrophy patients: insights from a population study.Frontiers in endocrinology · 2024Article
- Echocardiographic Alterations of Cardiac Geometry and Function in Patients with Familial Partial Lipodystrophy.Arquivos brasileiros de cardiologia · 2024Article
- Case report: Echocardiographic diagnosis of cardiac involvement caused by congenital generalized lipodystrophy in an infant.Frontiers in pediatrics · 2023Article
- Validity of resting strain/strain rate in prediction of myocardial viability.The international journal of cardiovascular imaging · 2022Article
- SARS-COV-2 infection outcomes in patients with congenital generalized lipodystrophy.Diabetology & metabolic syndrome · 2021Article
- Leu124Serfs*26, a novelDiabetology & metabolic syndrome · 2020Article
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17 authors at 2 institutions in 1 country.
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Abstract
purposeCongenital generalized lipodystrophy (CGL) is a rare autosomal recessive disorder characterized by the absence of functional adipocytes resulting in ectopic lipid storage, metabolic disorders and early cardiovascular disease. Two-dimensional speckle-tracking (2D-STE) allows the detection of early abnormalities in myocardial function. We aimed to evaluate myocardial deformation in a large sample of CGL patients using 2D-STE. PATIENTS AND
methodsA cross-sectional study of 22 patients with CGL and 22 healthy subjects, matched for sex and age, was conducted from 2013 to 2018. All participants had undergone standard conventional echocardiography (ECHO) and 2D-STE. Determination of blood glucose, lipids, insulin, and leptin were performed in all CGL patients.
resultsIn the CGL group the mean age was 14.6±10.7 years where 68.2% (n=15) were younger than 18 years old. All the patients had hypoleptinemia, 95.4% (21/22) low HDL-c, 86.4% (19/22) hypertriglyceridemia, 68.2% (15/22) diabetes, 50% (11/22) hepatic steatosis, 41% (9/22) insulin resistance, 41% (9/22) hypercholesterolemia, and 18.2% (4/22) hypertension. ECHO showed that 36.6% (8/22) of CGL patients presented diastolic dysfunction, 31.8% (7/22) left ventricular hypertrophy (LVH), 27.3% (6/22) increased left atrial volume index (LAVI), and 18.2% (4/22) increased left ventricular systolic diameter (LVDS) but normal ejection fraction (EF), whether using 2D-STE, 68.2% (15/22) of CGL patients showed abnormal global longitudinal strain (GLS) (p<0.01), and in almost LV segments. Positive association between abnormal GLS and A1c (r=0.57, p=0.005), glucose (r=0.5, p=0.018) and basal insulin (r= 0.69, p= 0.024), and negative association with leptin (r = -0.51, p = 0.005) were found in these patients.
conclusionThe 2D-STE revealed precocious left ventricular systolic dysfunction in a young CGL population with normal systolic function by ECHO. Early exposure to common metabolic abnormalities as insulin resistance, hyperglycemia, and hypoleptinemia must be involved in myocardial damage in these patients.
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