New England Journal of Medicine · 1980 · 248 citations · 24 references
Life SciencesBiochemistryPharmacologyHuman NutritionMacronutrient MetabolismPediatric CardiologyLipid Metabolism DisordersMetabolic SyndromeCardiac Fatty Acid OxidationCardiac Size MeasurementFatty Acid Oxidation DisordersGalactose-1-phosphate Uridylyltransferase DeficiencyKetone Body Utilization In Muscle
A 3 1/2-year-old boy presented at three months of age with an acute episode of lethargy, somnolence, hypoglycemia, hepatomegaly, and cardiomegaly, which responded poorly to restoration of the blood sugar level to normal. The absence of ketonuria during subsequent episodes of severe hypoglycemia prompted a search for a defect in fatty acid oxidation. Plasma carnitine (2.0 to 5.0 mumol per liter), muscle carnitine (0.01 to 0.02 mumol per gram, wet weight) and liver carnitine (0.021 to 0.065 mumol per gram, wet weight) were all less than 5 per cent of the normal mean. During a 36-hour fast, ketones were barely detectable. Prolonged treatment with oral carnitine over a six-month period resulted in increased muscle strength, a dramatic reduction in cardiac size, relief of cardiomyopathy, partial repletion of carnitine levels in plasma and muscle, and complete repletion in the liver. Systemic carnitine deficiency is an easily treatable cause of recurrent Reye's-like syndrome. Its diagnosis requires measurement of carnitine levels.
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The syndrome of systemic carnitine deficiency
George Karpati, Stirling Carpenter, Andrew G. Engel et al. · Neurology · 1975 · 400 citations