Plant sterol-enriched margarine consumption is known to significantly reduce concentrations of plasma low density lipoprotein (LDL) cholesterol. However, there is no evidence that such margarines may reduce cardiovascular events. This is the first report of a significant increase in the plasma concentration of phytosterols due to plant sterol-enriched margarine consumption leading to extravascular lipid deposits (xanthelasma), in a woman with hypercholesterolaemia. Because increased plasma levels of phytosterols may promote atherosclerosis, the present observation raises concern about potential cardiovascular risk after long-term consumption of these margarines.