SummaryBackgroundHyperlipidemia is an identified factor of premature vessel atherosclerosis. Lipemia retinalis is an unusual retinal manifestation of hyperlipidemia and is thought to be directly correlated with the serum triglyceride level.Case ReportThis paper discusses the case of a 55-year-old patient with lipemia retinalis, which deteriorated his visual acuity. The patient had an extremely high serum cholesterol level (1053 mg/dl) and a very high level of triglycerides (1513 mg/dl). The normalization of serum lipids, reversion of retinal vessels alterations and visual acuity improvement was achieved after an intensive statin lipid-lowering therapy. Pathological changes of the patient’s retina, connected with lipemia retinalis, disappeared completely.ConclusionsHyperlipidemia can cause lipemia retinalis, which is characterized by the hyperlipidemic vascular lesions-whitish color of vessels, lipid infiltration into the retina and decrease of visual acuity. The lipid-lowering therapy may lead to the normalization of the appearance of the fundus and restore the visual acuity.