The paper presents a case of ophthalmologic manifestations, episcleritis and retinal branch vein thrombosis, in a neonate born to a mother with antiphospholipid syndrome (APS) in the course of systemic lupus erythematosus. Female neonate (birth weight 1150 g, Apgar scores 6, 7 and 7) was born with respiratory distress syndrome, moderate anemia and grades I and II intraventricular hemorrhage. Ophthalmic examination revealed an enormous pre-retinal hemorrhage with accompanying thrombotic changes typical of retinal vein in the fundus of the left eye. Episcleritis was found in the anterior segment of the eye and later confirmed by ultrasound. Laboratory tests showed increased levels of maternal antibodies aCL IgG, antybeta2GP1 IgG and antybeta2GP1 IgM. No ANA, LA and SS-A/SS-B antibodies were detected. Increased concentrations of aCL IgG and a lengthening APTT were observed in newborn blood at first but the parameters normalized by 6 months of age. Our observations allowed us to conclude that early examination of neonates born to mothers with antiphospholipid syndrome is essential as it allows fast identification of pathological retinal changes by means of assessing the presence of aPL antibodies.