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Department of Neurosurgery, Ondokuz Mayis University Samsun, Türkiye Klippel - Trenaunay syndrome is a congenital angiodysplasia characterized by varicose veins, cutaeous haemangiomas and bony and soft tissue hypertrophy. This report describes a case of Klippel- Trenaunay Syndrome with dilated tortuous vessels in the retina and left occipital infaret . Keywords : Cerebral infaret. Klippel-Trenaunay Sydrome, Retinal Involvement