Congenital toxoplasmosis is caused by vertical transmission of Toxoplasma gondii from mother during pregnancy. The broad clinical spectrum of congenital toxoplasmosis ranges from stillbirth or death shortly after birth to survival with either cerebral damage or mild or subclinical disease, consisting usually of ocular involvement. The authors experienced a case of congenital toxoplasmosis in prematurity baby whose mother had lived recently in Africa. The diagnosis of congenital toxoplasmosis was confirmed on the basis of clinical finding of chorioretinitis and positive serologic test for toxoplasma IgM antibody.