We described a neonate with severe and multiple abnormalities following use of isotretinoin, in spite of the mother's previous knowledge of drug complications. Initial physical examination after delivery showed congenital absence of both eyes and both auricles as well as anal atresia and a cleft palate. Due to severe respiratory distress and atresia of the anus, the neonate was admitted to a neonatal intensive care unit and underwent reconstructive surgery. The drug should not have been used during pregnancy. Using this drug within pregnancy with awareness of its side effects may have legal consequences.