Fetal craniofacial malformations were identified sonographically in 13 cases. Holoprosencephaly and median cleft syndrome were the most frequent findings. Cleft lip, cleft palate, single nostril, cyclopia and hypotelorism were also seen. Seven of the 13 cases (54%) had polyhydramnios. Amniocentesis was performed on 12 fetuses, and 5 of them showed autosomal trisomy. Accordingly, when craniofacial malformation is recognized antenatally, a careful survey of the fetus for other associated structural anomalies and cytogenetic study are indicated to aid in diagnosis and subsequent obstetric and neonatal management. Also screening for fetal craniofacial malformation is recommended during mid-trimester ultrasound examinations.