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Background: A great concerns have been obtained for CNS anomalies due to their maternal and fetal morbidities and mortalities outcomes. The high prevalence of CNS anomalies has been linked to polyhydramnios in several studies. This study was conducted to evaluate the role of 3D/4D US in detection of CNS anomalies as compared to other non-CNS anomalies in pregnancy complicated with polyhydramnios. Methods: This was a cohort study conducted at the radiology department of University Hospital. The study was done on 110 pregnant females diagnosed with polyhydramnios. Regular visits and follow-up of patients were scheduled till either delivery or termination of pregnancy. Results: The most common fetal anomalies were CNS anomalies detected in 20 patients (18.18%) followed by musculoskeletal anomalies in 6 patients (5.5%) then cardiovascular anomalies in 5 patients (4.5%). Chiari malformation represented the most common malformation affecting about 6 fetuses (5.5%) of all the participants. There was a significant agreement between antenatal diagnosis of congenital anomalies as detected by 3D/4D US and postnatal confirmation of the diagnosis. Conclusions: The gold standard technique in detection of fetal anomalies is 3D/4D ultrasonography with high accuracy especially in detection of CNS malformations. Also, the incidence of CNS malformation in pregnant females complicated with polyhydramnios is more than other non-CNS anomalies. [ABSTRACT FROM AUTHOR]