Congenital malformations of the brain are relatively common in dysmorphic infants.Ultrasound has become an important method for delineation of the intracranial malformation, which have altered macroscopic morphology. Ultrasound has the advantages of being rapid, requires neither sedation nor radiation and its portability allows imaging at the bedside. Ultrasound is cheaper and much less invasive for the infant, as it is a bedside technique which only takes 5-10 minutes and does not interrupt intensive care treatment. Cranial ultrasound can be repeated over the ensuing weeks to follow the evolution of the lesions which are present immediately after the delivery and are considered to be of antenatal onset.We studied 20 patients with craniofacial dysmorphism with or without other congenital anomalies, who were subjected to history taking, clinical examination, laboratory investigations, karyotyping, and sonographic study of their brain with C.T. & M.R.I. for some cases. We found patients with Chromosomal anomalies like trisomy 13,18 & 21 and other dysmorphic syndromes. Cranial ultrasound reveals partial or complete agenesis of corpus callosum in 5 cases, ventricular dilatation in 6 cases and brain atrophy in 1 case.