Background:Congenital anomalies of the genitourinary system are found in approximately 10% of infants. Congenital anomalies of kidney and urinary tract may be of different types such as, ureteropelvic junction obstruction,multicystic dysplastic kidneys, hypoplastic kidneys, exstrophy of the bladder etc.,those of the female genital tract may be dysmorphic uterus, septate uterus, bicorporeal uterus, septate cervix, vaginal septum etc. and those of the male genital tract may be hypospadias, epispadias, cryptorchidismetc. The congenital anomalies of genitourinary system may be responsible for different types of reproductive problems. Hence, the present work has been carried out to study the congenital malformations of genitourinary system in perinates. Methods: The study was carried out in all consecutive born perinates, total 7985 births (both live and still born), from 28^th weeks of gestation to 7 days after birth, collected from the Department of Obstetrics and Gynaecology, Assam Medical College and Hospital, Dibrugarh, Assam, during 2014–16.After collection, the babies were examined and informations regarding congenital anomalies were noted in a pretested structured proforma, after taking written consents from the parents.Statistical calculations were done and the results were presented in tabular form. Results: In the present study, out of 7985 births, nine cases showed congenital anomalies of genitourinary system (1.127/1000 live births). Out of these nine cases, three cases showed undescended testis (bilateral), two cases showed polycystic kidney (one bilateral and one unilateral), two cases showed incomplete duplication of the ureter (unilateral), one case showed penile duplication with accessory perineal scrotum and one caseshowed exstrophy of the urinary bladder. Conclusion: The incidence of congenital malformations, in the present study, was comparatively lower than the previous studies, which may help in the management of these anomalies.