Pediatr Urol Case Rep 2021; 8(2):49-54DOI: 10.14534/j-pucr.2021267547Imaging based case report of ectopic urethral ureter in an infant: A case report and literature reviewKevin Nguyen, Mark Pisaneschi Department of Radiology, John H. Stroger Jr Cook County Hospital, Chicago, USA ABSTRACTWe present a case of ectopic urethral ureteral insertion in a 2-week old female diagnosed on a voiding cystourethrogram after abnormal prenatal and postnatal renal ultrasounds. The case is remarkable due to initial opacification of only the left upper pole moiety without bladder opacification on the voiding cystourethrogram due to direct canalization of the left ureter. Prompt diagnosis is important to prevent frequent urinary tract infections and urinary incontinence with lower ectopic urethral ureter insertions. Voiding cystourethrogram is an important tool in the workup of renal anomalies and is typically used to evaluate for reflux, however, keen attention can reveal other abnormalities such as ureteral ectopia in rare instances.