The authors describe four patients with unusual anatomic presentation of ectopic ureteroceles and their surgical treatment. Over a 3-year period, four cases of unusual ectopic ureteroceles were encountered. A 6-month-old girl had a complex cloacal anomaly with an ectopic ureterocele within the cloaca. A 10-year-old boy had two large diverticuli within an ectopic ureterocele combined with a blind-ending ipsilateral ureter. A 3-year-old girl had an ectopic ureterocele combined with a periureteral diverticulum and a completely duplicated ipsilateral kidney. A 4-year-old girl was found to have a vaginal ectopic ureterocele. Despite thorough radiological investigation in all patients, a correct assessment of the anatomic defect was achieved only by surgical exploration or endoscopic evaluation. If preoperative radiological evaluation is equivocal, a high index of suspicion and intraoperative recognition of an unusual anatomic presentation of the ectopic ureterocele are essential for appropriate management and a successful outcome.