Affiliations: Department of Pediatrics, Section of Neonatology, University of Calgary, AB, Canada | Alberta Children's Hospital Research Institute, University of Calgary, AB, Canada
Note:  Corresponding author: Dr. Prakash Loganathan, Foothills Medical Centre, Rm-C-211, 1403, 29th Street NW, Calgary, Alberta T2N 2T9, Canada. Tel.: +1 403 944 1087; Fax: +1 403 944 4892; E-mail: Prakash.email@example.com
Abstract: BACKGROUND: Persistent urogenital sinus is one of the rare urogenital anomalies, which commonly presents as hydrometrocolpos. Fetal urinary ascites as a presentation of persistent urogenital sinus is extremely rare. CASE REPORT: We report on a preterm infant with antenatal diagnosis of hydrometrocolpos and massive urinary ascites secondary to urogenital sinus without any bladder or renal abnormalities. CONCLUSION: This case report emphasizes the importance of maintaining a high index of suspicion in the diagnosis of persistent urogenital sinus especially in infants presenting with urinary ascites along with hydrometrocolpos.