Journal of pediatric surgery · 2018
Congenital prepubic sinus-An aborted dorsal urethral duplication or a cloacal remnant?
Клинический случайСтатьяНазвание и аннотация: Перевод в очереди
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Nazir Zafar, Khan Muhammad Arif Mateen, Qamar Javaria
Аннотация
BACKGROUND: Congenital Prepubic Sinus (CPS) is an uncommon urogenital anomaly characterized by a blind tract between the skin over the pubis to anterior of the urinary bladder, Urethra or umbilicus. We report four such cases to emphasize varied clinical presentation and embryological conundrum.
METHODS: Following Ethical Review Committee (ERC) approval, medical records of pediatric patients (<16 years) presenting with CPS (identified through operating room records and Hospital Information Management System (HIMS) between 1994 and 2018 were reviewed for demographics, clinical presentation, investigations including histopathology, management and outcome.
RESULTS: Four cases of CPS, 3 females and 1 male, age range 9 months to 13 years were managed over 25-years. Clinical presentation includes a discharging sinus and recurrent episodes of cellulitis and abscess formation in pubic area and labia majora. Urological investigations were mostly normal. Insertion of lacrimal probe or plastic sheath of intravenous cannula through the sinus opening was useful to determine the course of sinus and aid its excision. Histology of excised sinus highlights the possible embryological origin.
CONCLUSIONS: CPS is a rare anomaly with varied clinical presentation. It seems CPS is an aborted urethral duplication (Stephen Type 3) or a Cloacal remnant. Complete excision of the tract in the reported cases was curative.
TYPE OF STUDY: Case series.
LEVEL OF EVIDENCE: IV.
METHODS: Following Ethical Review Committee (ERC) approval, medical records of pediatric patients (<16 years) presenting with CPS (identified through operating room records and Hospital Information Management System (HIMS) between 1994 and 2018 were reviewed for demographics, clinical presentation, investigations including histopathology, management and outcome.
RESULTS: Four cases of CPS, 3 females and 1 male, age range 9 months to 13 years were managed over 25-years. Clinical presentation includes a discharging sinus and recurrent episodes of cellulitis and abscess formation in pubic area and labia majora. Urological investigations were mostly normal. Insertion of lacrimal probe or plastic sheath of intravenous cannula through the sinus opening was useful to determine the course of sinus and aid its excision. Histology of excised sinus highlights the possible embryological origin.
CONCLUSIONS: CPS is a rare anomaly with varied clinical presentation. It seems CPS is an aborted urethral duplication (Stephen Type 3) or a Cloacal remnant. Complete excision of the tract in the reported cases was curative.
TYPE OF STUDY: Case series.
LEVEL OF EVIDENCE: IV.