Complete penile agenesis (aphallia) is a very rare congenital anomaly. Around 80 cases have been reported to date. Diagnosis of this rare anomaly is made by clinical examination, but treatment options and parental counselling for gender assignment is difficult and challenging for the treating doctor. We report a case of an infant with complete penile agenesis with recto-urethral fistula, who was referred on day 6 of life. This baby developed obstructive uropathy which required vesicostomy. Surgical management consists of gender assignment after discussing in detail the pertinent situation with the parents and accepting their final decision.
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