Youssef's syndrome has been first described in 1957 as an atypical presentation of a vesicouterine fistula after lower segment Cesarean section. It is characterized by a triad of cyclic hematuria, amenorrhea, and absence of urinary incontinence, which is usually found in other forms of genitourinary fistulas. We describe a case report of a woman who developed a delayed Youssef's syndrome 3 months after her third Cesarean section. She was initially post-operatively treated conservatively and successfully for a bladder leakage through the wound with a bladder catheter for 1 month, and 2 months later, she developed symptoms as described above. She was successfully treated with uterus-sparing surgical fistula repair. Youssef's syndrome is a rare and atypical presentation. Physicians may easily be misled and diagnosis delayed. We describe the diagnostic approach and review the literature as to therapeutic approaches.
Keywords: Amenorrhea; Continence; Fistula; Hematuria.
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