Abstract

Primary cyclic hematuria occurring with the onset of menstruation can be a presenting symptom of a fistula between the Mullerian structures (uterus/cervix/upper vagina) and the urinary tract. When occurring, these fistulas are most often vesicovaginal connections caused by congenital Mullerian anomalies. We present a case of a fistula occurring between a bicornuate uterus and the ureteral stump that had been ligated at the time of ureteral reimplantation during childhood. The presenting symptom was cyclic hematuria beginning at the age of 14 years with the onset of menarche. Cystoscopy and retrograde pyelogram showed a fistula connecting the ureteral stump (which had been ligated at the time of ureteral reimplantation) to the small horn of the bicornuate uterus. Computed tomographic scan showed bilateral single orthotopic kidneys and collecting systems. The patient underwent successful transvaginal and transvesical surgical repair of the fistula, with complete resolution of her cyclic hematuria. We review genitourinary embryology in discussion of this unusual case.

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