Spontaneous vesicovaginal fistula caused by genitourinary aspergillosis

Genitourinary aspergillosis is rare in patients who are not immunocompromised. A 39-year-old woman (para 3) presented with vaginal urinary leakage with no history of previous trauma, procedure, or fever. The patient's last delivery had been by cesarean 12 years previously and she had no history of difficult labor. Six months prior to presenting, the patient had been scheduled to undergo abdominal hysterectomy for menorrhagia along with repair of a surgical hernia; only the hernia was repaired at that time as dense adhesions prevented the hysterectomy. The patient had been amenorrheic since that surgery and her incontinence began 6 days prior to presentation. A speculum could only be inserted 2–3 cm into the vagina because it was tender and inelastic. Examination revealed urine leakage and an indurated anterior vaginal wall with a 4-cm irregular growth (Fig. 1). Vaginal examination was painful and the growth was tender, nonfriable, hard, and nodular. The uterus was anteverted, comparable in size to 6–8 weeks, firm, and with bilateral parametrial thickening and tenderness. Histopathology of a biopsy of the lesion revealed invasive aspergillosis. Routine investigations such as CD4 cell count for immunosuppression, sweat chloride levels, urine examination, and vaginal swab culture for fungus were normal. Intravenous pyelography revealed a normal left kidney, a vesicovaginal fistula, and a nonfunctioning right kidney, confirmed by a renal scan. A computed tomography scan showed severe right hydroureteronephrosis. Cystoscopy was not performed because of infection. Speculum examination showing growth and fistula mimicking carcinoma of the cervix. The patient received 0.5 mg/kg per day of amphotericin B intravenously for 30 days, and the vaginal growth reduced markedly. A dye test confirmed a high lying vesicovaginal fistula. On cystoscopy, the bladder could not be distended because of a large trigonal fistula and the ureteric orifices could not be seen. The patient received 200 mg of ketoconazole twice daily. Three months later the growth had disappeared and the vagina was no longer indurated. Repeat biopsy was negative for aspergillosis. The patient underwent total abdominal hysterectomy, right salpingo-oophorectomy, appendectomy, right nephrectomy, and abdominal vesicovaginal fistula repair. Dense omental adhesions were observed intraoperatively and separated. A large posterior bladder and anterior vaginal wall defect were seen when the bladder was dissected. Bladder tissues were friable. The cervix was completely obliterated and the right adnexa formed a mass with the appendix. The vagina was mobilized with difficulty 1.5–2 cm below the fistula. The lower right ureter was completely destroyed and found to be detached from the bladder. The left ureter and ureteric orifice were normal. The bladder wall and vagina were repaired. The omentum was interposed between the vagina and bladder, and the bladder was drained via the urethra. Histopathology revealed aspergillosis in the kidney. The ureter showed submucosal chronic inflammation with muscle hypertrophy. The appendix showed recurrent appendicitis. Giant cells were observed in the lower uterus. The tubo-ovarian mass had foreign body giant cells with aspergillus hyphae. Ketoconazole was continued for 6 weeks postoperatively and the patient's recovery was uneventful. The silicone catheter was removed at 4 weeks. At follow-up the patient was continent and able to hold urine for nearly 2 hours. Vesicovaginal fistula is rarely caused by proven fungal infection, although cases have been reported with tuberculosis [1]. In low-income countries, obstructed labor causes 50% to 60% of all vesicovaginal fistulas. Infection is responsible for a much smaller percentage (0.74%). Genitourinary aspergillosis is rare with only a few cases reported [2], [3]. The abdominal route was chosen for repair as the fistula was large and the ureteric orifices were not visualized on cystoscopy. Omental interposition was done to increase vascularity of margins and also because it works like a physiological drain. Antifungal drugs were continued postoperatively to treat residual infection. Oral itraconazole, which has shown better results than amphotericin B for invasive aspergillosis, is unavailable in India.