CASE REPORT

Report of 2 Cases of Misdiagnosed Vesicouterine

Andrey Petrikovets, MD and Pierre F. Lespinasse, MD

catheter was placed. The bladder was adherent to the uterine Abstract: Vesicouterine fistula is a rare complication that may occur isthmus and the anterior aspect of the . A clam cystotomy after multiple cesarean deliveries. The following reports describe cases was made along the dome of the bladder, and the fistula tract where vesicouterine fistula was misdiagnosed; one was initially treated was identified. The fistula was then elevated with Allis clamps, for urge incontinence, and the other was treated for stress urinary and a scalpel was used to sharply dissect the fistula tract from incontinence. the bladder and . After the was performed, Key Words: vesicouterine fistula, menouria, urogenital fistula, stress the fistula was repaired in a 2-layer fashion. The first layer was urinary incontinence, urge incontinence closed with 2-0 Vicryl in a running fashion and the second layer Y with 2-0 Vicryl in an interrupted fashion. The clam cystotomy (Female Pelvic Med Reconstr Surg 2014;00: 00 00) incision was closed in a similar fashion. The bladder was then backfilled in a retrograde fashion with about 200 mL of saline, BACKGROUND and no extravasation of saline was identified. An omental J flap The increasing rate of cesarean delivery in the United States was placed between the bladder and the vaginal cuff. has led to an increasing prevalence of associated complications.1 The patient was discharged home on postoperative day 3 One infrequent complication after repeat cesarean deliveries is with a leg bag and Macrobid 100 mg by mouth daily for pro- a vesicouterine fistula, a pathologic communication between phylaxis for 15 days. The leg bag was removed 15 days later, the bladder and the uterus. Although case reports of vesicouterine and the patient had a successful voiding trial. Postoperatively, fistula have been often associated with repeat cesarean deliver- the patient has been doing well and has been asymptomatic for ies, it has also been diagnosed as a complication of pelvic 3 years. trauma after operative deliveries, malignancy, radiation, intra- uterine device placement, placenta percreta, uterine artery em- bolization, and more recently, endometrial ablation.2Y7 CASE 2 The classic presentation of vesicouterine fistula, described A 40-year-old woman (gravida 3, para 3) with 3 prior ce- as Youssef syndrome, consists of menouria with cyclic hematuria sarean deliveries presented to another physician complaining of and . It is a challenging diagnosis that describes about involuntary loss of with straining after her last cesarean 4% of all urogenital .8 The purpose of this report is to delivery, which was complicated by an inadvertent cystotomy. broaden the perspective of the different presentations of vesi- Her symptoms started 3 months after the cesarean delivery. She couterine fistula. The first case demonstrates vesicouterine fis- was evaluated, and her condition was diagnosed with stress uri- tula initially treated as urge incontinence, and the second case nary incontinence (SUI). The patient underwent a transobturator demonstrates the presentation as stress incontinence. sling procedure for SUI 12 months after her cesarean delivery. Postoperatively, her symptoms worsened. She was evaluated at CASE 1 another institution where urodynamics were negative for SUI. Cystoscopy and VCUG findings were normal as well. Because of A 60-year-old woman (gravida 3, para 3; last menstrual persistent leakage of urine, she presented to our institution a year period, 1992) with 3 prior cesarean deliveries presented to our and a half after she underwent the midurethral sling procedure. institution with involuntary loss of urine for 25 years after her Review of symptoms revealed blood in her urine with last cesarean delivery. During this course of time, she was seen menses, menorrhagia, and persistent loss of urine with coughing by multiple providers and was treated for urge incontinence and sneezing. The result of her physical examination was signif- with anticholinergic medications with limited improvement. Upon icant for leakage of urine with and without Valsalva maneuver, presenting to our office, the patient complained that her urinary stage I , a normal-sized uterus, and clear fluid in the frequency worsened during the course of the previous 5 years, vaginal vault. The vaginal fluid creatinine level was 22.4 mg/dL, often voiding every 30 minutes requiring her to wear diapers. and the hemoglobin level was 12.6 g/dL. The result of a methy- The physical examination revealed an atrophic vagina with clear lene blue dye test performed during a follow-up visit was initi- fluid pooling in the posterior fornix, no evidence of incontinence ally negative. Only after the patient voided did we identify dye from the with Valsalva maneuver, and a normal-sized leaking from her cervical os and not from the anterior vaginal uterus. Voiding cystourethrogram (VCUG) demonstrated a vesi- wall. Voiding cystourethrogram exhibited likely vesicouterine couterine fistula between the bladder dome and the uterus. fistula with contrast found in the vagina (Fig. 4). The patient underwent a total abdominal hysterectomy Y The patient underwent a robot-assisted hysterectomy with with fistula repair (Figs. 1 3). She received 1 dose of Levaquin vesicouterine fistula repair. She received 1 dose of Levaquin 500 mg intravenous and 1 dose of clindamycin 900 mg intra- 500 mg intravenous and 1 dose of clindamycin 900 mg intra- venous for surgical prophylaxis. Intraoperatively, a 3-way Foley venous for surgical prophylaxis. Intraoperatively, the uterus was From the Rutgers-New Jersey Medical School. densely adherent to the bladder dome. After careful dissection Reprints: Andrey Petrikovets, MD, of the uterus from the bladder, the fistula tract was identified. Rutgers, New Jersey Medical School, 185 South Orange Avenue, E-506 The margins of the fistula were trimmed, and the defect was Newark, NJ 07103. closed with 2-0 Vicryl in a running fashion. After the first layer E-mail: [email protected]; [email protected]. The authors have declared they have no conflicts of interest. was closed, the bladder was backfilled with 200 to 300 mL of Copyright * 2014 by Lippincott Williams & Wilkins saline using a 3-way Foley catheter with no spillage of saline DOI: 10.1097/SPV.0000000000000123 noted. A second layer, using 2-0 Vicryl, was approximated in an

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FIGURE 1. Probe through fistula tract. FIGURE 3. Intraoperative view of the bladder with probe in the bladder through the fistula tract. interrupted fashion with no evidence of saline extravasation after repeat backfilling. In comparison to the first case, we were incision. Often with repeat cesarean sections, there is a consider- unable to use the omentum to create an omental J flap because it able amount of scar tissue in this area, and there is an increased was too short. risk of identified and/or unidentified bladder injuries. Perhaps, She was discharged home on postoperative day 1 with a leg these complications can be prevented by performing a trial of bag and Macrobid 100 mg by mouth daily for 20 days for labor after cesarean delivery, decreasing the rate of elective ce- prophylaxis. The catheter was removed on postoperative day 20 sarean deliveries and generally avoiding the primary cesarean with a successful voiding trial. Postoperatively, she has been delivery. It is prudent to consider the possibility of a doing well for 2 and a half years without recurrence of her vesicouterine fistula in a patient who presents complaining symptoms. amenorrhea, cyclic hematuria, menouria, and pooling of vaginal fluid after multiple cesarean deliveries. DISCUSSION There are steps that should be taken in the evaluation of In 1957, Youssef 9 described vesicouterine fistula as a con- a suspected vesicouterine fistula. After the patients’ history and stellation of amenorrhea, cyclic hematuria, and menouria with or review of systems are elicited, a physical examination is per- without urinary incontinence, known as the Youssef syndrome. formed. If there is pooling of clear fluid, that is, urine in the This classic combination of symptoms depends on the location vagina, the patient may have a fistula. The fluid is then sent to of the fistula in relation to the uterine isthmus and the corre- determine its creatinine level. sponding pressure gradients. During normal menses, blood ac- If the creatinine level is similar to the serum, the fluid is cumulates over the uterine isthmus, and when the pressure is unlikely urine because the concentrates creati- greater than 25 mm Hg, the isthmus dilates, allowing menses to nine, resulting to a higher level than the serum. If the value is occur.10 Yearsafter Youssefdescribed the typical presentation of high, the creatinine has been concentrated by the urinary system, vesicouterine fistulas, Jozwik and Jozwik11 subclassified vesico- and a fistula is likely. A methylene blue dye test is also performed uterine fistulas into 3 types as follows: type I with menouria, type by retrograde filling of the bladder with È200 to 300 mL of II with flow from both uterus and the vagina, and type III with methylene blue. A speculum examination is then performed to normal menses. In Youssef syndrome, the fistula is found directly see if there is extravasation of methylene blue dye in the vagina. above the uterine isthmus and results in a type 1 vesicouterine If there is no extravasation of dye in the vagina, a tampon is fistula, the most common presentation. placed, and the patient is instructed to void. Increased bladder The increasing rate of cesarean delivery has led to an in- pressure during voiding creates a pressure gradient between the creased prevalence of vesicouterine fistulas.12 Multiple cesarean fistula tract and the bladder, thereby increasing the likelihood of deliveries are a risk factor for the development of vesicouterine dye extravasation through the fistula tract. In the case of a fis- fistulas because there is increased bladder dissection during the tula, the tampon is stained with blue dye, but the location of the cesarean section to develop a bladder flap before the uterine fistula tract is still unknown. The tampon is then removed, and a speculum examination is performed again. In a vesicouterine

FIGURE 2. Probe showing fistula tract between cervix and FIGURE 4. Voiding cystourethrogram with fistula between the bladder (dissected off). uterus and bladder.

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Copyright © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited. Female Pelvic Medicine & Reconstructive Surgery & Volume 00, Number 00, Month 2014 Two Misdiagnosed Vesicouterine Fistula fistula, there will be extravasation of dye from the cervix as REFERENCES seen in the second case. AVCUG is performed as a confirmatory 1. Minkoff H. Fear of litigation and cesarean section rates. Semin Perinatol test to locate the level of the fistula. With a VCUG, if the fistula 2012;36(1):390Y394. is small, the leakage will be significant if there is a pressure gradient, such as after voiding. Because the fistulas are often 2. Pelosi MA 3rd, Pelosi MA. Vesicouterine fistula: a rare complication of vaginal birth after cesarean. Obstet Gynecol 1996;87:160Y162. small, these defects may not be evident on cystoscopy without creating a pressure gradient. Cystoscopy will also not differen- 3. Gil A, Sultana CJ. 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