Abstract
Cervical cerclage is a common procedure employed to prevent preterm birth in women with cervical insufficiency. Complications such as injuries to the cervix and bladder, and premature membrane rupture are well-documented, while genitourinary fistulas are a rare occurrence. This article reports a rare case of vesicovaginal fistula (VVF) formation in a 27-year-old woman following the placement of a McDonald cervical cerclage. The patient presented with continuous watery vaginal leakage, which began during the last 2 months of her pregnancy. Clinical and cystoscopic examinations revealed the presence of a VVF, which was further confirmed through voiding cystourethrography and perineal magnetic resonance imaging. Notably, the patient had undergone the cerclage procedure 18 months prior to the onset of symptoms, making this case particularly unusual. We believe that the VVF formation was associated with the use of Mersilene tape, which may have slowly eroded through the cervix and subsequently breached the urothelium. This case underscores the importance of considering cerclage-related genitourinary fistulas as potential complications, especially when evaluating and counseling patients who have undergone cervical procedures like the McDonald technique. In conclusion, this case highlights the need for vigilant monitoring and a high index of suspicion in patients presenting with symptoms of genitourinary fistulas after such procedures. Further research and awareness in this area are warranted to better understand the risk factors and mechanisms underlying this unusual complication.
Plain language summary
Our patient presented with continuous watery leakage that started during the last 2 months of her pregnancy. The patient was diagnosed with an abnormal communication between the bladder and vagina following the placement of cervical Mersilene tape. After 18 months, the opening was repaired, and the patient fully recovered 2 months after surgery.
Complications associated with cerclage are rare and commonly related to injuries to the cervix or bladder, premature rupture of membranes, chorioamnionitis, and bleeding, as described in published reports. This case report described the unusual complication of vesicovaginal fistula formation after the placement of a McDonald suture. We emphasize that vesicovaginal fistula can have a serious impact on social, mental, and sexual health. Any practitioner dealing with such patients should be aware of these associated health problems during management.
Keywords
Introduction
Women with recurrent second-trimester pregnancy loss are frequently treated with cervical cerclage. Cerclage placement in an incompetent cervix is generally considered safe; however, its efficacy was recently questioned.1,2 Moreover, there is little evidence from well-controlled surgical trials that cerclage is successful in reducing the incidence of premature delivery in such patients. 3 In fact, multiple complications of cerclage placement are reported, with the highest number of cases in the antepartum or intrapartum period. 4 Common complications associated with cerclage include injuries to the cervix or bladder, premature rupture of membranes, chorioamnionitis, and bleeding. Remote sequelae and genitourinary injuries owing to cerclage placement are infrequent, 5 and cerclage-related genitourinary fistulas are rarely mentioned in worldwide reports since they are exceedingly rare.
A vesicovaginal fistula (VVF) is a genitourinary fistulous tract formed between the bladder and vagina that causes continuous loss of urine via the vagina. In the developed world, 80% of VVFs occur secondary to benign gynecological surgeries. Of these VVFs, 8% occur after obstetrical procedures, which predominantly include cesarean deliveries, hemorrhage-related hysterectomies, and, less commonly, surgical vaginal deliveries. 6 There are few reports on cerclage-related VVFs. Furthermore, although urethrovaginal, vesicovaginal, vesicocervical, vesicouterine, and ureterovaginal fistulas are documented as complications of cerclage placement, there is no consensus on the causative factors.7–11 Here, we describe a case of a VVF (vesicovaginal fistula) that occurred as a complication of cerclage placement during pregnancy. We believe that these findings will be useful for obstetricians who encounter similar cases and will aid in the timely diagnosis and treatment of such cases.
Case presentation
Our patient was a 27-year-old woman in her third pregnancy. Her two previous children had been delivered vaginally; she had undergone one miscarriage. Her medical and surgical history were unremarkable. During her third pregnancy, the cerclage was placed prophylactically at 14 weeks of gestation because she had a history of cervical funneling and shortening during her second pregnancy. At approximately 32 weeks, she noticed intermittent episodes of vaginal wetness; however, there was no clear evidence of ruptured membranes on ultrasound examination or the AmniSure test (Qiagen, Hilden, Germany).
The cerclage was removed at 38 weeks and the patient gave birth normally without any evidence of urine leakage. She experienced persistent vaginal wetness after delivery. She sought medical advice several times over a period of 18 months and was informed that the incontinence was a result of normal vaginal delivery and would be resolved with time. She was treated with solifenacin (Vesicare) (Astellas Pharma Inc., Tokyo, Japan) for 3 months but her symptoms did not improve. She occasionally used vaginal tampons for temporary relief. She had undergone investigations at various hospitals; however, the results were inconclusive.
The patient was psychologically depressed upon presentation to our hospital. The patient was examined in the lithotomy position using a light source. A speculum was used to expose the anterior and posterior vaginal walls, and urine leakage from the anterior vaginal wall was observed. For further confirmation, the bladder was filled with 300 ml of sterile water and 20 ml of methylene blue dye. Spillage of blue dye into the vagina immediately occurred after instillation, which confirmed the presence of a VVF approximately 1 cm in diameter at the anterior upper third of the vaginal wall. High-resolution perineal magnetic resonance imaging revealed a linear tract in the upper third of the anterior vaginal wall on a T1-weighted image and contrast enhancement of the tract on a T2-weighted image.
The patient was taken to the operating room and a vaginal examination was performed using a Sims speculum under general anesthesia. A fistula was observed in the upper anterior vaginal wall. The bladder was injected with methylene blue, and leakage of stained urine was observed from the fistula tract (Figure 1). A pediatric Foley catheter (size 5 Fr) (Medline Industries Healthcare Catheters, North Field, USA) was inserted into the fistula (Figure 2). The cystoscopy was performed and the fistula opening was confirmed. The pediatric catheter was observed near the ureteral orifice (Figure 3).
Leakage of stained urine from the fistula tract.
Insertion of a pediatric Foley catheter (size 5 Fr) into the fistula.
Cystoscopic image showing the fistula opening near the ureteral orifice.
We decided to insert a double-J stent before the repair since the fistula was located approximately 0.5 cm from the ureteral orifice. Subsequently, the old scar tissue was excised to expose its edges. This was followed by transvaginal repair of the fistula using the multilayer technique, which involved closing the defect using interrupted (3–0) delayed absorbable sutures. No leakage of methylene blue from the bladder was observed during the closure. Postoperative care at the hospital included continuous bladder drainage with a Foley catheter along with daily antibiotic coverage. The patient was discharged from the hospital in good condition. A follow-up conducted 3 weeks later indicated that her symptoms had not recurred; therefore, the urine catheter and double-J stent were removed.
The patient expressed her satisfaction with the treatment outcome, which positively impacted her health and psychological status. She was very thankful to the team involved in her treatment.
Discussion
Herein, we describe an unusual case of VVF occurring after cerclage placement in a pregnant woman.
Urinary tract injury at the time of cerclage placement is unusual, although cases of vesicovaginal, 8 urethrovaginal, vesicocervical, 7 and ureterovaginal fistulae are reported. 11 In most cases where cervical cerclage led to a fistula, the women had undergone cerclage placement during a previous pregnancy, with the procedure typically being reported as difficult.9–12 A review of the literature from PubMed and Google Scholar from 1 January 1957 to 31 December 2021 revealed 11 different cases of urogenital fistulas caused by cervical cerclage placement (Table 1).
Reported cases of patients with a cerclage placed during their previous pregnancy who underwent genitourinary fistula evaluation.
LEEP: Loop electrosurgical excision procedure.
Genitourinary fistulas can occur after McDonald or Shirodkar cerclage placement, which are the two main techniques used for cerclage placement. However, most fistulae in the reviewed data occurred after placement using the McDonald technique. Currently, the technique is chosen based on surgeon preference and training; however, most physicians continue to favor the McDonald technique because of its ease of placement and removal. This may explain the markedly high frequency of fistulas observed using this technique. 21 The published cases for both techniques seem to report similar success and complication rates. However, no study has compared these two techniques, and little is known about their comparable risks and rates of fistula formation. 22 It remains unclear whether dissection of the vesicovaginal space (as done in the Shirodkar technique) may protect against inadvertent suture placement into or through the bladder wall, although cystoscopy after cerclage placement may be beneficial to identify suture placements that involve the bladder, especially in difficult cases.
The findings of the present case indicate the importance of carefully evaluating cases of fluid leakage in women who have undergone cerclage placement by considering the possibility of urinary tract injury in their differential diagnosis. It is also important for obstetricians to avoid the assumption that premature rupture of membranes is the only complication that may account for this phenomenon. This complication can have a serious impact on the social, mental, and sexual health of a patient; therefore, practitioners who deal with such patients should be aware of the signs of this complication during the management of the condition and consider different causes and differential diagnoses before determining the best possible line of management. This is especially important in developing countries, since the incidence of VVF in resource-limited countries is difficult to ascertain, and most studies have only reported the rates of fistulas associated with obstetric causes.
Conclusion
In the present case, the Mersilene tape was most likely placed close to the bladder and subsequently eroded the urothelium over the months that followed. Erosion of the epithelium likely led to intermittent urinary incontinence; over time, widening of the fistula caused continuous urinary leakage.
We acknowledge that many patients undergoing cerclage may have these same risk factors and do not develop a genitourinary fistula. Thus, further research using a large sample size may be warranted to confirm the conclusion.
