Obstetric and Gynecologic Genitourinary Fistulas

MEGAN ABRAMS, MD, MPH,* and RACHEL POPE, MD, MPH† *Department of Female Pelvic Medicine and Reconstructive Surgery, University Hospitals Cleveland Medical Center; and †Urology Institute, University Hospitals Cleveland Medical Center, Cleveland, Ohio

Abstract: Urinary incontinence shortly after childbirth can be multifactorial and confusing to the or gynecologic surgery can be the result of obstetric or unsuspecting medical provider. While ves- iatrogenic fistula formation. This can be a confusing and challenging diagnosis for medical providers. While the icovaginal fistulas are rare, they occur after number of iatrogenic fistula cases is rising worldwide, 0.08% of hysterectomies in the United obstetric fistulas are an issue uniquely particular to States.1 Ureteral injury which may result resource poor settings. Appropriate treatment of genito- in ureterovaginal fistulas occur in 0.02% to urinary fistulas spans beyond surgical intervention of 0.33% of hysterectomies.2 Globally, genito- leakage, and includes re-integration into the community, widespread education and counseling, and battling social urinary fistulas occur at much higher rates. stigma and cultural biases. Current and future research Approximately 2 million women cur- must focus on rigorous, unified efforts to set evidence- rently live with an obstetric-related urinary based practices to help the millions of women affected. fistula. One systematic review found an Key words: obstetric fistula, vesicovaginal fistula, incidence of up to 4 cases of obstetric fistula iatrogenic fistula per 1000 deliveries and a prevalence of up to 81 obstetric fistula cases per 1000 women.3 While these estimates are on the higher end, there is a lack of robust data on incidence Introduction and prevalence. Urinary incontinence presenting shortly As the quality of obstetric care increases after childbirth or gynecologic surgery globally through the improved access to cesarean deliveries, iatrogenic fistulas ap- Correspondence: Rachel Pope, MD, MPH, Urology pear to increase. However, because of the Institute, University Hospitals Cleveland Medical Cen- ter, Cleveland, OH. E-mail: rachel.pope@uhhospitals. lack of data on fistulas in general, we do not org know if iatrogenic cases outnumber obstet- The authors declare that they have nothing to disclose. ric cases. Therefore, improved access to

CLINICAL OBSTETRICS AND GYNECOLOGY / VOLUME 64 / NUMBER 2 / JUNE 2021

www.clinicalobgyn.com | 321 Copyright r 2021 Wolters Kluwer Health, Inc. All rights reserved. 322 Abrams and Pope surgery and enhanced surgical technique are intraoperative ureteral injuries, including necessary across the board. lacerations, transection, crushing, avul- sion, suture ligation, or ischemia, lead to urinomas, and potential drainage from Gynecologic Genitourinary the vaginal cuff, leading to a ureterovaginal fistula. This is most often the lower Fistulas third of the ureter, and may be the result While obstetric fistulas are relatively un- of bleeding intraoperatively which ob- known in resource-rich settings, iatrogenic scures the operative field, a markedly genitourinary fistulas are known complica- enlarged uterus, or pelvic adhesions be- tion of gynecologic surgeries, and recent cause of prior surgery. studies have shown a growing incidence of Diagnosing an iatrogenic fistula requires iatrogenic fistula throughout the world.4 a thorough history, physical exam, and a Genitorurinary fistulas can occur during ob- high level of suspicion. Timing and presen- stetric or gynecologic surgery because of the tation vary widely from patient to patient close proximity of the bladder, ureters, uterus, and is dependent on etiology and location of and vagina and cause an abnormal commu- the injury. Some fistulas present immedi- nication between the bladder or ureter and ately after inciting trauma with leakage of the uterus, cervix, or vagina. While any urine from the vagina; however, iatrogenic surgery carries the risk of injury to nearby fistulas resulting from surgical intervention structures, fistula formation is a known maytakeupto30dayspostoperativelyto complication of several obstetric and gyneco- present.5 This is because of the slower logic procedures. Common obstetric and process of devascularization as a result gynecologic procedures, which may result in suture,clamp,orthermalinjury,which genitourinary fistulas include cesarean deliv- leads to necrosis and tract formation over ery, uterine rupture, and hysterectomy.4 time. Iatrogenic fistulas because of other Postsurgical fistulas tend to be small, causes, like radiation therapy, may take isolated, and surrounded by healthy tis- months or years to develop. The evaluation sue. In the United States, 80% of vesico- of size, number, and exact location of the vaginal fistulas are caused by benign fistula is important for diagnosis and surgi- gynecologic surgeries.5 Urinary tract in- cal planning. juries occur in ∼3/1000 gynecologic surgeries.5 Other risk-factors include a DIAGNOSING VESICOVAGINAL AND history of pelvic irradiation, gynecologic URETEROVAGINAL FISTULAS malignancy, endometriosis, pelvic inflam- Patients with a vesicovaginal and ureter- matory disease, infection, trauma, foreign ovaginal fistulas typically present several bodies, or history of pelvic surgery.6 days to months after a pelvic operation with Possible mechanisms of fistula devel- continuous leakage of urine through the opment include direct injury to the tissue vagina. This warrants a prompt physical during a surgical procedure or suboptimal exam. Using a speculum, the vagina should placement or use of surgical instruments be carefully inspected. If a hole or defect is while dissecting or clamping. Sutures identified, the surrounding mucosa may be placed too close to the bladder or ureter erythematous or inflamed. Patient rarely may also lead to necrosis and fistula cannot tolerate office exam and require formation. Though rare, fistula formation anesthesia. A careful physical exam is nec- has also been reported after uterine per- essary to evaluate for acute inflammation, foration at the time of dilation and cur- edema, necrosis, or other bladder pathol- retage or hysteroscopy or after procedures ogy, any of which may delay surgery. utilizing synthetic mesh.6 Unrecognized Evidence of scarring, fixation to adjacent www.clinicalobgyn.com Copyright r 2021 Wolters Kluwer Health, Inc. All rights reserved. Obstetric and Gynecologic Fistulas 323 organs, or postirradiation involvement may and inspected to identify suspected vesico- alter the surgical approach. If a vesicovagi- vaginal, ureterovaginal, or both vesico and nal fistula is identified, one must also ureterovaginal fistulas. Figure 2 displays evaluate to ensure there is not a concom- several possible outcomes of the double itant ureteral injury. dye tampon test. A dry, colorless tampon The standard office test for diagnosis of a suggests that there is not a fistula and other suspected fistula is the double dye tampon sources of incontinence should be consid- test. This allows the surgeon to identify if the ered. If only the distal edge of the tampon is patient has a vesicovaginal fistula, ureter- blue and the top is dry, stress incontinence ovaginal fistula, or both in the office setting. or transurethral urine leakage should be The patient is given 200 mg oral phenazo- considered (as in first image). A damp, pyridine at least 30 minutes before test to colorless tampon suggests a peritoneal fis- dye the urine orange. The bladder is then tula or even cuff dehiscence if a hysterec- retrograde filled with ∼250 mL of saline tomy was performed during the original dyed blue with methylene blue or indigo surgery. A damp, blue tampon (last image) carmine. For large vesicovaginal fistulas, suggests a vesicovaginal fistula. If the tam- blue dye can often be seen pooling in the pon is orange, a diagnosis of ureterovaginal vagina immediately (Fig. 1). After removal fistula is likely, while a damp tampon with of the catheter, a tampon is placed into the both blue and orange dyes suggests that vagina, and the patient is allowed to ambu- patient may have both a vesicovaginal and late for 30 minutes without voiding. It is ureterovaginal fistula. If either a vesicova- important that the patient not void during ginal or ureterovaginal fistula is suspected thetestasurinecanwickonthetamponand referral to urogyneoclogy or urology is obscure the results. The tampon is removed appropriate. A myriad of other tests and imaging may be utilized to further characterize and/or confirm lower urinary tract fistulas. Cysto- scopy is often performed to evaluate the size and location of the fistulous tract, its rela- tionship to the ureteral orifices, and the health of the surrounding urothelium, all of which guide the surgeon in when and how to repair the fistula. Leukocytosis may be seen on complete blood count. If there is fluid pooling in the posterior vagina, it can be sent for creatinine and compared with serum creatinine levels. Markedly higher levels of creatinine in the vaginal fluid than serum suggest urine can confirm urinary leakage; however, it does not differentiate bladder and ureteral fistulas. A study by Thayalan et al remarked that these additional tests often incurred high expense while not yielding useful additional infor- mation after a physical exam and double dye test have established the presence of a vesicovaginal fistula.7 Additional imaging should be ordered with discretion by the FIGURE 1. Dye test with methylene blue. diagnosing physician to evaluate other

FIGURE 2. Tampon dye test. pelvic abnormalitiesandtheureters. surgery is optimal for patient outcomes When additional imaging is ordered, we and decreases litigation. Intraoperative recommend computed tomography (CT) cystoscopy has a 97% detection rate for urogram rather than CT with intravenous lower urinary tract injury at the time of contrast or ultrasound. To accurately eval- hysterectomy and should be done rou- uate to the distal ureter, the delayed images tinely after hysterectomy.8 Cystoscopy is obtained with urography are essential. A also associated with better litigation out- CT with intravenous contrast or ultrasound comes. Unfortunately, however, not all can miss a distal ureteral injury. This will lower urinary tract injuries can or will be avoid delay of diagnosis and referral to a identified at the time of surgery and may urogynecologic surgeon or urologist. go on to fistulize. When disease of the upper urinary tracts is suspected, CT urogram or magnetic TREATMENT OF VESICOVAGINAL resonance imaging is used to further char- FISTULA acterize the fistula. Retrograde pyelography Most posthysterectomy vesicovaginal fistu- and voiding cystourethrogam may demon- las are repaired primarily, but multiple strate ureteral abnormalities. Findings on repairs are sometimes necessary. Small ves- imaging that may suggest ureteric fistula icovaginal fistulas may resolve with place- include extravasation of dye, hydronephro- ment of an indwelling bladder catheter for 4 sis, or a persistent column of contrast in the to 6 weeks. Most experts recommend im- ureter. CT findings may include contrast mediate early repair if the fistula is identified within the vagina, detection of air/fluid in in the first 7 to 10 days after the initial the vagina, or the fistulous tract itself. surgery; however, if the vesicovaginal fistula Imaging may also identify radiation or is diagnosed after this time frame, repair anatomic changes, pelvic masses, or adher- should be postponed for ∼6weeksanduntil ent thickened bowel, any of which will affect the surrounding vaginal epithelium and surgical planning. urothelium appear healthy. Vesicovaginal fistulas can be repaired MANAGEMENT OF GENITOURINARY using transvaginal or minimally invasive FISTULAS abdominal routes of access. Regardless, Identification and management of a gen- several important principles apply. It is itourinary injury at the time of the initial essential to have adequate visualization of www.clinicalobgyn.com Copyright r 2021 Wolters Kluwer Health, Inc. All rights reserved. Obstetric and Gynecologic Fistulas 325 the fistula and excellent mobilization of surgery; and 1 had a 1-year delay in the fistula from surrounding tissues. A diagnosis. Therefore, in the majority of two-layer closure is typically performed women who sustain ureterovaginal fistu- ensuring the repair is not left under any las after hysterectomy, ureteral stenting is tension. If the surrounding tissues do not an effective first line therapy. have adequate vascular supply, surgeons When endourologic management of often consider placement of vascular in- ureterovaginal fistula is not successful or terposition flaps (omentum if the repair is appropriate, surgical intervention is nec- abdominal and martius graft if it is essary. This may be accomplished by vaginal). Prolonged bladder drainage for reimplanting the ureter into the bladder 2 to 3 weeks is recommended. or reanastomosis of the injured ureter. Given that damage is most commonly in TREATMENT OF URETEROVAGINAL the lower 1/3 of the ureter after gyneco- FISTULA logic surgery, ureteral reimplantation Treatment of ureterovaginal fistula revolves through ureteroneocystotomy is the most around resolution of urinary leakage, pre- effective surgical correction. In this cir- vention of urosepsis, and preservation of cumstance, the ureter is identified at the renal function.9 Partial ureteral obstruction pelvic brim and mobilized to the fistula is commonly present, and therefore drainage site. The distal end of the ureter is trans- of the affected upper urinary tract is essential. ected, and any scarring or devitalized If a surgical subspecialist is not available, tissue excised. The ureter is spatulated gynecologists can consider referral to inter- and reimplanted into the posterior blad- ventional radiology for temporary manage- der near the dome under no tension. ment. Interventional radiologist can place a Adjuvant procedures, such as a psoas percutaneous nephrostomy tube and/or ante- hitch or Boari flap, may be utilized to rograde ureteral stent.10 facilitate ureteroneocystotomy repair Expert opinion regarding optimal without tension.9 In the rare instance management of ureterovaginal fistulas where direct reimplantation is not feasi- varies with some recommending surgical ble, end-to-end or end-to-side anastomo- repair or ureteral reimplanation11–13 and ses of ureteral segments may be others advocating for endourologic man- considered to repair the defect. agement as first line treatment.9,10 Most commonly, the patient will initially be taken to the operating room for retro- Obstetric Fistulas grade pyelography and ureteral stent Obstetric fistulas occur rarely in devel- placement. We recommend evaluating oped countries, but are prevalent in re- both ureters and kidneys. A recent case source poor areas of the world. In much series reported outcomes of 19 cases of of sub-Saharan Africa and south Asia, ureterovaginal fistula after obstetric or women deliver infants in facilities that gynecologic surgery (18 after hysterec- lack electricity, basic surgical instruments tomy and 1 after cesarean delivery) at a and materials, and personnel to provide single academic center.9 Twelve of the timely and high-quality obstetric inter- ureterovaginal fistulas were managed ventions such as a cesarean delivery in with initial ureteral stenting, which was the case of obstructed labor. The result is successful in 92% of the patients. Primary often death of the neonate and at times, ureteral reimplantation was selected in 6 the mother. Those who survive the diffi- patients for the following reasons: 3 had a cult childbirth may develop a vesicovagi- concomitant vesicovaginal fistula, 2 were nal fistula because of the fetal head identified intraoperatively at the index applying constant pressure on the pelvic

www.clinicalobgyn.com Copyright r 2021 Wolters Kluwer Health, Inc. All rights reserved. 326 Abrams and Pope tissues, interrupting the normal blood cutting, have been attributed to increasing supply. The tissues under pressure will the risk for developing a fistula. However, eventually necrose and slough off, leaving if these individuals receive access to intra- the fistula usually between the bladder or partum obstetric services, they would bladder neck and the vagina. In the worst likely not develop fistulas. While child cases, this connection continues into the marriage and genital cutting should come rectum, and resulting in urinary and rectal to an end to protect the rights of girls and fistulas. women, obstetric fistula will only come to These patients present with uncontrol- an end if poor women are prioritized in lable urinary leakage and often do under- improving access to health care and if stand its mechanism. In an effort to obstetric interventions are improved reduce the urinary leakage, women often worldwide. avoid drinking water, which in turn causes the urine to have a more concen- MEDICAL SEQUELAE trated putrid odor. For these women, Obstetric fistula can be seen as a larger even the most understanding partners injury “complex,” incorporating vesico- and family members find it difficult to vaginal fistula, rectovaginal fistula, stress share close quarters with them. As coping incontinence, renal involvement, secon- with the incontinence is often not possible dary infertility, vaginal stenosis, and foot- while working, many women experience a drop, and other physical ailments that lifetime of disability and social isolation. often accompany obstetric fistulas.15 Sim- Obstetric fistula has been described as ilarly, psychosocial effects and economic “the worst problem you have never heard detriment have also been documented, all of,” (http://www.operationfistula.org). Co- contributing to the injury complex. Ap- lloquially shortened to “fistula,” it is a proximately 90% of all births during problem often overlooked that leads to which obstetric fistulas occurs are accom- deep social embarrassment. The women panied by stillbirth.16 Wilson et al17 found affected either limit their interactions in that women with fistulas are more likely their communities or spend most of their to be depressed, experience post-trau- lives hiding their incontinence. In some matic stress disorder, have somatic com- contexts where fistula exists, communities plaints, and lack a social network. blame the woman for her child dying, and Community qualitative studies have dem- then she is blamed for the subsequent onstrated a deep layer of secrecy sur- uncontrollable leakage of urine. rounding obstetric fistulas and surgical Obstetric fistulas in low-resource set- repair. Barriers to accessing repair con- tings are largely caused by obstructed tinue to be unveiled in multiple countries. labor and could be prevented by high Baker et al18 found that financial barriers quality and skilled monitoring of labor were the most frequently mentioned and timely operative delivery. The women (65%). affected by fistula are generally of low socioeconomic status, with little to no education and often experience social Treatment of Obstetric Fistulas isolation and stigma as a result of the fistula. Browning et al14 examined phys- CATHETER PREVENTION ical attributes as risks for fistula and In 1841, Ryan in London described spon- found those affected are significantly taneously closing fistulas by keeping pa- shorter and have a smaller pelvic inter- tients on their face or side, tying a catheter tuberous space. Cultural patterns, such into the bladder and tamponing the vag- as young age of marriage and genital ina with oiled lint. We still recommended www.clinicalobgyn.com Copyright r 2021 Wolters Kluwer Health, Inc. All rights reserved. Obstetric and Gynecologic Fistulas 327 prolonged catheter drainage to facilitate junior surgeons, and visiting surgeons spontaneous vesicovaginal fistula healing, take into consideration the additional especially when the fistula is recognized trauma a failed repair presents to a soon after it develops. Prolonged catheter woman who has been suffering from drainage can be utilized for prevention in incontinence and also consider the de- individuals at high risk of developing a creased chance for healing with subse- fistula. Waaldijk reports healing rates of quent attempts before choosing to take on 15% with prolonged, early catheterization the surgery and not refer to a surgeon as conservative management of obstetric experienced in closing complex obstetric fistuas.19 fistulas.

REPAIRING VESICOVAGINAL CLASSIFICATION FISTULAS Frajzyngier et al23 examined prognostic Traditionally, surgeons have waited three values of classification systems and found months from delivery to operate on wom- them to be “poor to fair.” Bengtson et al24 en with vesicovaginal fistulas so that any looked for prediction of incontinence spontaneous closure may occur, and after surgery using Goh’s classification. granulation tissue is no longer present. Using a scoring algorithm based on clin- Some surgeons, however, advocate for ical and demographic characteristics, they operating on “fresh fistulas” rather than found age above 50 years, length of time waiting. In cases of newly identified fistu- with a fistula above 20 years, previous las, Waaldjik19 published on immediate surgical attempts, advanced Goh classifi- surgery after catheterization for 3 months. cation relative to the urethra, moderate to The catheter is kept until the fistula edge severe scarring, circumferential fistula, is no longer necrotic at which time the and urethral length of 1.5 cm or less were fistula is repaired. Outcomes using this all highly associated with residual incon- timing for repair were highly successful at tinence. The authors suggest using the 91.8% (n = 156). Short interval from fis- scoring tool before surgery to guide diag- tula development to repair could greatly nostic purposes and to assist with refer- decrease stigma and the social consequen- ring complex patients to expert-level ces of living with an obstetric fistula. surgeons.

PREDICTORS FOR SUCCESS and REPAIR TECHNIQUE FAILURE Vaginal approach to vesicovaginal fistula Reported rates of successful surgical clo- closer is generally described in the liter- sure of obstetric fistulas are high (84% to ature and preferred by most surgeons.25 94%), but increasingly complex fistulas The type of suture and number of layers are less likely to heal.20,21 Repair out- of closure have not been studied in a comes are associated with duration of the systematic way, but retrospective data fistula before surgery, fistula size, circum- demonstrates no superiority of 2 layers ferential fistulas or those with urethral when controlled for bladder size.26 Cir- involvement, and moderate to severe vag- cumferential fistulas appear to occur after inal scarring.21 Women with large fistulas longer and potentially more severely ob- ( > 3 cm) are 6 times more likely to fail structed labor (see Fig. 3). More bladder repair (P < 0.01).22 Previously failed at- tissue is lost and vaginal scarring is often tempts suggest that patients may not be also more severe with circumferential properly triaged or referred to surgeons fistulas resulting in residual incontinence with expertise in repairing complex cases. more frequently.27 Repeat procedures are It is imperative that training programs, common after failed closure, and some

FIGURE 3. Circumferential fistula. authors argue that an interpositional increase in diameter of fistula].30 Kopp layer with a new vascular supply in the et al31 examined 346 women after repair to form of a gracilis flap should be used as determine a significant pad weight for the Martius graft has not demonstrated incontinence. They determined that a improvements in patient outcomes.28,29 1-hour pad weight after catheter removal The first attempt is still widely considered of > 1.5 g had a positive predictive value of to be the best chance for success and 94% (confidence interval: 90.0-96.9) in pre- therefore, triaging cases according to dicting ongoing continence. Sexual dysfunc- surgical skill would optimize outcomes. tion is a challenge for a significant proportion of patients after surgery. Of RISKS FOR ONGOING INCONTINENCE 102 patients interviewed, 23 (22.5%) re- and SOLUTIONS ported not being able to engage in penetra- Defining “cure” after obstetric fistula repair tive vaginal intercourse and 12 (52%) of is not straight forward as many of these these patients ascribed this to a “narrow” women have severe transurethral urine leak- vagina.32 Reconstructive techniques to im- ageafterthefistulaisrepaired.Whileadye prove the quality of vaginal reconstruction tampon test can help differentiate between a and sexual function are imperative to im- healed fistula and urethral leakage, not all prove quality of life.33 surgeons perform dye tests at the time of catheter removal. Browning reported that persistent urinary leakage after obstetric Conclusion fistula repair was associated with the follow- Genitourinary fistulas in developed coun- ing: fistula involving the urethra [odds ratio tries are uncommon and most often a (OR): 8.4 (3.9-17.9)], small bladder (< 5cm sequalae of pelvic surgery and hysterec- depth from external ureteral orifice) [OR: tomy. Early identification and manage- 4.1 (1.2-13.8)], vaginal scarring [OR: 2.4 ment are essential to optimize outcomes (1.5-4.0)], and larger fistula size [increase for patients and minimize litigation. OR: 1.3 (1.16-1.56) with each centimeter In contrast, genitourinary fistulas in www.clinicalobgyn.com Copyright r 2021 Wolters Kluwer Health, Inc. All rights reserved. Obstetric and Gynecologic Fistulas 329 developing countries are often related to 14. Browning A, Lewis A, Whiteside S. Predicting obstetrics and obstructed labor. women at risk for developing obstetric fistula: a fistula index? An observational study comparison of two cohorts. BJOG. 2014;121:604–609. 15. Arrowsmith S, Hamlin EC, Wall LL. Obstructed labor injury complex: obstetric fistula formation References and the multifaceted morbidity of maternal birth 1. Medlen H, Barbier H Vesicovaginal fistula. Stat- trauma in the developing world. Obstet Gynecol – Pearls. Treasure Island, FL: StatPearls Publish- Surv. 1996;51:568 574. ing; 2020. 16. Ahmed S, Anastasi E, Laski L. Double burden of 2. Shaw J, Tunitsky-Bitton E, Barber MD, et al. tragedy: stillbirth and obstetric fistula. Lancet – Ureterovaginal fistula: a case series. Int Urogyne- Glob Health. 2016;4:e80 e82. col J. 2014;25:615–621. 17. Wilson SM, Sikkema Kh, Watt Mh, et al. Psy- 3. Cowgill KD, Bishop J, Norgaard Ak, et al. chological symptoms and social functioning fol- Obstetric fistula in low-resource countries: an lowing repair of obstetric fistula in a low-income under-valued and under-studied problem— setting. 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and recurrent obstetric fistulas. Int J Gynaecol 31. Kopp DM, Bengtson AM, Tang JH, et al. Use of a Obstet. 2018;143:390–392. postoperative pad test to identify continence status in 29. Tebeu PM, et al. Comparative study of the out- women after obstetric vesicovaginal fistula repair: a come of surgical management of vesico-vaginal prospective cohort study. BJOG. 2017;124:966–972. fistulas with and without interposition of the 32. Pope R, Ganesh P, Chalamanda C, et al. Sexual Martius graft: a Cameroonian experience. Prog function before and after vesicovaginal fistula Urol. 2015;25:1225–1231. repair. J Sex Med. 2018;15:1125–1132. 30. Browning A. Prevention of residual urinary in- 33. Pope R, Brown RH, Chipungu E, et al. The use of continence following successful repair of obstetric Singapore flaps for vaginal reconstruction in vesico-vaginal fistula using a fibro-muscular sling. women with vaginal stenosis with obstetric fistula: BJOG. 2004;111:357–361. a surgical technique. BJOG. 2017;125:751–756.