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The earliest evidence of gynecologic fistula dates back millennia, to Queen Henhenit, one of the wives of King Mentuhotep II of Egypt (11th Dynasty, c. 2050 bc). In his dissection of the mummy at the Cairo School of Medicine in 1923, Derry noted a large vesicovaginal fistula (VVF) in the presence of a severely contracted pelvis; he concluded that the fistula was a consequence of obstructed labor ( ). In 1663, van Roonhuyse of Amsterdam published Medico-Chirurgical Observations About the Infirmities of Women, commonly thought of as the first textbook on operative gynecology. In it, van Roonhuyse proposed a revolutionary surgical technique for the closure of VVFs based on the following principles: lithotomy position, good exposure of the fistula with a vaginal speculum, marginal denudation of the fistula edge, use of a fine scissors or knife, and approximation of the denuded edges with “stitching needles of stiff swans’ quills” ( ).
Centuries later, J. Marion Sims, advanced the surgical technique to repair vesicovaginal fistula. Sims practiced medicine at a time when treating women was considered distasteful and rarely done. With a modern lens his legacy is both complicated and divisive despite his contributions to the medical field. Sims published his classic paper “On the Treatment of Vesicovaginal Fistula” in 1852 in the American Journal of Medical Sciences , in which he deprecated the use of cautery and advocated for postoperative bladder drainage with a urethral catheter. His technique denuded the fistula edge, closing the defect in one layer with fine silver wire applied with leaden bars and perforated shot. From his work he developed novel surgical instruments including a vaginal speculum with a bent pewter spoon as a vaginal and utilized reflected light from a mirror. ( ). He later designed a silver sigmoid-shaped, self-retaining retractor for this purpose.
Sims is however a controversial figure as his research was conducted on enslaved Black women without anesthesia. This has led medical ethicists and historians to consider his use of enslaved Black bodies as medical test subjects falls into a long, ethically bereft history that includes the Tuskegee syphilis experiment and Henrietta Lacks. The American Medical Association recommends that if unethically acquired data are essential to science, any use or citation of these data should describe the unethical behavior and pay respect to the victims of the experimentation. A candid telling of the history of medicine can apply a new focus on the impact of the women and subjects in the narrative of Sims. Namely, the enslaved women, Lucy, Anarcha, and Betsy, who through their work, fortitude, and even suffering were equally responsible for the contemporaneous advances in surgical care. In this manner acknowledgment builds new ways to understand the history of medical research and to recognize the damage certain practices may continue to have as they reverberate over generations. ( )
The discovery of antibiotics and the development of general and regional anesthesia contributed significantly to the surgical treatment of VVFs in the twentieth century. Other notable milestones included urethral reconstruction using lateral vaginal flaps and labium minus grafts ( ); suprapubic intraperitoneal repair of posthysterectomy, high vesicovaginal and rectovaginal fistulas ( ); partial colpocleisis for posthysterectomy VVFs ( ); urethral reinforcement using pelvic floor muscles ( ); pedicled gracilis muscle flap ( ); bulbocavernosus flaps ( ); abdominal repair technique of VVFs involving bisection of the bladder ( ); pubococcygeus, bulbocavernosus, rectus abdominis, and gracilis flaps ( ); the use of pedicled omental flaps in the repair of extensive VVFs ( ); and urethral reconstruction ( ; ). Knowledge of effective repair of genitourinary fistulas became more widely disseminated with the publication of The Vesico-Vaginal Fistula ( ). Greater international attention was brought to the immense problem of genitourinary fistulas in developing countries with the foundation of the Second Fistula Hospital in Addis Ababa, Ethiopia, in 1975, and the report of 1789 fistulas repaired during an 11-year period from Nigeria ( ).
Fistulas, including those in the lower urinary tract, occur secondary to an initial insult followed by a defect or vulnerability in the wound-healing process. Wounded tissue undergoes four phases of healing: coagulation, inflammation, fibroplasia, and remodeling. These phases do not occur independently, but overlap one another. During the fibroplastic phase, collagen is laid down, reaching its peak on the seventh day after injury and continuing for 3 weeks. Between the first and third weeks, healing is most vulnerable to hypoxia, ischemia, malnutrition, radiation, and chemotherapy, so this is the time when most fistulas present. Conditions known to interfere with wound healing are associated with an increased risk of fistula formation, including diabetes mellitus, smoking, infection, peripheral vascular disease, chronic steroid use, malignancy, and previous tissue injury.
Most of the literature on the etiology, diagnosis, management, and prevention of lower urinary tract fistulas consist of expert opinions, case series, or cohort studies, with few randomized controlled trials. Many of the studies are based on experiences with obstetric fistulas, which primarily occur in women in developing countries. Research in these environments is often limited by resource scarcity and plagued with short-term follow-up periods. It is important to review the information presented in this chapter with these limitations in mind.
The vast majority of urogenital fistulas that occur in women in developing countries are caused by obstetric trauma, accounting for an estimated 95% of genitourinary fistulas ( ). Obstetric fistula occurs when women experience prolonged, obstructed labor, typically associated with young childbearing age and lack of access to emergency obstetric care allowing cesarean section ( ). Prolonged pressure of the fetal head within the maternal pelvis results in ischemia and necrosis of tissue trapped between the fetal head and the maternal bony pelvis, with consequent tissue breakdown and eventual fistula formation. The consequences of obstetrical fistula can be severe and life-altering. The obstructed labor injury complex described by includes urinary incontinence that is typified by constant urinary leakage from the vagina. Additional devastating symptoms of the resultant fistula include gynecologic injury such as vaginal stenosis, chronic pelvic infection, secondary infertility, cutaneous manifestations such as vulvar urinary dermatitis, and neurologic injury such as foot drop or complex neuropathic bladder dysfunction ( ). Tragically, the cruelest outcome is high rates of fetal loss, with over 90% of pregnancies in women who develop fistulas resulting in stillbirth. Because of the intractable urine loss, associated odor, and lack of feminine hygiene resources, women who develop fistulas often suffer ostracism from their communities and families ( ). There is a subsequent loss of psychological and social support resulting in limited means to access appropriate care. In some series, over 50% of affected women were divorced by or separated from their husbands, and over 70% experienced depression attributed to their symptoms. See for an example of an obstetrical fistula.
Obstetric fistula is estimated to affect 2 to 3.5 million women in the developing world, including sub-Saharan Africa and South Asia. Prevalence estimates of obstetric fistula from demographic health surveys and other population-based surveys range widely from 0.16% to 4.7% in sub-Saharan Africa and 0.08% to 2.7% in South Asia. The World Health Organization estimates that between 50,000 and 100,000 women develop obstetric fistula annually ( ). However, the true disease burden is unknown and believed to be significantly underreported. Most estimates are based on models that combine extrapolations from facility-based studies, which include only individuals with access to health care, and from cross-sectional community-based surveys, which lack standardization and validation for fistula diagnosis and suffer from both potential overestimation attributed to other causes of incontinence and underreporting because of embarrassment associated with incontinence.
Commonly described physical and social risk factors for development of obstetric fistula in developing countries include prolonged labor, early marriage and pregnancy (mean age 15.5 years), short stature (<150 cm), and limited access to emergency obstetric care ( ). In a retrospective review of 93 patients treated for obstetric fistula in Central Uganda, the average time spent in labor was 2.56 days, with an average of 1.3 days’ delay in seeking medical care ( ). concluded in a case control study that postprimary education was associated with a protective effect against development of fistula. Women at risk for and with obstetric fistula commonly live in rural areas, deliver at home without access to established medical care, and may go unnoticed by public health officials or health care providers. Many women are unaware that the condition is treatable, and thus lack appropriate care because of social isolation as a result of their urine leakage symptoms. Poverty, lack of knowledge, long travel distances, and waiting lists deter women from traveling to major centers and receiving care. As a result, many of these women live with fistula symptoms for years and face abandonment by their families and isolation from society ( ).
The commonly accepted archetype of the fistula patient is a poor and malnourished young woman with limited formal education, from a rural area, who attempts a home delivery without a trained birth attendant, which results in prolonged, obstructed labor and delivery of a stillborn infant. In various hospital-based case series, the mean age of women with obstetric fistula ranges from 19 to 35 years, and in approximately half (31%–81%) of women, the fistula developed during their first delivery. Female circumcision has not been shown to be associated with an increased risk for fistula development; however, Gishiri cutting (i.e., longitudinal cuts in the vagina, made for various obstetric and gynecologic disorders) and the use of traditional postpartum vaginal caustic agents are associated with fistula development. Fistula resulting from obstructed labor is often associated with cephalopelvic disproportion. Supporting this concept are several case series that have documented the prevalence of short stature in women with fistulas, with average heights of 149 to 156 cm, and smaller foot sizes. The concomitant anthropomorphic factors of reduced pelvic inlet with inadequate obstetrical conjugate distance as a result of early childbearing, chronic disease, malnutrition, and even rickets can increase dystocia risk. VVFs can also follow cesarean delivery or peripartum hysterectomy, particularly after a prolonged and difficult labor. Endometritis and postpartum hemorrhage also increase the incidence. Furthermore, labor management with an operative vaginal delivery utilizing forceps or vacuum can increase risk of fistula formation.
That obstetric fistula is still a public health problem in some countries demonstrates the enormous gap in maternal health care between high-income and low-income countries. Women with obstetric fistula are indicators of health system shortcomings in terms of the ability to deliver accessible, timely, and appropriate intrapartum care. Obstetric fistula consistently and disproportionately affects the poorest women, whose voices are hardly heard.
VVFs represent the majority of lower urinary tract gynecologic fistulas. In developed countries, obstetric causes are rare. The majority of genitourinary fistulas in high-income countries are associated with iatrogenic factors, specifically surgery and pelvic radiation. The most common surgeries associated with fistula development are benign gynecologic procedures, specifically hysterectomy. A recent meta-analysis describing 35 years of reported cases in the literature concluded that, among 2055 reported urogenital fistulas in developed countries, 46% were attributed to abdominal hysterectomy (with a total of 62% associated with hysterectomy by any route). Overall, over 96% of urogenital fistulas were attributed to surgery (83.2%) or radiation therapy (13%) ( ). Minimally invasive approaches have gained in popularity in recent years, and a meta-analysis of outcomes from laparoscopic versus abdominal hysterectomy demonstrated a higher risk of intraoperative and postoperative urologic complications associated with a laparoscopic approach. However, this difference was not seen in the subgroup of studies published after 2012, suggesting that increasing experience with minimally invasive approaches may lead to a comparable complication rate ( ).
Genitourinary fistulas that result from gynecologic surgery occur secondary to either direct urinary tract injuries or devascularization, often with the use of diathermy (electrocautery). Bladder injuries resulting from hysterectomy occur primarily during blunt dissection of the bladder off the lower uterine segment. Devascularization or an unrecognized tear in the posterior bladder wall subsequently results in tissue ischemia, necrosis, and fistula formation. Operative risk factors for lower urinary tract fistulas include intraoperative cystotomies that extend into the trigone or bladder neck, hysterectomy for a large uterus, pelvic adhesions from prior surgery, and intraoperative blood loss greater than 1000 mL ( ). Patient risk factors can include previous pelvic operations including cesarean section, history of pelvic inflammatory disease, prior pelvic irradiation, endometriosis, body mass index greater than 30, diabetes mellitus, tobacco use, and concurrent infection ( ).
In a study in Finland, reviewed the incidence of urinary tract injury on a national scale. During the study period, 62,379 hysterectomies were performed, and 142 urinary tract injuries were reported. The incidence of bladder injury was 1.3 per 1000 hysterectomies. The incidence of VVF was 1 in 1200 procedures: 1 in 455 after laparoscopic hysterectomy, 1 in 958 after total abdominal hysterectomy, and 1 in 5636 after vaginal hysterectomy. Notably, the risk of ureteral injury was greater with laparoscopic procedures than with open or vaginal procedures.
Bladder and urethral injury are also known complications of antiincontinence procedures and repair of pelvic organ prolapse. Broad adoption of synthetic mesh slings for the treatment of urinary incontinence can carry risk of mesh-related complications. Whether because of intraoperative injury or postoperative erosion of synthetic material into the bladder or urethra, vesicovaginal and urethrovaginal fistula (UVF) formation after antiincontinence sling procedures have been reported to occur in up to 0.5% to 1% of cases ( ). There are also case reports of UVF development after urethral diverticulectomy and periurethral injection of a bulking agent.
Radiation therapy, used for carcinoma of the cervix or other pelvic malignancies, may also result in fistula formation. Healthy tissues of the anterior vaginal wall tolerate radiation doses as high as 8000 cGy. Fistulas may first appear during the course of radiotherapy, usually from necrosis of the tumor itself, or after treatment is completed. Late fistulas arise secondary to endarteritis obliterans, usually within the first 2 years, but they can occur anytime from 6 months to 5 years after treatment completion.
A subset of genitourinary fistulas occur secondary to advanced malignancy, with estimates ranging between 3% and 5%. The most common types of associated malignancies include cervical, vaginal, and endometrial carcinoma ( ).
Ureterovaginal fistulas occur uncommonly, but generally have a similar etiological profile and risk factors as VVFs. Although the majority are attributed to gynecologic surgery, other reported causes include obstetric surgery, vaginal delivery, infertility treatment with oocyte retrieval, pelvic radiation, and retained pessary ( ).
Patients with genitourinary fistulas present in many ways, but virtually all share the symptom of some form of persistent insensate urinary leakage from the vagina. The timing of symptom onset and the severity of symptoms coincide with the size, location, and nature of the injury. After surgery, radiation, or other inciting insult or injury to the tissue, symptoms may develop after an interval of days, weeks (surgical and obstetric fistulas), months, or even years (radiotherapy-related fistulas). Postsurgical fistulas, the most common type in developed countries, usually present in the first 14 days after surgery. The classic presentation is a patient who describes continuous urinary leakage (day and night) or persistent watery vaginal discharge. If the fistula is very small, leakage may be intermittent, occurring only at maximal bladder capacity or with particular body positions, such as lying supine. Other associated signs and symptoms include unexplained fever, hematuria, recurrent cystitis or pyelonephritis, vaginal pain, suprapubic pain, flank pain, and abnormal urinary stream.
The size, location, and number of fistulous connections should be ascertained to facilitate the best curative measures. Office evaluation, cystourethroscopy, and imaging studies such as intravenous urography permit the localization and characterization of suspected fistula, and additional exclusion or identification other types of urinary tract injury. The initial evaluation of all patients with symptoms of genitourinary fistulas starts with a complete physical examination. In a patient with vaginal leakage, a thorough speculum examination of the vagina may reveal the source. Oftentimes, the first examination finding consistent with fistula is urine pooling in the vaginal vault. Depending on the patient’s anatomy, the fistula may best be seen using a full speculum to retract both the anterior and posterior vaginal wall. This is particularly true when the fistula is located at the vaginal cuff, which is most common for VVFs occurring as a result of hysterectomy. When the fistula is more distal, it is useful to use the bottom blade of a Grave’s speculum to allow careful visualization of the anterior vaginal wall. During examination, the surrounding tissue should be assessed for signs of inflammation, edema, and necrosis, which portend poor wound healing and should be addressed before repair.
If a urethral fistula is suspected but not immediately seen, it may be necessary to place a urethral catheter to elongate the urethra and allow for inspection of the entire vaginal epithelium overlying the urethra. If any portion of the catheter becomes visible on vaginal examination, a sizable fistula is present.
If leakage is seen, and a fistula suspected without certainty based on exam, the fluid may be analyzed for urea or creatinine concentrate to confirm the presence of urine. If leakage is not immediately demonstrated, the bladder may be filled (with saline or methylene blue saline), and provocative maneuvers, such as Valsalva or manual pressure over the bladder, may be used to reproduce and confirm the patient’s symptoms.
Further office-based evaluation to confirm the presence of a small-diameter VVF and/or evaluate for ureteral involvement includes the tampon test. Instillation of methylene blue or indigo carmine into the bladder typically stains a vaginal swab or tampon blue in the presence of a VVF. If instillation volume is inadequate because of urethral leakage, a transurethral Foley catheter should be placed to prevent any staining of the distal tampon from leakage of fluid through the urethral meatus. Unstained but wet swabs may indicate a ureterovaginal fistula. If a ureterovaginal fistula is suspected, oral phenazopyridine can be administered at the time of methylene blue bladder instillation, which will cause orange-colored staining of the tampon. Other options include intravenous indigo carmine, which would stain the tampon blue, and sodium fluorescein. Sodium fluorescein (25 mg of 10% intravenous sodium fluorescein [0.25 mL]) is quickly renally excreted and stains the urine a greenish hue ( ). However, both of these dyes require intravenous access, and may be most appropriate if performing an examination without anesthesia. Intravenous methylene blue must be used with caution because of the risk of methemoglobinemia, a rare but serious complication.
Cystourethroscopy or combined cystoscopy-vaginoscopy is recommended to further characterize the fistula size, number, complexity, and location within the bladder, along with associated lesions and condition of surrounding tissue. Key observations include the fistula’s size and proximity to the bladder neck, urethral sphincter, and ureteral orifices, as well as the presence of tissue edema, slough, infection, induration, stones, foreign material (sutures, mesh), and scarring. An additional benefit is the opportunity to perform retrograde pyelogram in order to assess for ureteral involvement. Cystoscopy can be difficult with large fistulas because of leakage of irrigation; in such cases, a Foley catheter may be placed within the fistula tract to decrease leakage and observe the rest of the bladder. Bladder biopsy should be performed as needed if there is a history or suspicion of malignancy or lesions suspicious for infectious agents, such as tuberculosis, contributing to the development of fistula.
There is no clear consensus on the optimal imaging modality that should be performed to diagnose and determine the extent of a urinary tract fistula. In fact, imaging is frequently not required to make a diagnosis and treat a vesicovaginal or UVF, but it may be helpful. In a multiinstitutional study of 226 VVFs diagnosed in the United States, 23% of fistulas were diagnosed and managed based on physical examination alone, while the other 77% were assessed with some form of imaging or cystoscopy ( ).
If one chooses to pursue imaging, the choice of study should be tailored to maximize the likelihood of detecting the fistula based on suspected location. Suspected bladder injuries can be evaluated using retrograde cystourethrography, but for suspected urethral injuries, voiding cystourethrogram is better. However, we prefer intravenous urography with cross-sectional imaging, such as computed tomography (CT) or magnetic resonance urogram, which evaluates not only the bladder but also the ureters for fistula or obstruction ( Fig 38.1 ). However, this technique may not be optimal for suspected concomitant ureteral and bladder fistulas, as they are sometimes difficult to distinguish and, under distension of the bladder, may lead to poor or no visualization of a small VVF. If the fistula is clearly seen on physical exam, imaging may be of less value. In the uncommon setting of vesicouterine fistula, hysterosalpingography may be useful. Other advanced imaging techniques include subtraction magnetic resonance fistulography and endocavitary ultrasound with or without Doppler and contrast ( ; ).
Various conservative or minimally invasive therapies are available for lower urinary tract fistulas, although the true viability and success of these treatment modalities are unknown, as the literature consists mostly of case reports and small retrospective series. The most conservative treatment of a VVF is simply prolonged bladder drainage with a Foley catheter. In a retrospective analysis of 1716 women with obstetric VVF, continuous catheter drainage resulted in spontaneous fistula closure in 15% of patients. Spontaneous closure occurred in 50% to 60% of patients with fistulas that were 2 cm or less and who presented for care no later than 4 to 6 weeks after delivery ( ; ). Because it may be logistically difficult to repair a VVF immediately, and/or there may be tissue factors such as necrosis preventing immediate repair, it is reasonable to proceed with a trial of continuous bladder drainage for 4 weeks in fistulas that are less than 1 cm in diameter and unrelated to malignancy or radiation, as these fistulas may spontaneously resolve in 12% to 80% of cases.
Other less invasive options include curetting, electrofulguration, and laser ablation to deepithelialize the fistula tract and allow it to spontaneously heal while the bladder is continuously drained. Once the fistula tract has been deepithelialized, injection of agents such as fibrin glue and collagen into the fistula tract has been shown to successfully promote closure. Most of these studies report successful treatment in small fistulas (≤5 mm) resulting from gynecologic surgery. There have also been reports of using these methods to successfully close radiation-induced fistulas. Agents such as cyanoacrylic glue administered percutaneously, endoscopically, or vaginally have been used in a series of 13 urinary tract fistulas of differing etiology, with an 85% success rate after a mean follow-up period of 35 months ( ). The failures were in larger fistulas (>1 cm). Nonsurgical fistula closure is best attempted in a patient who has a small fistula, who is a poor surgical candidate, or who desires to first try less invasive options.
Since the 1990s, the recommended initial management of an isolated ureterovaginal fistula has been ureteral stenting when feasible. In 1995, Selzman et al. reported 100% successful curative treatment of ureterovaginal fistula among seven patients in whom stent placement was feasible. More recently, described their 13-year experience with ureteral stent placement. Among 12 patients who underwent conservative management, 11 (92%) were able to be successfully stented (8 retrograde, 3 antegrade). At a median stent duration of 10 weeks, all patients had successful closure of ureterovaginal fistula. Stenting may be more successful when performed sooner rather than later. In a series by , stent failure occurred in two patients who underwent stent placement at 90 and 390 days after initial surgery. However, one patient who was stented 780 days after surgery experienced full resolution of symptoms.
Patients who undergo stent placement should be counseled about the risks of infection, stone formation, and ureteral stricture formation. After 8 to 12 weeks, a retrograde pyelogram can be performed to evaluate for persistence of the fistula. If the fistula has healed, the stent may be removed, and a renal ultrasound, CT urogram, or nuclear renogram may be performed after 4 to 6 weeks to rule out subsequent stricture formation and obstruction.
If stenting is not possible, or the patient fails treatment, interval management with percutaneous nephrostomy may be used until inflammation has subsided and the patient can safely undergo surgery.
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