HISTORIC PERSPECTIVES

The earliest evidence of a vesicovaginal fistula was reported by Derry (1935) in the mummified remains of Queen Henhenit, one of the wives of King Mentuhotep II of Egypt (11th Dynasty, circa 2050 b.c.). In his dissection of the mummy at the Cairo School of Medicine in 1923, Derry noted a large vesicovaginal fistula in the presence of a severely contracted pelvis; he concluded that the fistula was a consequence of obstructed labor. Hippocrates (460–377 b.c.) recognized the problem of urinary incontinence after confinement but offered no clue as to its cause. In his textbook, Al Kanoun, celebrated Persian physician Avicenna (980–1037) was the first to recognize that urinary incontinence after difficult labor was caused by communication between the bladder and vagina.

No further reference to vesicovaginal fistula appeared until 1597, when both Felix Platter of Basle and Luiz de Mercado of Valladolid separately reviewed the problem but offered no constructive therapeutic advice. Zacharin (1988) states that de Mercado first used the term fistula instead of the usual term ruptura.

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, this text was translated into English in 1676. The fourth chapter is titled “Rupture of the Bladder: The Signs, Causes, Prognostics and Cure Thereof.” Van Roonhuyse proposed a revolutionary surgical technique for the closure of vesicovaginal fistulas 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.” No record has been found that van Roonhuyse operated on patients using this technique. In 1752, a medical text by Swiss physician Fatio was posthumously published. In it were recorded two successful fistula repairs performed by Fatio in 1675 and 1684, using van Roonhuyse’s technique.

Volter in 1687 suggested that sutures should be interrupted, and he introduced the use of a retention urinary catheter. During this same period, Pietro DiMarchettis (1675) claimed complete cures, using cautery. In later years Monteggia, Dupuytren, and others also recommended cautery.

The nineteenth century was the dawn of a new era in the surgical treatment of vesicovaginal fistula. In 1834, Jobert de Lamballe (1852) successfully repaired a small number of fistulas using pedicled skin flaps (autoplastie vaginale par la methode indienne). A second technique (autoplastie par glissment ou par locomotion) later enabled him to close a greater number of fistulas. This technique involved dissecting the bladder from the cervix and vagina with the additional use of curved releasing incisions in the vagina to facilitate mobilization and closure without tension.

In a letter to the Boston Medical and Surgical Journal in August 1838, Mettauer (1840) of Virginia stated that he had repaired a vesicovaginal fistula about the size of a half dollar piece using lead wire, with good results. This was the first successful repair in the United States.

On June 21, 1849, in an eight-bed infirmary on Perry Street in Montgomery, Alabama, Sims (1852) operated on a young slave woman named Anarcha for the thirtieth time. Using the genupectoral position, a bent pewter spoon as a vaginal speculum, and reflected light from a mirror, Sims denuded the fistula edge, closing the defect in one layer with fine silver wire applied with leaden bars and perforated shot. On the eighth day, Sims reexamined the patient and noted that the wound was well healed. In 1852, he published his classic paper “On the Treatment of Vesicovaginal Fistula” in the American Journal of Medical Sciences. He deprecated both cautery as advocated by Dupuytren for small fistulas and obturation of the vulva as practiced by Vidal de Cassis (whereby the bladder and vagina are converted into a common reservoir for urinary and menstrual discharge). Sims insisted on liberal use of opium for perioperative analgesia and stressed the importance of postoperative bladder drainage with a urethral catheter. He later designed a silver sigmoid-shaped, self-retaining catheter for this purpose. In 1853 Sims moved to New York, and in 1855 he became chief surgeon in the newly built Woman’s Hospital, where he was later joined by a brilliant young assistant, Thomas Addis Emmet. Sims and Emmet worked closely together, with Emmet perfecting many of his mentor’s techniques.

In his text Vesico-Vaginal Fistula from Parturition and Other Causes with Cases of Recto-Vaginal Fistula (1868), dedicated to Sims, Emmet reported on 270 consecutive patients treated in the Woman’s Hospital: 200 were cured, 65 were improved, and 5 were considered incurable. Emmet eventually succeeded Sims at the Woman’s Hospital. Probably his greatest contribution to obstetric care was his insistence that frequent catheterization of the bladder in labor, together with the judicious use of forceps for second-stage delay, would prevent the majority of labor-related vesicovaginal fistulas.

In 1861, Collis of Dublin advocated the flap-splitting technique, whereby the anterior vaginal wall is widely dissected from the bladder with separate closure of the two defects. This method was later popularized by Mackenrodt (1894) in Berlin.

In the 1880s and 1890s, Trendelenburg and von Dittel reported failed attempts at fistula repair, using extraperitoneal and suprapubic approaches, respectively. Schuchardt (1893) also devised a parasacral incision, which permitted better access to high fistulas, particularly when associated with vaginal stenosis.

The discovery of antibiotics and the development of general and regional anesthesia contributed significantly to the surgical treatment of vesicovaginal fistulas in the twentieth century. Other notable milestones included urethral reconstruction using lateral vaginal flaps and labium minus grafts (Noble, 1901), suprapubic intraperitoneal repair of posthysterectomy, high vesicovaginal and rectovaginal fistulas (Kelly, 1902), partial colpocleisis for posthysterectomy vesicovaginal fistulas (Latzko, 1942), urethral reinforcement using pelvic floor muscles (Martius, 1928), pedicled gracilis muscle flap (Garlock, 1928), bulbocavernosus flaps (Martius, 1942), pubococcygeus, bulbocavernosus, rectus abdominis, and gracilis flaps (Ingelman-Sundberg, 1960), the use of pedicled omental flaps in the repair of extensive vesicovaginal fistulas (Kiricuta and Goldstein, 1972), and urethral reconstruction (Symmonds and Hill, 1978; Tanagho and Smith, 1972). Knowledge of effective repair of genitourinary fistulas became more widely disseminated with the publication of The Vesico-Vaginal Fistula (Moir, 1961). 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 over an 11-year period from Nigeria (Ward, 1980).

EPIDEMIOLOGY AND ETIOLOGY

To understand the cause of fistulas one must understand wound healing because it is a defect or vulnerability in this process that results in fistula development. 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 increased risk of fistula formation, including diabetes mellitus, smoking, infection, peripheral vascular disease, chronic steroid use, malignancy, or previous tissue injury.

Obstetric Fistulas

The vast majority of vesicovaginal fistulas that occur in developing countries are caused by obstetric trauma. Of 377 cases reported by Lawson (1989) from Ibadan, Nigeria, 369 (97.9%) were obstetric and 343 were a consequence of obstructed labor.

Because knowledge of vesicovaginal fistulas in the developing world is based on women treated in hospitals, the prevalence is probably underestimated considerably. Many women are unaware that the condition is treatable and are prevented from learning about appropriate care by severe social isolation as a result of their incontinence. Few area hospitals have the staff, equipment, or expertise to manage the overwhelming problem. Poverty, long distances, and long waiting lists deter women from traveling to major centers.

Obstructed labor remains the most important cause of vesicovaginal fistulas in developing countries. Absent or untrained birth attendants, reduced pelvic dimensions (caused by early childbearing, chronic disease, malnutrition, and rickets), uncorrected inefficient uterine action, malpresentations, hydrocephalus, and introital stenosis secondary to tribal circumcision all contribute to obstructed labor. Prolonged impaction of the presenting fetal part against a distended edematous bladder eventually leads to pressure necrosis and fistula formation. Fistulas may be caused by trauma from forceps, instruments used to dismember and deliver stillborn infants, and surgical abortion. They are also associated with symphysiotomy, the practice of Gishiri cuts (i.e., an incision in the anterior vaginal wall, made for various obstetric and gynecologic disorders), and the use of traditional postpartum vaginal caustics.

In modern obstetrics, most of these conditions do not exist; however, genitourinary fistulas still occur. Vesicovaginal fistulas can follow cesarean delivery or peripartum hysterectomy (particularly in the presence of distorted anatomy, e.g., massive fibroids), massive hemorrhage, and surgical inexperience. Vesicouterine fistulas have occurred more frequently as more women attempt vaginal birth after previous cesarean.

Gynecologic Fistulas

In developed countries, abdominal surgery, particularly total abdominal hysterectomy, is the major cause of genitourinary fistulas. In the United States, vesicovaginal fistulas are caused by benign gynecologic surgery (80%), obstetric events (10%), surgery for malignancies of the cervix, uterus, or ovary (5%), and pelvic radiotherapy (5%). Of 166 cases from the United Kingdom, 116 (69.9%) were related to surgery and only 21 (12.6%) to obstetrics. Fistulas related to surgery performed by obstetrician-gynecologists account for approximately 80% of all urogenital fistulas. The remaining 20% is divided among urologists and colorectal, vascular, and general surgeons. Because gynecologic surgery is the cause of the majority of urogenital fistulas, gynecologic surgeons should be skilled in their identification and management. Most genitourinary fistulas that result from gynecologic surgery occur secondarily to urinary tract injuries. These injuries can occur with pelvic surgery performed by even the most experienced surgeons. Most post-surgical vesicovaginal fistulas are caused by vascular trauma or by unrecognized cystotomies. These injuries occur from primarily blunt dissection of the bladder during mobilization of the bladder flap during total abdominal hysterectomy. This maneuver can result in devascularization or an unrecognized tear in the posterior bladder wall. This results in tissue ischemia, necrosis, and, finally, fistula formation.

Predisposing risk factors for vesicovaginal fistula include a history of pelvic irradiation, cesarean section, endometriosis, previous pelvic surgery or pelvic inflammatory disease, diabetes mellitus, concurrent infection, vasculopathy, and tobacco use. The reported incidence of vesicovaginal fistula after hysterectomy is approximately 1 in 1300 surgeries. However, only a few large series are known in which to base reliable estimates of risks. In a study from Dublin on 17 cases of posthysterectomy fistulas reported over a 15-year period, the estimated risk of fistula was 1 in 605 for total abdominal hysterectomy, 1 in 571 for vaginal hysterectomy, and 1 in 81 for radical hysterectomy. In a recent study in Finland, Harki-Siren et al. (1998) 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 reported. The incidence of bladder injury was 1.3 per 1000 hysterectomies. The incidence of vesicovaginal fistula 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. The risk of ureteral injury was greater with laparoscopic procedures than with open procedures.

Bladder and urethral injury are also known complications of anti-incontinence procedures and procedures for pelvic organ prolapse. A recent increase has occurred in these types of fistulas seen with the popularity of numerous synthetic materials used for sling procedures and prolapse repairs. Kobashi et al. (1999) reviewed cases in which sling removal is required after the use of a woven polyester sling treated with pressure injected bovine collagen (ProteGen, Boston Scientific, Neddick, MA). Thirty-four women in a 2-year period required sling removal, and six had developed a urethrovaginal fistula. Also, a few case reports are known of periurethral injection of a bulking agent, resulting in the development of a bladder neck fistula.

Radiation therapy, used for carcinoma of the cervix or other pelvic malignancies, may rarely result in fistula formation. Healthy tissues of the anterior vaginal wall tolerate radiation doses as high as 8000 rads. Fistulas may first appear during the course of radiotherapy, usually from necrosis of the tumor itself, or after treatment is completed. Late fistulas arise secondarily to endarteritis obliterans usually within the first 2 years. In planning a repair, ruling out recurrent malignancy with biopsy of the fistula margins is essential. Because of decreased vascularity of the adjacent tissue, healing is impaired and must be considered when planning repair of a radiation-induced fistula.

Lee et al. (1988) reviewed 303 women with genitourinary fistulas treated at the Mayo Clinic. Gynecologic surgery was responsible for 82%, obstetric events for 8%, radiation therapy for 6%, and trauma or fulguration for 4%. Seventy-four percent of fistulas resulted from gynecologic surgery for benign conditions, most commonly fibroids, dysfunctional uterine bleeding, prolapse, incontinence, carcinoma in situ, and endometriosis. Fistulas occurred after surgery for malignancy in 42 patients (14%). This review included 53 patients with urethrovaginal fistulas, of whom 10 also had vesicovaginal fistula. Antecedent events included vaginal surgery for incontinence or cystocele, urethral diverticulum repair, treatments for gynecologic cancer, and the use of forceps.

Although suture ligation causing necrosis may lead to a fistula, the mere presence of suture probably does not. Using a rabbit model, Meeks et al. (1997) demonstrated that suture placed through the vaginal cuff and bladder was not associated with the development of vesicovaginal fistula. Posthysterectomy fistulas are usually located above the interureteric ridge, medial to both ureteral orifices. Unlike obstetric fistulas, massive tissue loss is uncommon.

Ureterovaginal fistulas are preceded by benign gynecologic surgery in 90% of cases. The remaining 10% are divided between surgery for malignant disease, pelvic radiotherapy, and obstetrics.

Urethrovaginal fistulas have historically been relatively uncommon, although they are becoming more common with the increased popularity of synthetic midurethral slings. They can also occur after surgery for urethral diverticulum, anterior colporrhaphy, needle suspension procedures, or radiation therapy. Although rare, pressure necrosis that results in a urethrovaginal fistula can occur from prolonged bladder drainage with an indwelling transurethral Foley catheter.

Vesicouterine and vesicocervical fistulas are rare and are usually complications of obstetrical surgery, occurring most frequently after a cesarean section.

Gynecologic vesicovaginal and urethrovaginal fistulas are best classified as being either simple or complicated. Complicated urethrovaginal fistulas are those that involve the proximal urethra and bladder neck and radiation-induced fistulas. Complicated vesicovaginal fistulas include previous radiation, pelvic malignancy, compromised vaginal length more than 3 cm in size, and fistulas involving the trigone.

PRESENTATION AND INVESTIGATION

Patients with genitourinary fistulas present in many ways. Gross hematuria or abnormal intraperitoneal fluid accumulation (urinoma) noted during or after surgery should raise suspicion of an unrecognized urinary tract injury and dictates immediate investigation. In the postoperative period, symptoms may develop after an interval of days, weeks (surgical and obstetric fistulas), months, or even years (radiotherapy-related fistulas). Post-surgical fistulas usually present 7 to 21 days after surgery. Most patients have urinary incontinence or persistent vaginal discharge. If the fistula is very small, leakage may be intermittent, occurring only at maximal bladder capacity or with particular body positions. Other signs and symptoms include unexplained fever; hematuria; recurrent cystitis or pyelonephritis; vaginal, suprapubic, or flank pain; and abnormal urinary stream.

The initial evaluation of all patients with symptoms of genitourinary fistulas starts with a complete physical examination. A thorough speculum examination of the vagina may reveal the source of fluid, which can then be collected; measurement of its urea concentration may identify it as urine. Urine should be examined microscopically and cultured, and appropriate treatment should be instituted for infection. Urethrovaginal fistulas are usually easily diagnosed on physical examination. Further office evaluation, cystourethroscopy, and intravenous urogram permit the physician to localize the fistula, determine adequacy of renal function, and exclude or identify other types of urinary tract injury.

Office testing is often able to distinguish between fistulas involving the bladder or ureters. Instillation of methylene blue or sterile milk into the bladder stains vaginal swabs or tampons in the presence of a vesicovaginal fistula. If this test is not diagnostic, a transurethral Foley catheter should be placed to prevent any staining of the distal tampon from the urethral meatus. Unstained, but wet, swabs may indicate a ureterovaginal fistula. Intravenous indigo carmine can be given and the tampon observed for blue staining. Use of intravenous methylene blue must be chosen with caution because of the risk of methemoglobinemia, a rare but serious complication. If leakage is not demonstrated, the bladder is filled to maximum capacity and provocative maneuvers, such as Valsalva or manual pressure over the bladder, are used to reproduce and confirm the patient’s symptoms.

Radiologic imaging is recommended in most cases and usually includes intravenous urography or cystoscopic retrograde urography. Renal ultrasound may miss up to 20% of ureteral injuries. A Tratner catheter may be useful in cases of suspected urethrovaginal fistula.

Cystourethroscopy is indicated in most cases. The size, site, and number of fistulas and condition of local tissues are carefully noted. Key observations include the fistula’s proximity to the bladder neck, urethral sphincter, and ureteral orifices as well as the presence of tissue edema, slough, infection, induration, scarring, and fixity to bone. Water cystoscopy may be impossible with large fistulas. Placing the patient in the genupectoral position allows the bladder to fill with air, thus permitting dry cystoscopy. Bladder calculi and nonabsorbable sutures should be removed. Associated problems, such as rectovaginal fistulas, anal sphincter disruption, and vaginal stenosis, must be identified and plans made for appropriate treatment.

CONSERVATIVE MANAGEMENT

Various conservative or minimally invasive therapies are available for vesicovaginal and ureterovaginal fistulas. However, the true viability and success of these treatment modalities remain to be determined. The most conservative treatment of a vesicovaginal fistula is simple bladder drainage. A small series described four patients who underwent successful postoperative vesicovaginal closure by simple bladder drainage (Davits and Miranda, 1992). Previously, it has been empirically stated that fistulas diagnosed within 7 days of occurrence, that are less than 1 cm in diameter and unrelated to malignancy or radiation, may spontaneously resolve with up to 4 weeks of continuous drainage in anywhere from 12% to 80% of cases. Again, this outcome has not been proven and still remains unpredictable. Fibrin therapy has been used to treat rectovaginal fistulas with varying degrees of success and is now being applied to treat vesicovaginal fistulas. Case reports have shown success when using the treatment in fistulas that are smaller than 3 mm (Morita and Tokue, 1999). Other methods of treatment of vesicovaginal fistula repairs include electrofulguration and laser ablation of the fistulous tract (Dogra and Nabi, 2001).

Once the diagnosis of a ureterovaginal fistula is confirmed, recommended initial management is ureteral stenting (de Baere et al., 1995; Selzman et al., 1995). Stenting is more successful when performed sooner rather than later; in one study, 82% of attempts in patients whose fistulas were less than 1 month old were successful, compared with 33% with older fistulas. High success rates of stenting and complete resolution of fistulas have been reported when both antegrade and retrograde techniques were used together.

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