Introduction

The most frequent urogenital fistulas following delivery are vesicovaginal fistulas. Vesicouterine fistulas (VUF) are much less common and occur when the uterus has been breached either by rupture in labour or more frequently during caesarean section [1]. As the rate of caesarean sections has increased in the last 2 decades, VUF have been reported more and more often. Up until 1986 Tancer had collected 92 cases of VUF [2], while in 1999 Jozwik et al. [1] reported 796 cases, mostly iatrogenically complicated caesarean sections. These figures suggest that VUF are now more frequently encountered than before. In this paper we report a new case of VUF and discuss diagnostic and treatment issues of this condition.

Case report

A 68-year-old woman, mother of three children, was hospitalised for a moderate urine leak. Her history was significant with urine leak from the vagina for 35 years, starting just after a forceps delivery. The patient suffered three spontaneous abortions in the following 4 years. Questioning revealed a history of cyclic hematuria starting concomitantly with vaginal urine leak. Physical examination revealed a moderate urine loss from the vagina. Speculum examination showed spontaneous urine leak through the cervical ostium. These urine losses turned to blue after instillation of dilute methylene blue into the bladder which led to suspecting the diagnosis of VUF. This diagnosis was confirmed by cystography showing a fistulous track between the uterus and bladder (Fig. 1).

Fig. 1
figure 1

Oblique view of retrograde urethrocystography showing a communication between the posterior aspect of the bladder (B) and the isthmus of the uterus (U).

Full size image

An excretory urogram showed a normal appearance of both kidneys and ureters. The fistula was approached transperitoneally. The posterior bladder wall was adherent to the anterior uterine wall at the isthmus level with dense fibrous tissue. The bladder and uterus were mobilised and the fistulous track, 1.5 cm large, was excised. The bladder was closed in two layers with interrupted resorbable sutures and the uterus was closed in a single layer. The bladder was drained with a Foley catheter for 14 days and the patient was discharged after catheter removal. Convalescence was uneventful and the patient became dry.

Discussion

Vesicouterine fistula is an infrequent condition representing only 1% of all bladder fistulas [1] and affects mostly young women. Although the majority of reported cases were iatrogenic secondary to caesarean sections [1], occasionally other causes have been reported such as rupture of the lower uterine segment and bladder during complicated labour [3], uterine curettage [4], manual removal of placenta [5], tuberculosis of the bladder [6] and an intrauterine contraceptive device [7].

The main symptom of a VUF is urinary leak from the vagina and cyclic hematuria also referred to as menouria. Vesicouterine fistulas can have one of three clinical presentations: (1) amenorrhea and cyclical hematuria, (2) urinary loss from the vagina and (3) the combination of all these symptoms and signs [8].

Cyclic hematuria suggests urinary endometriosis, but the combination of a caesarean section, amenorrhea and cyclic hematuria in the absence of urinary incontinence had been described by Youssef in 1957 as a syndrome that is pathognomonic of VUF and was given Youssef’s eponym [9]. Youssef explains the lack of urinary loss via the uterus in this case by the fact that the isthmic sphincter may maintain sufficient pressure to prevent the escape of urine [10].

In the presence of urine leak, the diagnosis can be confirmed with the aid of methylene blue dye injected into the bladder. If the dye does not come through the cervical ostium, a ureterovaginal fistula must be excluded. Cystography confirms the communication between bladder and uterus and is a very important diagnostic tool. Cystoscopy is necessary to assess the site of the fistula in relation to the ureteral orifices, and intravenous urography is essential to assess the condition of the upper urinary tract and eliminate associated ureteral lesions.

Many authors reported the success of conservative treatment [11]. This requires two conditions: early fistula discovery—less than 3 months—and stopping the fluid flow through the fistula. In fact, when detected early, Youssef’s syndrome can be treated conservatively with continuous bladder drainage by means of an indwelling catheter and oral administration of an estrogen and progesterone combination to induce amenorrhea and stop menouria [11]. A few cases of spontaneous closure of tiny fistulas with or without use of continuous drainage of the bladder have been reported [12].

Transurethral electrocoagulation of the fistulous track may be tried in the presence of a small fistula [8]. Open surgery represents the most common treatment modality. Injuries of the bladder discovered during caesarean operation should be repaired immediately; otherwise, surgical repair should be delayed for at least 2–3 months to allow the inflammation to subside and the uterus to involute completely.

The transperitoneal approach is preferred to the intravesical one. Treatment consists of an excision of the fistulous track and multilayered closure of the bladder and uterus with absorbable sutures. Hysterectomy with closure of the bladder is also proposed if future pregnancies are not desired. Laparoscopic treatment was also proposed to treat vesicocervical fistulas [13].

Although all the studies reported regression of urinary symptoms after surgery, it was classically thought that fertility was compromised after VUF repair [14], presumably because of a deleterious effect of urine on the genital tract. Nevertheless, Lotocki et al. in 1996 [15] reported on a 31.25% pregnancy rate and 25% full-term deliveries among 16 women who had had their VUF surgically repaired. It is noteworthy, however, that among these 16 patients, 1 patient had had her fistula open for 80 months before surgery. This patient became pregnant 18 months after reconstruction proving that urine action on the genital tract is reversible.

Conclusion

VUF should be suspected in women with leakage of urine from the vagina, with or without menouria. Diagnosis is based on clinical and radiological findings. Surgical repair is recommended, but conservative management may be tried in some acute cases.