Iatrogenic vesicouterine fistula manifesting as youssef syndrome: A rare case following uterine rupture

A 36-year-old woman, gravida 4, para 4 was referred to us with a history of recurrent, painless hematuria lasting 3 to 4 days each month for the last five years. This bleeding cycle coincided with her menses, though she reported on the other side, oligomenorrhea. Neither vaginal urine leakage nor other associated symptoms were reported. She sought consultation at various healthcare facilities over five years without any accurate diagnosis. She denied any background suggestive of schistosomiasis. Nevertheless, she had an obstetric history of four Cesarean sections (CS). The last one was an emergency CS indicated for obstructed labor and uterus rupture. This was performed with no perioperative accident reported. However, a few months after by, the patient started presenting hematuria, occurring three to four days each month, along with oligomenorrhea. At clinical examination, the patient was afebrile, conscious, with stable vital signs. Abdomen was normal with a Pfannenstiel scar, with no tenderness or palpable masses. Matching clinical history, the symptoms and the clinical features, differential diagnoses of post-caesarian VUF, vesical schistosomiasis and endometriosis were retained. First, schistosomiasis haematobium was excluded: urine filtration and microscopy test was negative. Transvaginal ultrasound coming out normal, endometriosis was excluded as well. Advanced imaging investigations were indicated to exclude VUF. Unfortunately, neither cystoscopy, hysteroscopy, CT, nor MRI was available. We pushed exploration further, doing a 3-swab test instead. Speculum was placed to expose the vagina, and three dry gauze swabs were placed sequentially into the vagina: swab 1 near the urethra, swab 2 in the mid-vagina and swab 3 in contact with the cervix. Catheter was then inserted into the bladder, and diluted methylene blue was instilled until the bladder was full. The patient walked around for 30 minutes. Then, the swabs were carefully removed. The deepest swab was stained with the dye, confirming communication between urinary and upper genital tracts, in our case more likely VUF. Based on the patient's history, the symptoms and the positive 3-swab test, diagnosis of VUF was retained. Laparotomy was indicated and then performed. Intraoperatively, the diagnosis of VUF was confirmed. The uterus was found in anteversion with multiple epiploic-utero-vesical adhesions surrounding it ( Fig. 1 ). Following adhesiolysis, a small fistula (diameter = 5 mm) was identified at the roof of the bladder, communicating with a second fistula on the anterior uterine corpus. The Goh classification ( Fig. 2 ) was 1Ai (distance from the urethra to the VUF more than 4 cm, diameter<1.5 cm without fibrosis). The surgical procedure consisted of adhesiolysis (release of adhesions) and fistulorrhaphy (closure of the fistula). Fig. 1 Intraoperative photograph during laparotomy showing extensive epiploic-utero-vesical adhesions. After complete adhesiolysis, the surgeon is using a probe to delineate the 5 mm fistulous tract located at the roof of the bladder. These findings were consistent with the patient's surgical history of four Cesarean sections and a prior uterine rupture, which likely facilitated the mechanical tethering of the bladder to the uterine corpus. Fig. 1 Fig. 2 Identification of the vesico-uterine fistula (VUF). This site was found to communicate directly with a corresponding defect on the anterior uterine corpus. According to the Goh classification, this was categorized as a 1Ai fistula (distance from the urethra >4 cm, diameter <1.5 cm without fibrosis). Fig. 2 Intraoperative photograph during laparotomy showing extensive epiploic-utero-vesical adhesions. After complete adhesiolysis, the surgeon is using a probe to delineate the 5 mm fistulous tract located at the roof of the bladder. These findings were consistent with the patient's surgical history of four Cesarean sections and a prior uterine rupture, which likely facilitated the mechanical tethering of the bladder to the uterine corpus. Identification of the vesico-uterine fistula (VUF). This site was found to communicate directly with a corresponding defect on the anterior uterine corpus. According to the Goh classification, this was categorized as a 1Ai fistula (distance from the urethra >4 cm, diameter <1.5 cm without fibrosis). In post operative care, urinary catheter was placed for 14 days and the patient received antibiotics (Ceftriaxone 2 g IV as a single prophylactic dose, followed by Cloxacillin 500 mg tablets TDS for 5 days. After removal of the catheter, 3-swab test come out negative. The patient was then discharged. Follow-up at 1, 3, and 6 months after discharge confirmed the complete resolution of the symptoms, the patient reported no more hematuria.

Albin Baraka Munyanderu: Writing – review & editing, Writing – original draft, Validation, Supervision, Methodology, Conceptualization. Lucien Wasingya Kasereka: Writing – review & editing, Visualization, Validation, Methodology, Formal analysis, Conceptualization. Dieumerci Kaseso: Writing – review & editing, Writing – original draft, Data curation. Olivier Mumbere Mulisya: Writing – review & editing, Conceptualization. Augustin Kanyali: Writing – review & editing, Formal analysis. Gabriel Muhini: Writing – review & editing, Formal analysis. Patrick Kahindo Muyayalo: Writing – original draft, Validation, Supervision.

Written informed consent was obtained from the patient for the publication of this case report.

No financial statement to disclose.

Vesico-uterine fistula is a rare but curable cause of cyclic hematuria. The main reported etiology is Cesarean section. Appropriate imaging, especially MRI, CT, cystoscopy, hysteroscopy are crucial for prompt diagnosis. Nevertheless, in settings with limited resources, a critical clinical assessment and the 3-swab test can be crucial for establishing the diagnosis. The definitive treatment is an abdominal surgical approach (laparotomy) to close the fistula.

Vesico-uterine fistula (VUF) is the rarest type of urogenital fistula 1 , 2 , 3 . The overwhelming majority of VUF cases are iatrogenic, with Cesarean section being the main cause. 6 , 10 The risk is particularly elevated in cases of emergency CS, obstructed labor, or when there are complications like wound infection and re-suturing, as often seen in the literature. 5 Our patient's history of four CS, the emergency nature and uterine rupture constituted a high-risk surgical environment where bladder injury was more probable. A high index of suspicion is mandatory in any woman with a history of prior CS presenting with hematuria. The symptoms presentations may vary and often be delayed from the surgical procedure. 5 As in our case, the patient experienced first hematuria, some months after her CS. This implies that a serious approach is necessary to conduct the diagnosis. Nevertheless, painless cyclic hematuria (bleeding synchronized with menses), is the key symptom, of VUF, often termed menouria (urinary bleeding with menstruation). 11 , 12 A classification of VUF based on the routes of menstrual flow has been proposed by Jo'zwik that divides VUF into three types. Type I, characterized by amenorrhea, menouria and complete continence of urine has been known as Youssef's syndrome. Type II is associated with dual menstrual flow via both the bladder and vagina. Type III is associated with normal vaginal menses and lack of menouria, 13 In our case, the clinical presentation aligns with Type I of this classification system, nevertheless, instead of amenorrhea, the patient reported oligomenorrhea. Hematuria is either macroscopic or microscopic. General urinalysis may show red blood cells only during menses. In suspicion of VUF being a probable cause, imaging is the key. 14 Pelvic MRI is recognized as the gold standard, demonstrating the exact location, size, and course of the fistulous tract. Cystoscopy allows for direct visualization of the bladder orifice, which may appear as an area of hyperemic, edematous, or bullous mucosa. Other diagnostic tools may include intravenous urography, hysterosalpingography with low accuracy. 8 In limited context, a 3-swab test may be a highly effective method, critically correlating with clinical history, as the dye passage into the vagina (via the cervix) confirms the communication to diagnose a VUF. 14 In our case, neither cystoscopy, hysteroscopy, MRI, nor intravenous urography were available. An abdominal ultrasound was performed and came out normal and non-specific. In fact, ultrasound is unconventional and ineffective in diagnosis a VUF. 2 Thus, our diagnosis was clinical. We completed by a bed side 3-swab test with Methylene Blue. The deepest swab came out stained with dye. Matching this with meticulous clinical examination and patient's history, we concluded for probable VUF in our patient. We then indicated the surgery for the fistulorraphy, through a laparotomic access, provided that we had no precision on the location, size, and course of the fistulous tract. The findings revealed the uterus in anteversion with multiple epiploic-utero-vesical adhesions surrounding it. After adhesiolysis, a small VUF was identified at the roof of the bladder, which communicated with a second fistula located on the anterior uterine corpus. In fact, adhesions are common after complicated abdominal surgery (like the reported emergency CS with uterine rupture in our patient) and may have contributed to the mechanism of fistula formation by pulling or fixing the bladder to the lower segment of the uterus. Per operatively, we made the fistulorraphy, with closure of the fistula on the bladder and on the uterine separately. In fact, surgical approach by laparotomy is the standard and appropriate management for VUF, aiming for a multilayer closure with interposition of healthy tissue after adhesiolysis. 15 In post-operative care, a bladder catheter was left in place for 14 days and the patient was submitted to antibiotic prophylaxis. In fact, for small VUF, continuous bladder drainage leaving the bladder catheter for at least 10 days often yields good results. 16 In some other cases, for recent and small post-surgery VUF, hormonal treatment to suppress menstruation for 3 to 6 months has also been found to potentially aid in cicatrization. 17 These non-surgical options were not suitable for our case due to the long duration of the patient's symptoms. The fistula was fibrosclerotic. Laparoscopic treatment has also been proposed for VUFs located near the uterine cervix. 18 Our facility had no laparoscopic equipment. We planned an abdominal laparotomy regarding the difficulty of information about the location and the size of the fistula. The surgical procedure was oriented in an exploratory and therapeutical approach. The 14-day indwelling urinary catheter and prophylactic antibiotics in postoperative management is standard designed to ensure continuous bladder drainage and minimize tension on the repair site, promoting healing. 16 The surgical outcome was satisfactory, resolving the symptoms. Follow-up visits at 1, 3, and 6 months confirmed the complete resolution of the cyclic hematuria and the maintenance of normal micturition.

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Vesico-uterine fistula (VUF), also known as uterovesical fistula, represents a rare subset of urogenital fistulae, accounting for approximately 1-4% of all cases 1 , 2 , 3 . The main symptoms of VUF include cyclic hematuria, amenorrhea, and urinary incontinence. In some particular cases, it can present as Youssef's syndrome; classically with cyclic hematuria with menses), amenorrhea (absence of vaginal bleeding), and urinary continence (no urine leakage from the vagina). 4 Although uncommon, the overwhelming majority of VUF cases are iatrogenic, with Cesarean section (CS) identified as the main cause. The mechanism often involves direct injury to the bladder, an abnormal suture line, or inadequate mobilization of the bladder flap during the procedure, with the risk particularly elevated in complicated surgeries, such as emergency CS or obstructed labor. 5 Suspicion is mandatory in any woman with a history of prior CS, presenting with hematuria. 6 Precise assessment is the main prerequisite for successful treatment. Radiological investigations play pivotal role in the evaluation of size and location of fistula, especially CT, MRI or hysterography. 2 , 7 , 8 Cystoscopy is crucial to localize the fistula and assess the diameter. 9 Nevertheless, ultrasound is neither common nor accurate. 2 In limited context without imaging investigations, clinical exploration must be critical in order to establish a diagnosis. We are presenting here a case of a 36-year-old woman with painless cyclic hematuria that began shortly after CS. In a limited-resources context, it came up as a particular case, without advanced imaging investigations, the diagnosis was critical and challenging. Relying on the patient's history, clinical examination, symptoms, and a 3-swab test an assumptive diagnosis of VUF could be established. A laparotomy was performed and revealed a VUF with surrounding omental adhesions, that was successfully repaired.

The authors declare no competing interest.