Introduction

Ileal conduit is the eldest procedure of urinary diversion after cystectomy and has been practised for more than half a century. It is preferred to orthotopic neobladder in patients with a shorter life expectancy or patients of whom is expected that they cannot complete the necessary rehabilitation or education following orthotopic neobladder [1,2,3]. Long-term studies have shown that the rate of complications in ileal conduits may reach up to 66%. Fistulas are a rare complication of ileal conduit [2]. Fistulas of the ileal conduit or ureter with vessels, bowel, or skin are reported in the existing literature. To our knowledge, no data about ureteroureteral fistulas has been published yet.

We present a rare clinical case and discuss it with the available literature. Finally, we describe a solution using fluoroscopy/endoscopy to determine the optimal place to re-anastomose the ileal conduit to the upper urinary tract.

Case Presentation

A 73-year-old Caucasian man with non-muscle invasive high-grade urothelial carcinoma of the bladder failed to BCG due to intolerance and developed a cripple bladder. Prior to the surgery, he was a retired man living independently with few comorbidities and a good performance status (WHO 0). He underwent a cystoprostatectomy, pelvic lymphadenectomy, and an ileal conduit urinary diversion. The patient had a large right inguinal hernia which was repaired without mesh during this operative session too. A previous large incisional hernia post cholecystectomy, to be repaired with a foreign body mesh, was not repaired in order to limit the impact of the operative procedure and avoid potential infectious complication of the mesh. The urinary stoma was planned for the left abdominal under-quadrant, due to the aforementioned incisional hernia. As a result, the right ureter was tunnelled beneath the sigmoid to the left side. Both ureters were spatulated and splinted. The right ureter was sutured using two running sutures with 5.0 monofilament PDS to the ileal conduit as an end-to-end anastomosis and the left ureter as an end-to-side using the same suturing technique. There was approximately 1 cm of ileal tissue between these two ureters anastomosis. Ureteric splints were removed on day 14. Post-operative recovery was unremarkable. The pathology report revealed radical resection of non-muscle invasive (T1) high-grade urothelial carcinoma.

Four months after the surgery, the patient presented to the emergency department due to dyspnoea caused by segmental embolisms of the lungs which were treated successfully without residual cardiopulmonary issues by initially using a therapeutic dosage of nadroparin and after by a 3-month edoxaban treatment regime. During the routine blood test, a raised creatinine level of 129 µmol/l (normal range for men 60–110 µmol/l) was discovered. Sonography revealed bilateral hydro-ureteronephrosis (Fig. 1).

Shortly after, the stomal urinary production became minimal, and a nephrostomy was introduced to the right kidney after informed consent of the patient was obtained. To limit the impact for the patient, only a one-sided nephrostomy was performed in the acute situation. The right kidney was chosen as it was deemed to be more compromised but still had sufficient renal cortex. As a result, unexpectedly, the right- as well as the left-sided hydronephrosis diminished, as assessed by ultrasound. Stomal urinary production ceased entirely. Meanwhile, the creatinine levels improved the following days to a normal value of 93 µmol/l.

First, an X-ray with contrast via the urostomy as loopogram was conducted. There was no reflux from the ileal conduit into the ureters (Fig. 2). This finding indicated that there had to be a stenosis at the junction of the ileal conduit and ureters. Thereafter, an anterograde pyelography was performed. A contrast agent was introduced through the nephrostomy of the right kidney. Contrast went down the right ureter and did not pass through the ureteroileal anastomosis but passed right through the left ureter up to the left kidney (Fig. 3). Thereby, the stenosis at the ureteroileal anastomosis as well as the connection between the two ureters was proven.

Before further interventions, the renal function was assessed by MAG3 scans. It revealed 66% function of the left and 33% of the right kidney. Moreover, there was a significant outflow obstruction of both kidneys when the right nephrostomy was clamped, which dissolved when the nephrostomy was open.

Endoscopic intervention to resolve the obstruction took place without any success. By trying to antegradly catheterize the ileal conduit through the nephrostomy, a perforation occurred of the distal ureter which was managed conservatively.

Approximately 4 months after the initial presentation to the emergency department, a re-laparotomy was performed following informed consent to make a neo-anastomosis from the ureters to the ileal conduit. An antegrade pyelogram using contrast and blue agent was performed during the laparotomy to visualise the ureter-ureter fistula and to estimate the best position of the new anastomosis between the ileal conduit and the ureters-loop (Fig. 4). The fibrotic end of the ileal conduit was resected and sent to pathology which revealed sclerotic connective tissue with no recognisable urothelial tissue and no sign of malignancy. The ileal conduit was then anastomosed end-to-side with the ureter-ureter loop with running 5.0 PDS monofilament using a single J stent for 10 days.

In the days after the neo-anastomosis, the nephrostomy catheter could be removed, and the stomal production as well as the creatinine levels remained within the normal range. During the 4-year follow-up, the kidney function remained stable.

Conclusions

Ureteroureteral fistula following ileal conduit diversion according to Bricker is extremely rare and has not been reported earlier. We suspect that total obliteration of the oral end of the ileal conduit, which is a rare phenomenon on its own, caused fistula formation of the ureters as observed in antegrade pyelography. Other types of fistula between the ureters and the vessels or bowel have been related to radiotherapy or surgery and fluid leakage and are more common [4].

Studies have shown that 4 to 14% of the patients developed a ureteroileal anastomotic stricture after ileal conduit urinary diversion, mostly unilaterally at the left ureter [5, 6]. Ureteral stents are common practice to prevent ureteroileal anastomotic stricture. However, it has been shown that the incidence of the strictures is higher in stented patients when compared to non-stented patients. The complications related to the stents may even be the potential cause for the stricture formation. Therefore, routine prophylactic stents might prove to be obsolete in the future [7]. Other surgical factors associated with ureteroileal anastomotic stenosis are the technique of anastomosis, ureter ischemia, and non-refluxing anastomosis [8,9,10]. Non-surgical factors associated with stenosis are urinary fistulas and urinary tract infections. Tumour recurrence has to be taken into account as well [11, 12].

The presentation of patients with a ureteroileal anastomotic stenosis may be symptomatic or asymptomatic (71%). Possible symptoms are flank pain, urinary tract infections, a decrease in renal function, or the incidental radiological finding of hydronephrosis. Early detection is important to preserve renal function. Relative renal function and outflow obstruction can be identified by diuretic nuclide renogram as has been done in our patient [13]. To ensure the preservation of the renal function, timely kidney drainage is required. Depending on the severity of the stenosis, interventions to dilate the stricture, or as in our case revise the anastomosis, can be indicated. The endoscopic approach with balloon dilatation or ureterotomy is the preferred option with a success rate of 70% [14]. If the endoscopic approach fails, surgical revision is required [13, 15].

We would like to address this rare phenomenon as a complication of ileal conduit to be mentioned in the differential diagnosis if the outcome of urine is in question. It is not a simple stenosis of the distal portion of the ureters and demands an adapted surgical reconstructing technique as well. It requires the positioning of the anastomosis to be tension-free using fine mono-filament sutures. This could be challenging while the surgical background is fibrotic and difficult to recognise and reconstruct. A combination of X-ray and endoscopic view during the laparotomy may give guidance.