Challenging case: Persistent hemorrhage following ureteral stent exchange

“Challenging Cases in Urology” is a new Urology Times section in which residents from the nation’s leading urology programs present their toughest cases and how they ultimately managed them. Cases inform readers of the problem-solving process and provide a lesson from the authors’ experience.

History

A 69-year-old woman was transferred from an outside institution to our inpatient service with recurrent gross hematuria. Two weeks prior to transfer, she underwent routine bilateral ureteral stent exchange, which was complicated by gross hematuria with significant clot burden. Over the course of 10 days, she continued to have intermittent hematuria requiring continuous bladder irrigation with associated blood loss anemia necessitating transfusion with 4 units of packed red blood cells (RBCs).

Prior to transfer to our institution, cystoscopy demonstrated slow oozing from the left ureteral orifice, which became brisk upon removal of the left ureteral stent. A retrograde pyelogram revealed a normal-caliber ureter and renal pelvis without filling defects or extravasation. The stent was replaced, which reduced the bloody efflux. At this point a CT without contrast was obtained, which revealed no significant upper tract lesions or abnormalities. The patient was offered a left nephroureterectomy. However, her family desired a second opinion, which prompted transfer to our hospital.

The patient reported no prior episodes of gross hematuria and her previous stent exchanges were uneventful. Additionally, she did not report any flank pain, flank mass, weight loss, voiding symptoms, fevers, or chills. Despite requiring multiple transfusions, she reported no dyspnea, chest pain, or light-headedness.

Her medical history is notable for colon cancer treated by sigmoidectomy in 1995, endometrial cancer, and stage III chronic kidney disease presumably due to ureteral obstruction. In 2009, her endometrial cancer was treated with a total abdominal hysterectomy and bilateral salpingo-oophorectomy (TAH+BSO) followed by pelvic external beam radiation. In 2011, she required a small bowel resection for radiation enteritis. In 2012, she was noted to have bilateral hydroureteronephrosis and an atrophic left kidney, which has been managed with chronic indwelling double-J ureteral stents that are exchanged every 4 months.

She is a retired secretary who has an occasional glass of wine and has never smoked. Her mother was diagnosed with uterine cancer, but her family history was otherwise unremarkable.

Next: Exam, lab, and imaging results

Examination

On exam, our patient was a thin woman who appeared older than her stated age of 69 years. She was hemodynamically stable and a cardiopulmonary exam was unremarkable. Her abdomen was soft without flank masses or tenderness. Midline scars from her prior TAH+BSO, sigmoidectomy, and small bowel resection were noted. Her urine was light pink without clots on slow saline irrigation. Her exam was otherwise unremarkable.

Laboratory workup revealed an eGFR of 44 mL/min and a hemoglobin of 9.4 g/dL. Urinalysis showed gross blood without evidence of infection. A coagulation panel was negative and she was cross-matched for blood.

Vascular surgery and interventional radiology (IR) were consulted to develop a multidisciplinary approach. The patient was given IV hydration and a CT angiogram was obtained (figure 1), which noted no evidence of uretero-arterial fistula, no concerning upper tract or pelvic masses, and well-positioned stents bilaterally.

Figure 1. CT angiogram, axial cut showing the left ureter and stent crossing over the external and internal iliac arteries close to the bifurcation. No communication can be seen.

Next:What's your diagnosis?Discussion and diagnosis

After further discussion, we proceeded to the IR suite for an angiogram. The urology team was present with the patient positioned in “frog-leg” position, prepped, and draped in the event the ureteral stent would need to be removed over a wire to provoke bleeding. Several units of blood were immediately available. Angiography revealed a fistulous connection between the left pelvic ureter and the internal iliac artery (figure 2). The internal iliac artery was embolized with coils and a vascular plug (figure 3).

Figure 2. Angiography showing opacification of the left internal iliac artery with antegrade and retrograde flow of contrast into the ureter.

Figure 3. Angiography of the common iliac artery bifurcation after endovascular embolization of the internal iliac artery. Contrast is no longer seen in the ureter.

During the procedure, she experienced pelvic pressure concerning for bladder filling and voided a moderate volume of clot. She became hypotensive at this time, but improved significantly following embolization and transfusion of a unit of RBCs.

The patient’s hematuria resolved completely by day one after embolization. A percutaneous nephrostomy tube was placed to manage her chronically obstructed left kidney and she was discharged home without a urethral catheter on post-procedure day 2. Her left ureteral stent was removed as an outpatient 2 weeks after discharge.

Uretero-arterial fistulas are uncommon and require a high index of suspicion-and occasionally multiple imaging studies-to diagnose. Our patient underwent a non-contrast CT, retrograde pyelogram, and CT angiogram before her fistula was diagnosed and treated. While data on uretero-arterial fistulas are limited to small retrospective studies, prior series suggest 78% are missed on initial workup (Urology 2009; 74:251–5). IR angiography is the gold standard for diagnosis with a sensitivity of 60% to 100% when combined with ureteral stent manipulation (Ann Vasc Surg 2017; 44:459–65). In comparison, CT angiography and retrograde pyelograms miss 60% and 50% of fistulas, respectively (Am J Roentgenol 2015; 204:W592–8).

Our patient’s presentation and risk factors are consistent with the existing literature. The most common presentation is intermittent hematuria that may be exacerbated by stent exchange and often requires blood transfusion. Multiple risk factors are typically present and include prior pelvic surgery (89% of cases), chronic indwelling ureteral stents (67%), pelvic radiation (43%), prior vascular surgery, malignancy, and intrinsic vascular abnormalities (15%) (Urology 2009; 74:251–5).

Historically, treatment of uretero-arterial fistulas was surgical, which was often complex and morbid due to the frequent incidence of prior surgery and radiation. The development of endovascular technologies has shifted management to the IR suite. The most common sites of fistulization include the common iliac, internal iliac, and external iliac arteries. In patients with urinary diversion, uretero-aortic fistulas may occur (Am J Roentgenol 2015; 204:W592–8).

Endovascular treatment is dictated by the location of the fistula; common iliac and external iliac fistulas can be treated with a covered stent graft, while fistulas to the internal iliac artery can be embolized. If a covered stent is used and traverses the internal iliac artery, the internal iliac should be embolized to prevent an endoleak. In our patient, although surgical therapy in the form of nephroureterectomy was initially offered by an outside institution, she was ultimately successfully managed with IR embolization.

Conclusion

Uretero-arterial fistulas are uncommon and often present a diagnostic and therapeutic challenge. Successful management requires a high index of suspicion and a multidisciplinary approach. In our case, the patient underwent multiple imaging studies and received 4 units of RBCs before angiography ultimately revealed the diagnosis.

Kyle Ericson, MD

Molly E. DeWitt-Foy, MD

Khaled Fareed, MD

Dr. Ericson and Dr. DeWitt-Foy are urology residents, and Dr. Fareed is a staff physician at the Cleveland Clinic Glickman Urological & Kidney Institute, Cleveland.