This column summarizes patient cases posted to SIR Connect (SIR’s popular online member community), the responses from other SIR members and how that feedback helped the original poster. To see how SIR’s online community can help you, visit SIR Connect at connect.sirweb.org.
Original post, lightly edited for flow:
75 yo F with a history of SCC involving the urethra. The patient is post cystectomy, ileal conduit and pelvic chemoradiation in 2015.
In late 2018, the patient had a left ureteral stricture and hydronephrosis managed by perc neph -> antegrade NU -> retrograde trans-stomal NU. The retrograde NU was upsized to 12 and then 14 Fr with the intent of getting her tube free.
Has anyone ever been this aggressive with NU upsizing? Up to 14 Fr?
She developed massive hematuria earlier this month (attached pyelogram through multi side hole catheter). Further evaluation led to a diagnosis of left ureter-iliac artery fistula. I managed it with a covered stent graft-atrium iCAST.
She is now post procedure day 5 and recovering very well. The bleeding has stopped, urine output is clear with no signs of infection, and kidney function is improving.
Vascular surgery/uro-oncology want to take her for an RP approach iliac resection and venous bypass graft with possible nephrectomy or proximal ureteral ligation-perc nephrostomy.
I find that aggressive. I feel that a conservative approach has a role and have been trying to look up reports/literature.
Has anyone managed this with chronic prophylactic antibiotics and continued a diverting NU stent? Is there any literature that you find compelling one way or another?
I felt that NU upsizing alone is not the cause of this problem but that, due to the setting of radiation and prior operative mobilization of ureter, aggressive upsizing could have caused this problem. Thoughts?
What specifically prompted you to reach out regarding this case?
Long-term management remains controversial and decisions regarding next steps and long-term solutions were difficult. I managed the initial emergent problem with a covered iliac artery stent graft and the patient was doing well. My options were to pursue a conservative approach (continued urinary diversion and long-term antibiotics) or a major surgical intervention which would include resection of the iliac artery covered stent graft alongside venous bypass, ureteral implantation or nephrectomy.
The decision was tough due to the rarity of this condition and lack of existing guidelines. Case reports and series are available, but none focus sufficiently on long-term management and outcomes. Therefore, I reached out to the SIR Connect community hoping to receive a collective opinion from those who have been in this situation.
What post or posts were most valuable to you and why?
I received replies from various IRs who are more experienced than me. They shared their own cases (for most n=1), images, experience and any literature that they were aware of. It was interesting that the opinions were different and almost evenly split between conservative and surgical management. Their replies reinforced my thought that there is no well-defined algorithm.
Do you find that the responses will shape your practice pattern and lead to an outcome that you did not think could have been achieved otherwise?
Overall, yes. However, this is a rare diagnosis and there is a good chance I may not see it again in my career. I believe most IRs have never had to manage this condition. As far as the outcome in my case: the patient underwent surgery but did not do well, had poor quality of life from postoperative complications and passed away 5 months after surgery.
This experience has made me realize that we need to do our best to compile the published literature out there for the benefit of the IR community. Therefore, we plan to write a manuscript about this case, focusing on the dilemma of long-term management of uretero-iliac fistula by reviewing the available IR, urology and vascular surgery literature.
Additional commentary
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Retrograde multi-sidehole ureteral stent contrast injection demonstrating antegrade contrast flow within the left common iliac artery (arrow) due to the presence of a uretero-arterial fistula (UAF).
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Distal aortogram depicting successful initial left UAF exclusion using a covered stent graft (arrow).
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Contrast-enhanced computed tomography 2.5 months post iliac resection, venous bypass and nephrectomy demonstrating contrast extravasation and pseudoaneurysm formation (arrow) at the site of bypass.
Uretero-arterial fistula (UAF) is an extremely rare entity that is postulated to be increasing in frequency due to increased cancer survival, multimodal pelvic cancer treatments and chronic ureteral stenting.1,2 van den Bergh et al. evaluated 139 cases reported between 1899 and 2008, 93% of which were after 1970.3
Generally, UAF is classified as primary (15%) or secondary (85%).2,4,5 Primary UAFs are native while secondary UAFs can form after surgery, vascular graft placement, irradiation, retroperitoneal fibrosis or ureteral stenting.
The pathophysiology of secondary UAF is complex and appears to involve a combination of inflammation, ischemia and fibrosis.2,5 Ultimately, the UAF forms most often where a ureter crosses an artery (usually the common or external iliac) or anastomosis, and an associated pseudoaneurysm is reported in 38% of cases.2,3
Patients most commonly present with hematuria which can be microscopic or macroscopic and range from intermittent to massive, often declaring itself at ureteral stent exchange/removal.2,3
Diagnosis of UAF is challenging. Cystoscopy can demonstrate bleeding at the ureteral orifice. Contrast-enhanced CT was reported to be helpful in 42% of cases3,6 and can show an enhancing pseudoaneurysm or hydronephrosis with ureteral clot; however, the UAF will not be readily apparent.5 Angiography will demonstrate flow between the ureter and artery, active extravasation, or intimal irregularity and is reportedly confirmatory of UAF in 69% of cases.3,5
Endovascular treatment involves stent-graft placement which allows exclusion of the UAF while permitting perfusion of distal vasculature. Alternatively, coil-embolization of the involved artery may be performed although vascular compromise may result. As treatment is often multidisciplinary, surgical options include vascular bypass, simple ligation, sewing over the fistula, placing a vein patch, interposition grafting, ureteral re-implantation or nephrectomy/ureterectomy.5
Although outcomes data remains sparse, a recent retrospective study by Fox et al. examined at 19 patients with similar circumstances to the original poster’s index case. These patients developed UAF after pelvic surgery, radiation and long-term ureteral stent placement.7 Approximately 70% of patients underwent endovascular repair and the authors reported high rates of lower extremity complications (including ischemia) after endovascular or surgical repair—50% and 67%, respectively.5,7 Moreover, recurrent hemorrhage was reported in up to 14% of patients with 50% mortality. Continual reporting, retrospective analyses and prospective data collection will be paramount for understanding treatment options in UAF.
References
- Turo R, Hadome E, Somov P, et al. Uretero-arterial fistula: Not so rare? Curr Urol. 2018;12(1):54–56.
- Madoff DC, Gupta S, Toombs BD, et al. Arterioureteral fistulas: A clinical, diagnostic, and therapeutic dilemma. AJR Am J Roentgenol. 2004;182(5):1241–1250.
- van den Bergh RC, Moll FL, de Vries JP, Lock TM. Arterioureteral fistulas: Unusual suspects-systematic review of 139 cases. Urology. 2009;74(2):251–255.
- Bergqvist D, Pärsson H, Sherif A. Arterio-ureteral fistula: A systematic review. Eur J Vasc Endovasc Surg. 2001;22(3):191–196.
- Pillai AK, Anderson ME, Reddick MA, Sutphin PD, Kalva SP. Ureteroarterial fistula: Diagnosis and management. AJR Am J Roentgenol. 2015;204(5):W592–598.
- Muraoka N, Sakai T, Kimura H, et al. Endovascular treatment for an iliac artery-ureteral fistula with a covered stent. J Vasc Interv Radiol. 2006;17(10):1681–1685.
- Fox JA, Krambeck A, McPhail EF, Lightner D. Ureteroarterial fistula treatment with open surgery versus endovascular management: Long-term outcomes. J Urol. 2011;185(3):945–950.