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Hematuria and death secondary to aortoureteric fistula
CASE REPORT
HEMATURIA AND DEATH SECONDARY TO AORTOURETERIC FISTULA MICHAEL HOLMES, NOELYN HUNG,
AND
MICHAEL HUNTER
ABSTRACT Ureteroarterial fistulae are extremely rare after previous vascular surgery. Eight cases have been described in the English literature. This is the first example of a vascular communication between the aorta and the ureter. All previous cases were ureteroiliac fistulae. Known hydronephrosis in the presence of prior vascular grafting and heavy hematuria should alert the clinician to the possibility of a ureteroarterial fistula. UROLOGY 52: 720–722, 1998. © 1998, Elsevier Science Inc. All rights reserved.
A
n 83-year-old man presented with a history of collapse after the onset of severe urgency, but prior to attempting to void. On regaining consciousness in the emergency department, he complained of severe back pain and voided gross hematuria. He had been discharged 3 days previously from the urology ward after a similar episode requiring transfusion of 4 U of packed cells. This episode had been labeled “clot colic” and he was awaiting cystoscopy. Sixteen years previously he had undergone an elective aortobifemoral graft. Transurethral resection of the prostate had been carried out 12 months before this admission for benign prostatic hypertrophy. He had been admitted with clot retention 6 months ago when he was noted to have left-sided hydronephrosis on a computed tomography (CT) scan of the abdomen, undertaken to investigate his chronic renal failure. The patient also had a stable myeloproliferative disorder and epilepsy. He was not taking aspirin, nonsteroidal anti-inflammatory agents, or anticoagulants. At admission the patient was awake, pale, and sweating profusely. His blood pressure was 200/60 mm Hg and his pulse was 68 beats per minute. The abdomen was soft and nontender to examination with a prominent aortic impulse. Catheterization of the bladder revealed gross hematuria without clots. Hemoglobin was 77 g/L, creatinine 0.22 mmol/L, and urea 12 mmol/L. Electrocardio-
From the Department of Urology, Waikato Hospital, Hamilton, New Zealand; and the Departments of Pathology and General Surgery, Dunedin Hospital, Dunedin, New Zealand Reprint requests: Michael Holmes, M.D., Department of Urology, Waikato Hospital, P.O. Box 19-210 Hamilton, New Zealand Submitted: December 15, 1997, accepted: May 1, 1998
720
© 1998, ELSEVIER SCIENCE INC. ALL RIGHTS RESERVED
graphic sinus rhythm, chest x-ray, and supine abdominal films were normal. The presumptive diagnosis for the collapse was a retroperitoneal hemorrhage from his aortic graft. CT scan of the abdomen documented a large periaortic hematoma believed to be arising from the graft or graft suture line. Left-sided hydronephrosis was again noted, and the left ureter was poorly visualized (Fig. 1). Conservative management was undertaken given the patient’s chronic renal failure and the undetermined nature of his hematuria. His hematuria resolved, and after transfusion he was discharged. He returned less than 24 hours later with a further catastrophic episode of hematuria and died that day. At postmortem examination the graft could be seen to have pulled away at its proximal suture line from the aortic intima. A dissection of the aorta proper at this level had occurred with the formation of a pseudoaneurysm and recent thrombosis. The left ureter was incorporated into the pseudoaneurysm. A guide wire could be placed from the left ureter via the pseudoaneurysm to the disrupted suture line. The left kidney was small, and hydronephrotic (Fig. 2). COMMENT Ureteroarterial fistulae occurring as a result of or complication of vascular graft surgery are extremely rare. In a review by Blasco and Saladie,1 6 cases of such fistulae, 3 in the English literature, were noted with a single case published in French of an aortoureteric fistula. In a Medline search, 5 additional cases of ureteroiliac fistulae occurring after vascular graft surgery were found in the En0090-4295/98/$19.00 PII S0090-4295(98)00281-7
FIGURE 1. Abdominal CT at initial admission with gross hematuria. Left-sided periaortic collection is visible.
glish literature.2–9 All of these 8 previous cases are ureteroiliac fistulae. Ureteroarterial fistulae from other causes are also rare. Quillin and coworkers10 in 1994 stated that there were 33 cases from all causes (including vascular surgery) in the world literature. In all cases the communication was from the iliac artery to the overlying ureter. In addition to previous surgery, fistula may occur secondary to prolonged indwelling stents or pelvic irradiation.11 A single case of a spontaneous common iliac artery aneurysm eroding a ureter also exists.12 Of the 8 cases following prior vascular surgery, 1 occurred from an iatrogenic suture injury2 and was immediately apparent postoperatively. The other 7 all occurred at least 1 year after vascular surgery.3–9 No patient had received radiotherapy. The median time to presentation from time of surgery was 10 years with a range of 1 to 20 years. Two patients had long-term indwelling ureteric stents on the involved side.3,4 In one instance this was for 1 month but postdated the appearance of a urinoma,3 and in the second case the stent was in situ for more than a year before the fistula developed.4 In 2 cases aneurysms had developed in the iliac artery at the site of fistula formation.3,5 In all cases, at operation, the ureter and graft were densely adherent and encased in scar tissue. The presenting feature in all cases was hematuria. This was severe in 3 patients,3,4,6 with the other patients describing mild to moderate intermittent hematuria. Occasionally patients also described flank pain. In 5 of 5 cases, where pre-existing appropriate imaging was available, hydronephrosis had preceded the onset of hematuria.3,4,6,7 Investigations utilized included intravenous urogram (IVU), CT, retrograde pyelograms, and aortograms. In 5 of 5 IVUs performed, hydronephrosis was present5–9 but in no case was a fistula documented. CT performed in 1 case was unhelpful.3 Four patients underwent a retrograde pyelogram3,7–9 and in 2 cases this proved diagnostic.8,9 UROLOGY 52 (4), 1998
FIGURE 2. Postmortem findings, posterior view.
In 1 patient a fistula was displayed8 and in a second patient a noncommunicating tract was seen passing medially from the ureter.9 Aortogram was performed in 5 cases3–5,7,8 and demonstrated the fistula in 1 case alone,8 where very brisk bleeding occurred following the removal of the ureteric catheter. Baum et al.3 described the presence of a fluidfluid line within the ureter in ureteroarterial fistulae on CT. This implies bleeding above the site of the stricture, but not the cause. Digital subtraction angiography or manipulation of a stent if in situ during the angiography can improve the sensitivity of interventional studies for the site of bleeding.10 Given the difficulty (as described) in demonstrating a fistula, it is not surprising that in 2 of the 8 cases involving prior vascular surgery the patient underwent a nephrectomy3,5 on the erroneous assumption that this was the site of the bleeding. Both patients subsequently underwent further exploration after hematuria persisted. In addition, 1 patient in this series died of hematuria while being investigated, before the site of bleeding was delineated.6 Of the remaining 5 patients, 3 underwent primary repair of the graft and ureterolysis2,8,9 and in 2 patients the graft was excised or tied off.4,7 The documented unilateral hydronephrosis, previous vascular grafting (in particular more than 10 years ago), and the intermittent gross, almost catastrophic, hematuria in the present case are therefore characteristic of a ureteroarterial fistula. In addition, the CT findings of an aortic pseudoaneurysm suggests the site of fistula formation. In summary, the triad of unilateral hydronephrosis, intermittent hematuria, and previous aortic or iliac arterial surgery should alert the clinician to this life threatening and potentially curable disease. The optimal investigations would appear to 721
be a retrograde pyelogram with digital subtraction angiography. Ureteric strictures and/or an aneurysm suggest the site of the fistula. In a well patient, exploration with ureterolysis and repair of the graft should be undertaken and should achieve acceptable results in both graft and renal unit. REFERENCES 1. Blasco FJ, and Saladie JM: Ureteral obstruction and ureteral fistula after aortofemoral or aortoiliac bypass surgery. J Urol 145: 237–242, 1991. 2. Schapira HE, Li R, Gribetz M, et al: Ureteral injuries during vascular surgery. J Urol 125: 293–297, 1981. 3. Baum ML, Baum RD, Plaine L, et al: Computed tomography in the diagnosis of fistula. J Comput Assist Tomogr 11: 719 –721, 1987. 4. Nelson HN, and Freid FA: Iliac artery-ureteral fistula associated with Gibbons’ catheter: a case report and review of the literature. J Urol 125: 878 – 880, 1981.
722
5. Joost G, Bartsch S, Weimann S, et al: Ilioureteric fistula. Br J Urol 53: 477, 1981. 6. Shetty SD, Read JR, and Newling DWW: Uretero-arterial fistula. Br J Urol 62: 382–383, 1988. 7. Wheatley JK, Ansley JD, Smith RB, et al: Ureteroarterial fistula. Urology 18: 498 –502, 1981. 8. Shultz ML, Ewing DD, and Lovett VF: Fistula between iliac aneurysm and distal stump of ureter with haematuria: a case report. J Urol 112: 585–586, 1974. 9. Beard JD, Somerville PG, Ward JP, et al: Massive haematuria due to an ilio-ureteric fistula. Br J Urol 58: 332– 343, 1986. 10. Quillin SP, Darcy MD, and Picus D: Angiographic evaluation and therapy of ureteroarterial fistulas. AJR Am J Roentgenol 162: 873– 878, 1994. 11. Abal VC, Veiga FG, Escandon AS, et al: Common iliac artery-ureteral fistula: a case report and literature review. Arch Esp Urol 46: 843– 846, 1993. 12. Rennick JM, Link DP, and Palmer JM: Spontaneous rupture of an iliac artery aneurysm into a ureter: a case report and review of the literature. J Urol 116: 111–113, 1976.
UROLOGY 52 (4), 1998
HEMATURIA AND DEATH SECONDARY TO AORTOURETERIC FISTULA MICHAEL HOLMES, NOELYN HUNG,
AND
MICHAEL HUNTER
ABSTRACT Ureteroarterial fistulae are extremely rare after previous vascular surgery. Eight cases have been described in the English literature. This is the first example of a vascular communication between the aorta and the ureter. All previous cases were ureteroiliac fistulae. Known hydronephrosis in the presence of prior vascular grafting and heavy hematuria should alert the clinician to the possibility of a ureteroarterial fistula. UROLOGY 52: 720–722, 1998. © 1998, Elsevier Science Inc. All rights reserved.
A
n 83-year-old man presented with a history of collapse after the onset of severe urgency, but prior to attempting to void. On regaining consciousness in the emergency department, he complained of severe back pain and voided gross hematuria. He had been discharged 3 days previously from the urology ward after a similar episode requiring transfusion of 4 U of packed cells. This episode had been labeled “clot colic” and he was awaiting cystoscopy. Sixteen years previously he had undergone an elective aortobifemoral graft. Transurethral resection of the prostate had been carried out 12 months before this admission for benign prostatic hypertrophy. He had been admitted with clot retention 6 months ago when he was noted to have left-sided hydronephrosis on a computed tomography (CT) scan of the abdomen, undertaken to investigate his chronic renal failure. The patient also had a stable myeloproliferative disorder and epilepsy. He was not taking aspirin, nonsteroidal anti-inflammatory agents, or anticoagulants. At admission the patient was awake, pale, and sweating profusely. His blood pressure was 200/60 mm Hg and his pulse was 68 beats per minute. The abdomen was soft and nontender to examination with a prominent aortic impulse. Catheterization of the bladder revealed gross hematuria without clots. Hemoglobin was 77 g/L, creatinine 0.22 mmol/L, and urea 12 mmol/L. Electrocardio-
From the Department of Urology, Waikato Hospital, Hamilton, New Zealand; and the Departments of Pathology and General Surgery, Dunedin Hospital, Dunedin, New Zealand Reprint requests: Michael Holmes, M.D., Department of Urology, Waikato Hospital, P.O. Box 19-210 Hamilton, New Zealand Submitted: December 15, 1997, accepted: May 1, 1998
720
© 1998, ELSEVIER SCIENCE INC. ALL RIGHTS RESERVED
graphic sinus rhythm, chest x-ray, and supine abdominal films were normal. The presumptive diagnosis for the collapse was a retroperitoneal hemorrhage from his aortic graft. CT scan of the abdomen documented a large periaortic hematoma believed to be arising from the graft or graft suture line. Left-sided hydronephrosis was again noted, and the left ureter was poorly visualized (Fig. 1). Conservative management was undertaken given the patient’s chronic renal failure and the undetermined nature of his hematuria. His hematuria resolved, and after transfusion he was discharged. He returned less than 24 hours later with a further catastrophic episode of hematuria and died that day. At postmortem examination the graft could be seen to have pulled away at its proximal suture line from the aortic intima. A dissection of the aorta proper at this level had occurred with the formation of a pseudoaneurysm and recent thrombosis. The left ureter was incorporated into the pseudoaneurysm. A guide wire could be placed from the left ureter via the pseudoaneurysm to the disrupted suture line. The left kidney was small, and hydronephrotic (Fig. 2). COMMENT Ureteroarterial fistulae occurring as a result of or complication of vascular graft surgery are extremely rare. In a review by Blasco and Saladie,1 6 cases of such fistulae, 3 in the English literature, were noted with a single case published in French of an aortoureteric fistula. In a Medline search, 5 additional cases of ureteroiliac fistulae occurring after vascular graft surgery were found in the En0090-4295/98/$19.00 PII S0090-4295(98)00281-7
FIGURE 1. Abdominal CT at initial admission with gross hematuria. Left-sided periaortic collection is visible.
glish literature.2–9 All of these 8 previous cases are ureteroiliac fistulae. Ureteroarterial fistulae from other causes are also rare. Quillin and coworkers10 in 1994 stated that there were 33 cases from all causes (including vascular surgery) in the world literature. In all cases the communication was from the iliac artery to the overlying ureter. In addition to previous surgery, fistula may occur secondary to prolonged indwelling stents or pelvic irradiation.11 A single case of a spontaneous common iliac artery aneurysm eroding a ureter also exists.12 Of the 8 cases following prior vascular surgery, 1 occurred from an iatrogenic suture injury2 and was immediately apparent postoperatively. The other 7 all occurred at least 1 year after vascular surgery.3–9 No patient had received radiotherapy. The median time to presentation from time of surgery was 10 years with a range of 1 to 20 years. Two patients had long-term indwelling ureteric stents on the involved side.3,4 In one instance this was for 1 month but postdated the appearance of a urinoma,3 and in the second case the stent was in situ for more than a year before the fistula developed.4 In 2 cases aneurysms had developed in the iliac artery at the site of fistula formation.3,5 In all cases, at operation, the ureter and graft were densely adherent and encased in scar tissue. The presenting feature in all cases was hematuria. This was severe in 3 patients,3,4,6 with the other patients describing mild to moderate intermittent hematuria. Occasionally patients also described flank pain. In 5 of 5 cases, where pre-existing appropriate imaging was available, hydronephrosis had preceded the onset of hematuria.3,4,6,7 Investigations utilized included intravenous urogram (IVU), CT, retrograde pyelograms, and aortograms. In 5 of 5 IVUs performed, hydronephrosis was present5–9 but in no case was a fistula documented. CT performed in 1 case was unhelpful.3 Four patients underwent a retrograde pyelogram3,7–9 and in 2 cases this proved diagnostic.8,9 UROLOGY 52 (4), 1998
FIGURE 2. Postmortem findings, posterior view.
In 1 patient a fistula was displayed8 and in a second patient a noncommunicating tract was seen passing medially from the ureter.9 Aortogram was performed in 5 cases3–5,7,8 and demonstrated the fistula in 1 case alone,8 where very brisk bleeding occurred following the removal of the ureteric catheter. Baum et al.3 described the presence of a fluidfluid line within the ureter in ureteroarterial fistulae on CT. This implies bleeding above the site of the stricture, but not the cause. Digital subtraction angiography or manipulation of a stent if in situ during the angiography can improve the sensitivity of interventional studies for the site of bleeding.10 Given the difficulty (as described) in demonstrating a fistula, it is not surprising that in 2 of the 8 cases involving prior vascular surgery the patient underwent a nephrectomy3,5 on the erroneous assumption that this was the site of the bleeding. Both patients subsequently underwent further exploration after hematuria persisted. In addition, 1 patient in this series died of hematuria while being investigated, before the site of bleeding was delineated.6 Of the remaining 5 patients, 3 underwent primary repair of the graft and ureterolysis2,8,9 and in 2 patients the graft was excised or tied off.4,7 The documented unilateral hydronephrosis, previous vascular grafting (in particular more than 10 years ago), and the intermittent gross, almost catastrophic, hematuria in the present case are therefore characteristic of a ureteroarterial fistula. In addition, the CT findings of an aortic pseudoaneurysm suggests the site of fistula formation. In summary, the triad of unilateral hydronephrosis, intermittent hematuria, and previous aortic or iliac arterial surgery should alert the clinician to this life threatening and potentially curable disease. The optimal investigations would appear to 721
be a retrograde pyelogram with digital subtraction angiography. Ureteric strictures and/or an aneurysm suggest the site of the fistula. In a well patient, exploration with ureterolysis and repair of the graft should be undertaken and should achieve acceptable results in both graft and renal unit. REFERENCES 1. Blasco FJ, and Saladie JM: Ureteral obstruction and ureteral fistula after aortofemoral or aortoiliac bypass surgery. J Urol 145: 237–242, 1991. 2. Schapira HE, Li R, Gribetz M, et al: Ureteral injuries during vascular surgery. J Urol 125: 293–297, 1981. 3. Baum ML, Baum RD, Plaine L, et al: Computed tomography in the diagnosis of fistula. J Comput Assist Tomogr 11: 719 –721, 1987. 4. Nelson HN, and Freid FA: Iliac artery-ureteral fistula associated with Gibbons’ catheter: a case report and review of the literature. J Urol 125: 878 – 880, 1981.
722
5. Joost G, Bartsch S, Weimann S, et al: Ilioureteric fistula. Br J Urol 53: 477, 1981. 6. Shetty SD, Read JR, and Newling DWW: Uretero-arterial fistula. Br J Urol 62: 382–383, 1988. 7. Wheatley JK, Ansley JD, Smith RB, et al: Ureteroarterial fistula. Urology 18: 498 –502, 1981. 8. Shultz ML, Ewing DD, and Lovett VF: Fistula between iliac aneurysm and distal stump of ureter with haematuria: a case report. J Urol 112: 585–586, 1974. 9. Beard JD, Somerville PG, Ward JP, et al: Massive haematuria due to an ilio-ureteric fistula. Br J Urol 58: 332– 343, 1986. 10. Quillin SP, Darcy MD, and Picus D: Angiographic evaluation and therapy of ureteroarterial fistulas. AJR Am J Roentgenol 162: 873– 878, 1994. 11. Abal VC, Veiga FG, Escandon AS, et al: Common iliac artery-ureteral fistula: a case report and literature review. Arch Esp Urol 46: 843– 846, 1993. 12. Rennick JM, Link DP, and Palmer JM: Spontaneous rupture of an iliac artery aneurysm into a ureter: a case report and review of the literature. J Urol 116: 111–113, 1976.
UROLOGY 52 (4), 1998