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THE
Vol. 124, August Printed in U.S.A.
JOURNAL OF UROLOGY
Copyright © 1980 by The Williams & Wilkins Co.
Case Reports IN SITU AND EX SITU RENAL ARTERY ANEURYSMECTOMY IN THE SOLITARY KIDNEYS HIDEO HIDAI, KEN IDE
AND
SATORU FUJISHIMA
From the Departments of Urology, Surgery and Medicine, Yokohama City University School of Medicine, Yokohama, Japan
ABSTRACT
We report 2 cases of renal artery aneurysms in congenital solitary kidneys, which were treated respectively by in situ and ex situ aneurysmectomy. Hyperosmolar intracellular electrolyte solution was valid in preserving renal function during renal ischemia in both cases. The occurrence of an aneurysm in a solitary kidney, ex situ operation and renal preservation during ischemia are discussed. Although rare, a renal artery aneurysm in a solitary kidney is critical because of possible rupture of the aneurysm and because renal preservation during operation is mandatory. Herein we report 2 cases of renal artery aneurysms in solitary kidneys treated by different surgical methods, namely in situ and ex situ aneurysmectomy. CASE REPORTS
Case 1. C. M., a 33-year-old housewife, had been in good health until January 30, 1977 when she noticed vertigo followed by transient unconsciousness. Lower abdominal pain developed and the patient was hospitalized elsewhere. She had anemia and, because of its progressiveness despite blood transfusion, she was referred to our hospital on February 7. A lower abdominal soft tender mass was palpated, which extended to the left flank. The mass showed a cystic pattern on sonography. An excretory urogram (IVP) revealed non-visualization of the right kidney and deviation of the lower third of the left ureter (fig. 1, A). Exploratory laparotomy on February 10 revealed a huge left retroperitoneal hematoma. Arteriography during operation showed absence of the right renal artery and a ruptured non-calcified aneurysm located at the main renal artery (fig. 1, B). Hemorrhage into the retroperitoneal space was considered to cease temporarily at that time. Since the patient had a single kidney and no arrangement for renal preservation during ischemia had been made a definitive operation was postponed and the wound was closed. On February 16 the abdomen was re-explored through a left anterior subcostal incision. The aorta and the renal hilar region were embedded in dense hematoma and our greatest difficulty was encountered during dissection of the renal artery and vein. After securing the renal artery by a vessel tape the ruptured aneurysmal sac began to bleed again. The kidney was cannulated through the aneurysmal sac and flushed with ice cold lactate Ringer's solution followed by slow infusion of 140 ml. hyperosmolar intracellular electrolyte solution (Sacks' solution)1 under electrocardiogram monitoring. The renal artery was transected distal and proximal to the aneurysm. End-toend anastomosis of the renal artery by 5-zero polyester suture was attempted but this was complicated by poor exposure of the operative field owing to an organizing hematoma and friable arterial wall. Despite renal ischemia for 90 minutes the kidney functioned well postoperatively. Convalescence was relatively uneventful except for a tranAccepted for publication October 19, 1979.
sient minimal increase of serum creatinine up to 2.2 mg./dl. and pleural effusion. An IVP 2 weeks postoperatively showed normal renal function. The 24-hour endogenous creatinine clearance 5 weeks postoperatively was 135 1. per day. Although followup angiography revealed mild stenosis and irregularity at the renal arterial anastomosis renal function, plasma renin activity and blood pressure have remained within normal range for the last 2 years (fig. l, C). Case 2. H. T., a 51-year-old housewife, was hospitalized on November 8, 1977 for evaluation of hypertension and backache. In 1967 she had been hospitalized elsewhere because of hypertension and non-visualization of the left kidney and multiple right renal artery aneurysms were found. Backache on the right side had been aggravating during the last several months, necessitating daily analgesics. Physical examination revealed blood pressure of 170/100 mm. Hg and a grade 2 upper abdominal bruit. Urinalysis showed no albumin, and 4 to 6 red and white blood cells per high power field. Blood urea nitrogen was 9 mg./dl., creatinine 0.6 mg./dl., maximum specific gravity on a Fishberg concentration test 1.017, endogenous creatinine clearance 104 1. per day and renal plasma flow 517 ml. per minute. The ocular fundus change was H1S1. Plasma renin activity examinations, including renal vein renin ratio, were within normal limits. A chest film and electrocardiogram were interpreted as normal. Cystoscopic examination revealed an absent left ureteral orifice together with a defective interureteral ridge on the left side. A scout film showed 1 almost completely calcified wreathlike shadow and 3 other crescent calcifications corresponding to the right kidney (fig. 2, A). The left kidney was not visualized on an IVP. Aortography revealed an absent left renal artery and 7 calcified and non-calcified renal artery aneurysms, including a non-calcified aneurysm 30 mm. in diameter, which was located at the main trunk of the solitary kidney. Arteriography also demonstrated mild stenosis of the right internal iliac artery (fig. 2, B). In addition to the renal artery aneurysms, gynecological and neurological examinations revealed other abnormalities, including uterus bicornus and 2 small aneurysms in the cerebral artery region. Initially, we hesitated to perform an aneurysmectomy because of the multiplicity of the renal artery aneurysms, state of solitary kidney and associated intracranial lesions. However, the aggravating backache necessitated more analgesics and suggested urgent rupture of the non-calcified aneurysm. On April 24, 1978 the right kidney was explored through an anterior 274
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FIG. 1. Case 1. A, preope!ative IVP reveals non-visu~tion of right kidney and medial deviation of left ureter. B, intraoperative abdominal aortogr_aPl?-Y sho"".s absent nght r1;nal artery and non-calcified saccular aneurysm on left renal artery. C, postoperative abdominal aortography. Arrow mdicates site of anastomosis.
subcostal incision. After recognition of minimal retroperitoneal bleeding from the largest non-calcified aneurysm, the left iliac fossa was opened for preparation of renal autotransplantation. However, we could not locate the left iliac vein despite the presence of the left iliac artery. An ipsilateral dissection of the iliac fossa for renal transplantation was necessary. Without dividing the ureter, the kidney was extirpated and brought down to the right iliac fossa through a retroperitoneal tunnel between the 2 incisions. The kidney was flushed immediately with 250 ml. heparin added to ice cold Sacks' solution under 100 cm. water pressure through the cannulated renal artery. Excluding 2 intraparenchymal completely calcified aneurysms, 5 were resected and the renal artery was reconstructed by an angioplastic technique, including a pair-of-pants anastomosis and ovarian vein patch graft (figs. 2, C and 3). The kidney was autotransplanted·,to the right iliac fossa in an upside-down fashion. The ureter was left in place in an inverted S fashion without a fixing suture. During 6 hours 15 minutes of renal ischemia no surface cooling was attempted and the kidney was kept in ambithermic condition except for flushing with 50 ml. ice cold Sacks' solution 3 times. Postoperative anuria for 6 days was controlled by daily hemodialysis. The blood urea nitrogen and serum creatinine returned to normal levels 19 days postoperatively. The 24-hour endogenous creatinine clearance 9 weeks postoperatively was 68.5 1. per day. The patient's general condition improved gradually, despite persistent nausea and vomiting owing to refractory hyponatremia. The 24-hour endogenous creatinine clearance was 116 1. per day and the renal plasma flow was 330 ml. per minute 3 months postoperatively. Angiography showed patent renal blood flow except for stenosis at the lower polar branch anastomosis and no progress of
stenosis at the internal iliac artery, which had been demonstrated preoperatively (fig. 4). Although there was no change in the hypertensive state the patient was discharged from the hospital on August 8 in good condition. She was doing well 1 year postoperatively. DISCUSSION
An operation for a renal artery aneurysm is indicated when hypertension is attributable to that aneurysm or when the aneurysm has the possibility of rupture. A non-calcified or incompletely calcified aneurysm and a large aneurysm (> 15 mm. in diameter) are believed to have the possibility of rupture.2 If possible renal revascularization following aneurysmectomy is the method of choice. In our 2 cases preservation of the solitary kidney was mandatory. In situ aneurysmectomy was done in case 1 because of the possibility of future pregnancy and the state of the aneurysm. Although poor exposure of the operative field owing to dense hematoma and friable arterial wall necessitated 90 minutes of ischemia, renal damage was prevented by flushing with ice cold Sacks' solution. A preoperative plan to drain Sacks' solution from renal outflow by cannulating the renal vein was not possible because of sudden bleeding from the aneurysmal sac. Although the hazard of hyperkalemia was prevented by slow infusion of the solution and by electrocardiogram monitoring, ex situ flushing would have been the safe method in this case. The validity of hyperosmolar intracellular electrolyte solution in preserving renal function during an ambithermic condition has been well documented. 1 Although reversible acute renal failure developed in case 2 we verified previously the considerable renal protective effect of this solution in an am-
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Fm. 2. Case 2. A, film of kidneys and ureters shows completely calcified and several incompletely calcified rings at right renal fossa. B, abdominal aortography reveals multiple calcified and non-calcified renal artery aneurysms. C, aneurysms at hilar region were exposed during extracorporeal maneuver.
(1)
Fm. 3. Schematic drawing of aneurysmectomy and angioplasty in case 2 shows end-to-end anastomosis (J), side-to-end anastomosis (2), ovarian vein patch graft (3) covered by cuff of arterial stump (4) and pair-of-pants anastomosis (5).
bithermic condition by experimental and clinical studies, including 3 cases of ex situ operation. 3 ' 4 The combination of flushing Sacks' solution and surface cooling will be the method of choice if renal ischemia time exceeds 5 hours. Gynecological abnormalities associated with a congenital solitary kidney have been well documented. However, in addition to renal malformation of duplication, ectopia and cystic change, reports regarding an associated renal artery aneurysm are scanty, 5 In 1959 Mathe,6 and Harrow and Sloane7 reported
independently a case of a renal artery aneurysm in a solitary kidney. Since then 5 cases have been reported. 8 - 12 Although Mathe reported a case of a pelvic solitary kidney there have been no previous reports as our case 2, namely association of intracranial aneurysms, uterus bicornus and absent left iliac vein. The absence of the left iliac vein, which we could not anticipate preoperatively, resulted in a needless and time-consuming exploration of the left iliac fossa. Treatment of these 7 reported cases consisted of exploration
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7. Harrow, B. R. and Sloane, J. A.: Aneurysm of renal artery: report of five cases. J. Urol., 81: 35, 1959. 8. Ekestroem, S.: Surgical treatment of renal artery aneurysm. Acta Chir. Scand., 127: 149, 1964. 9. Hall, J. W., Factor, S. M. and Cerny, J. C.: Traumatic renal artery aneurysm in a solitary kidney. J. Urol., 107: 17, 1972. 10. Hodges, C. V., Lawson, R. K., Pearse, H. D. and Stranburg, C. 0.: Autotransplantation of the kidney. J. Urol., 110: 20, 1973. 'IL Belzer, F. 0., Salvatierra, 0., Palubinskas, A. and Stoney, R. J.: Ex vivo renal artery reconstruction. Ann. Surg., 182: 456, 1975. 12. Aubert, J. and Casamayou, J.: Anevrysme arteriel sur rein unique; chirurgie conservatrice. J. Urol. Nephrol., 84: 215, 1978. 13. Faidutti, B., Megevand, R., Humair, L., Zeltner, L., Simonet, F., Schneider, P., Fournial, G. and Hahn, C. H.: Anevrisme intrahilaire de l'artere renale sur rein unique. Reparation ex vivo et autotransplantation. Ann. Chir. Thorac. Cardiovasc., 13: 309, 1974. 14. Archimbaud, J.P., Calcat, P., Gelet, A., Gammondes, J.P., Vaiton, J., Banssillon, V. et G., Bouletrot, P., Petit, P. and Pinet, P.: Anevrisme geant de l'artere renale Sur rein unique. -Reparation par chirurgie extra corporeele suivie d'autotransplantation. Guerison Avec un an de Recul Chirurgie, 101: 408, 1975.__ . 15. Salvatierra, 0., Jr., Olcott, C., IV and Stoney, R. J.:Ex vivo renal artery reconstruction using perfusion preservation. J. Urol., 119: 16, 1978. EDITORIAL COMMENT
FIG. 4. Postoperative angiography in case 2 shows patent blood flow of autotransplanted kidney. in 2, in situ aneurysmectomy in 2, ligation of a polar branch in 1 and ex situ aneurysmectomy in 2. The first ex situ aneurysmectomy on a congenital solitary kidney was conducted in 1973 by Hodges and associates, using flushing as a preservation method. 10 However, the transplanted kidney lost its function owing to 12 hours of renal ischemia. Belzer and associates performed the first successful ex situ aneurysmectomy on a congenital solitary kidney using their own preservation machine.11 As for the solitary kidney after contralateral nephrectomy, Faidutti, 13 Archimbaud 14 and Salvatierra15 and their associates succeeded in ex situ aneurysmectomy, preserving the kidney either by the flushing method 13• 14 or the Belzer method. 15 REFERENCES 1. Sacks, S. A., Petritsch, P.H., Linder, R. and Kaufman, J. J.: Renal
2. 3.
4. 5. 6.
autotransplantation. Further use of a new perfusate. Amer. J. Surg., 128: 402, 1974. Poutasse, E. F.: Renal artery aneurysms. J. Urol., 113: 443, 1975. Hidai, H., Miyai, K., Saito, K., Kubota, Y. and Koshimizu, K.: Renal artery aneurysm treated by ex vivo surgery under ambithermic condition: report of a case and review of the literatures. Jap. J. Urol., 68: 425, 1977. Hidai, H.: Ex vivo operation. Jap. J. Urol., 69: 1161, 1978. Campbell, M. F.: Anomalies of the kidney. In: Urology, 3rd ed. Edited by M. F. Campbell and J. H. Harrison. Philadelphia: W. B. Saunders Co., p. 1418, 1970. Mathe, C. P.: Aneurysm of renal artery causing hypertension; report of three cases. J. Urol., 82: 412, 1959.
This article crosses several areas of my interest and writings: renovascular surgery, autotransplantation, renal preservation and operations on the solitary kidney. It is a good testimonial to the value of initial kidney flush and the clemency of ambiothermia vis-a-vis warm ischemia (body temperature). It also supports the concept shared by many urologists that the solitary kidney is privileged in that it will withstand ischemic insult better than the paired kidney. With regard to case 1, I wonder if the 140 ml. hyperosmolar intracellular electrolyte solution after a flush of the kidney with ice cold Ringer's lactate solution was of any value. The authors perfused the kidney in situ with Sacks' solution, which contains 126 mEq./1. potassium, and allowed the venous effluent to enter the systemic circulation. They did this slowly and monitored the electrocardiogram to avoid cardiac complications. The 140 ml. solution provides almost 20 mEq. potassium and I doubt that Sacks' solution, as it was used in this case, had any real effect on renal ischemic tolerance. Nonetheless, the good result obtained is commendable and the authors are to be congratulated for their resourcefulness. I am more critical of the management of case 2, since I believe that the extensive operation to remove all of the aneurysms in a 51-year-old woman with a solitary kidney was unwarranted. The largest of the 5 aneurysms required surgical treatment in my opinion but the other aneurysms did not constitute a threat, since they were relatively small and fairly well protected. Furthermore, the extensive ex vivo operation required 6 hours 15 minutes of renal ischemia and, despite the fact that the kidney was kept in an ambiothermic condition except for periodic flushing with Sacks' solution, the patient did, indeed, have postoperative anuria. Also, the hypertension was not corrected, perhaps because of the presence of the hypogastric artery stenosis, although this does not appear to be stenosed to a critical enough degree to cause hypertension. Furthermore, since the hypogastric artery was known to be narrow I would have preferred in situ surgery in this case, merely excising the large aneurysm and doing an aneurysmorrhaphy with or without a patch graft, or after removing the aneurysm to use a vein graft or monofilament knitted polypropylene graft from the aorta to the renal artery distal to the resected aneurysm. This procedure would have been safer and would have presented less risk to the patient. Joseph J. Kaufman Department of Surgery University of California School of Medicine Los Angeles, California REPLY BY AUTHORS As Doctor Kaufman has indicated case 1 was managed by infusion of 17.6 mEq. potassium into the systemic circulation, causing 1.6 mEq./ 1. serum potassium elevation estimated from the extracellular fluid volume, although actual serum potassium elevation was 1.0 mEq./1. (preoperative serum potassium 3.8 mEq./1., post-infusion 4.8 mEq./1.). Although no T wave change was recognized on the electrocardiogi 1m