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Ruptured Horseshoe (fused) Kidney: A Review and Report of a case with Traumatic Renal Hypertension
THE JOURNAL OF UROLOGY
Vol. 92, No. 5 November 1964 Copynght © 1964 by The Williams & Wilkins Co. Printed in U.S.A.
RUPTURED HORSESHOE (FUSED) KIDNEY: A REVIEW AND REPORT OF A · CASE WITH TRAUMATIC RENAL HYPERTENSION G. R. GIBSON* Horseshoe kidney is the result of fusion of the renal blastemas, between the fourth and eighth weeks of fetal life, before the usual migration and rotation have occurred. This produces the abnormal relation of the pelvis (anterior) to the vessels, and results in the ureters coursing across the isthmus anteriorly. The fusion is almost always of the lower poles, and lies across the body of the fourth lumbar vertebra. The incidence of horseshoe kidney is thought to be about 1 in 400 (Glenn1). Lowsley2 states that the diagnosis is most commonly made on pyelogrnphic investigation of some unrelated symptom, but nevertheless, horseshoe kidney often presents with secondary renal lesions. Hydronephrosis, infection and calculus formation are the rnost common, and are associated with the abnormal course of the ureters and their tendency to obstruction. A surgery rate of approximately 25 per cent was reported by Culp 3 and by Glenn. 1 The site of the isthmus lying over the most prominent part of the lordosis of the bony lumbar spine, and the medial and anterior displacement of the lower poles of the kidneys, would appear to render horseshoe kidneys peculiarly liable to injury. Despite this, and the prevalence of the anornaly, there is a dearth of recorded cases of injury. In 1925, Eisendrath4 reviewed 124 cases of horseshoe kidney then recorded, and found four with injury, two of which were diaO'nosed after death. The recent English literatu;e reveals only 1 case of trauma to a horseshoe kidney (Dowse and Kihn5). In the light of these considerations it is con~cc_epted for publication April 17, 1964. Ciba Research Fellow in Urology. 1 Glenn, J. F.: Analysis of 51 patients with horseshoe kidney. New Eng. J. Med 261· 684
1959.
.,
.
'
Lowsley, 0. S.: Surgery of the horseshoe kidney. J. Urol., 67: 565, 1952. 3 Culp, 0. S. and Winterringer, J. R.: Surgical treatment of horseshoe kidney. J. Urol 73· 747 • 2
1955.
.,
.
'
Eisendrath, D_. N., Phifer, F. M. and Culver, H. 5B: Horseshoe kidney. ~nn. Surg., 82: 735, 1925. . Do~se, J. L. and Kihn, R. B.: Renal injunes. Bnt. J. Surg., 50: 353, 1963. 4
sidered appropriate to report this case of ruptured horseshoe kidney. CASE REPORT
The patient was ag~d 36 when he sustained multiple injuries in a motor accident. After treatment of shock and fractures, he had heavy, though decreasing hematuria. Three weeks after the injury hypertension and oliguria developed and a mass appeared in the right side of the abdomen. Excretory urography showed a poorly functioning right kidney, and calyceal distortion in the left, which was attributed to a retroperitoneal hematoma. A retrograde catheter was ~locked at 18 cm. up the right ureter, and no urme passed through it. Transperitoneal exploration and drainage of a large retroperitoneal hematoma were then performed. The diagnosis of horseshoe kidney was made 2 months later by an aortogram (fig. 1, A) after persistent urinary drainage through the wound. This eventually ceased 16 months after the injury. The patient had sustained hypertension during this time, and another aortogram 27 months after injury showed a shrunken rio·ht hemi-kidney with no function and a diminished arterial supply (fig. 1, B). Operative rernoval of the fibrotic right segment of a horseshoe kidney was carried out a month after the second aortogram. The ureter was found to have been completely divided and ended blindly in dense scar tissue. The pathologist's report on the kidney stated: "The ureter is filled with thrombus and is completely obstructed by fibrous tissue at the ureteropelvic junction. There is marked hydronephrosis with a reddish lining to the pelvis and calyces, and a rin1 of about 0.5 cn1. of renal tissue· cortex and medulla cannot be distinguished'. Sections show severe chronic pyelonephritis, and also blood pigment and scarrino· indicatino· " "' past hemorrhage." The patient's blood pressure fell during the operation and has rernained around the level of 160/100 in the short followup period.
374
RUPTURED HORSESHOE (FUSED) KIDNEY
375
FIG. 1. Aortograms. A, May 16, 1961, shows horseshoe kidney. B, July 22, 1963. Note decrease in arterial supply to right segment.
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FIG. 2. Chart of recorded blood pressure DISCUSSION
The diagnosis of horseshoe kidney in this case was obscured, even to the surgeon approaching the right kidney transperitoneally, by the huge retroperitoneal hematoma which developed after the rupture. The diagnosis was eventually made by an aortogram 3 months after the injury. In
the next 2 years, the patient's health was impaired by the discharging urinary fistula with associated infection, and by sustained hypertension. It was this hypertension which prompted the subsequent surgery in 1963. Renal trauma has not often been incriminated as a cause of hypertension. Slade and colleagues 6 6 Slade, N., Evans, K. T. and Roylance, J_:
376
GIBSON
found no cases of hypertension in the followup study of 59 patients with renal trauma. Zimmerman and Radding7 presented a case of hypertension secondary to renal trauma in a 27-year-old woman, relieved by nephrectomy. Dowse and Kihn,5 in the followup evaluation of 67 cases of renal trauma, found 3 cases of hypertension which they attributed to the trauma. They suggested that the criteria necessary to link renal trauma with hypertension are 1) a pre-existing normotensive state, 2) progressive rise in blood pressure within a year of the trauma and 3) return to normotension after nephrectomy. In the present case, criteria 1 and 3 are not satisfied. The patient had several recorded blood pressure estimations before the accident, both of the order of 150/90, and the blood pressure has remained above normal levels since the right heminephrectomy (fig. 2). Nonetheless, this case is presented as one of renal hypertension The late results of closed renal injuries. Brit. J. Surg., 49: 194, 1961. 7 Zimmerman, S. J. and Radding, R. S.: Hypertension due to trauma of kidney. New Eng. J. Med., 264: 238, 1961.
secondary to trauma, because it exhibits 1) a known level of blood pressure before the injury (albeit mildly elevated), 2) a marked and sustained increase in blood pressure within a year of the injury and 3) a return to a level comparable to the original after nephrectomy (fig. 2). The mechanism of development of hypertension after renal trauma is presumed to be renal ischemia, produced by compression of renal tissue by the hematoma. In this case, there is also a progressive decrease in arterial supply to the injured right half of the kidney. This is clearly seen on the aortograms 2 years apart. SUMMARY
Some of the more recent literature covering horseshoe kidney and renal trauma has been reviewed, and the rarity of injury to a horseshoe kidney noted. A case of ruptured horseshoe kidney is presented, with the subsequent development of severe hypertension. It is considered that the hypertension was a consequence of the injury, and it was largely relieved by excision of the damaged half of the horseshoe kidney.
Vol. 92, No. 5 November 1964 Copynght © 1964 by The Williams & Wilkins Co. Printed in U.S.A.
RUPTURED HORSESHOE (FUSED) KIDNEY: A REVIEW AND REPORT OF A · CASE WITH TRAUMATIC RENAL HYPERTENSION G. R. GIBSON* Horseshoe kidney is the result of fusion of the renal blastemas, between the fourth and eighth weeks of fetal life, before the usual migration and rotation have occurred. This produces the abnormal relation of the pelvis (anterior) to the vessels, and results in the ureters coursing across the isthmus anteriorly. The fusion is almost always of the lower poles, and lies across the body of the fourth lumbar vertebra. The incidence of horseshoe kidney is thought to be about 1 in 400 (Glenn1). Lowsley2 states that the diagnosis is most commonly made on pyelogrnphic investigation of some unrelated symptom, but nevertheless, horseshoe kidney often presents with secondary renal lesions. Hydronephrosis, infection and calculus formation are the rnost common, and are associated with the abnormal course of the ureters and their tendency to obstruction. A surgery rate of approximately 25 per cent was reported by Culp 3 and by Glenn. 1 The site of the isthmus lying over the most prominent part of the lordosis of the bony lumbar spine, and the medial and anterior displacement of the lower poles of the kidneys, would appear to render horseshoe kidneys peculiarly liable to injury. Despite this, and the prevalence of the anornaly, there is a dearth of recorded cases of injury. In 1925, Eisendrath4 reviewed 124 cases of horseshoe kidney then recorded, and found four with injury, two of which were diaO'nosed after death. The recent English literatu;e reveals only 1 case of trauma to a horseshoe kidney (Dowse and Kihn5). In the light of these considerations it is con~cc_epted for publication April 17, 1964. Ciba Research Fellow in Urology. 1 Glenn, J. F.: Analysis of 51 patients with horseshoe kidney. New Eng. J. Med 261· 684
1959.
.,
.
'
Lowsley, 0. S.: Surgery of the horseshoe kidney. J. Urol., 67: 565, 1952. 3 Culp, 0. S. and Winterringer, J. R.: Surgical treatment of horseshoe kidney. J. Urol 73· 747 • 2
1955.
.,
.
'
Eisendrath, D_. N., Phifer, F. M. and Culver, H. 5B: Horseshoe kidney. ~nn. Surg., 82: 735, 1925. . Do~se, J. L. and Kihn, R. B.: Renal injunes. Bnt. J. Surg., 50: 353, 1963. 4
sidered appropriate to report this case of ruptured horseshoe kidney. CASE REPORT
The patient was ag~d 36 when he sustained multiple injuries in a motor accident. After treatment of shock and fractures, he had heavy, though decreasing hematuria. Three weeks after the injury hypertension and oliguria developed and a mass appeared in the right side of the abdomen. Excretory urography showed a poorly functioning right kidney, and calyceal distortion in the left, which was attributed to a retroperitoneal hematoma. A retrograde catheter was ~locked at 18 cm. up the right ureter, and no urme passed through it. Transperitoneal exploration and drainage of a large retroperitoneal hematoma were then performed. The diagnosis of horseshoe kidney was made 2 months later by an aortogram (fig. 1, A) after persistent urinary drainage through the wound. This eventually ceased 16 months after the injury. The patient had sustained hypertension during this time, and another aortogram 27 months after injury showed a shrunken rio·ht hemi-kidney with no function and a diminished arterial supply (fig. 1, B). Operative rernoval of the fibrotic right segment of a horseshoe kidney was carried out a month after the second aortogram. The ureter was found to have been completely divided and ended blindly in dense scar tissue. The pathologist's report on the kidney stated: "The ureter is filled with thrombus and is completely obstructed by fibrous tissue at the ureteropelvic junction. There is marked hydronephrosis with a reddish lining to the pelvis and calyces, and a rin1 of about 0.5 cn1. of renal tissue· cortex and medulla cannot be distinguished'. Sections show severe chronic pyelonephritis, and also blood pigment and scarrino· indicatino· " "' past hemorrhage." The patient's blood pressure fell during the operation and has rernained around the level of 160/100 in the short followup period.
374
RUPTURED HORSESHOE (FUSED) KIDNEY
375
FIG. 1. Aortograms. A, May 16, 1961, shows horseshoe kidney. B, July 22, 1963. Note decrease in arterial supply to right segment.
300
I
I:
250
200
150
120 100
BO 70 60 50
,o 30
~~ ~~ ~le
>-lo
:::>N "'-
1
:-: ~;:: lg;;
~a
~~
Q.N
9
~
[
N
FIG. 2. Chart of recorded blood pressure DISCUSSION
The diagnosis of horseshoe kidney in this case was obscured, even to the surgeon approaching the right kidney transperitoneally, by the huge retroperitoneal hematoma which developed after the rupture. The diagnosis was eventually made by an aortogram 3 months after the injury. In
the next 2 years, the patient's health was impaired by the discharging urinary fistula with associated infection, and by sustained hypertension. It was this hypertension which prompted the subsequent surgery in 1963. Renal trauma has not often been incriminated as a cause of hypertension. Slade and colleagues 6 6 Slade, N., Evans, K. T. and Roylance, J_:
376
GIBSON
found no cases of hypertension in the followup study of 59 patients with renal trauma. Zimmerman and Radding7 presented a case of hypertension secondary to renal trauma in a 27-year-old woman, relieved by nephrectomy. Dowse and Kihn,5 in the followup evaluation of 67 cases of renal trauma, found 3 cases of hypertension which they attributed to the trauma. They suggested that the criteria necessary to link renal trauma with hypertension are 1) a pre-existing normotensive state, 2) progressive rise in blood pressure within a year of the trauma and 3) return to normotension after nephrectomy. In the present case, criteria 1 and 3 are not satisfied. The patient had several recorded blood pressure estimations before the accident, both of the order of 150/90, and the blood pressure has remained above normal levels since the right heminephrectomy (fig. 2). Nonetheless, this case is presented as one of renal hypertension The late results of closed renal injuries. Brit. J. Surg., 49: 194, 1961. 7 Zimmerman, S. J. and Radding, R. S.: Hypertension due to trauma of kidney. New Eng. J. Med., 264: 238, 1961.
secondary to trauma, because it exhibits 1) a known level of blood pressure before the injury (albeit mildly elevated), 2) a marked and sustained increase in blood pressure within a year of the injury and 3) a return to a level comparable to the original after nephrectomy (fig. 2). The mechanism of development of hypertension after renal trauma is presumed to be renal ischemia, produced by compression of renal tissue by the hematoma. In this case, there is also a progressive decrease in arterial supply to the injured right half of the kidney. This is clearly seen on the aortograms 2 years apart. SUMMARY
Some of the more recent literature covering horseshoe kidney and renal trauma has been reviewed, and the rarity of injury to a horseshoe kidney noted. A case of ruptured horseshoe kidney is presented, with the subsequent development of severe hypertension. It is considered that the hypertension was a consequence of the injury, and it was largely relieved by excision of the damaged half of the horseshoe kidney.