COMPLICATIONS
OF HORSESHOE
J. E. CASTRO,
M.S., F.R.C.S.
N. A. GREEN,
M.S.,
KIDNEY
F.R.C.S.
From the Urological Units, United Norwich Hospitals, Norwich, and Hammer-smith Hospital, London, England
ABSTRACT -A case of horseshoe kidney complicated by hydronephrosis, renal calculi, urinary infection, and tumor of the renal pelvis is reported. The incidence and etiology of the complications are discussed and the frequency of tumors in horseshoe kidneys described.
This case demonstrates four complications associated with horseshoe kidney, namely, hydronephrosis, renal calculi, urinary infection, and tumor of the renal pelvis, It is of particular interest since the occurrence of tumor in a horseshoe kidney is unusual. Eighty-three cases have been described previously, 20 with carcinoma of the renal pelvis. Case Report The patient, a married female, eighty years of age, was seen in 1965 because of two episodes of hematuria and dysuria. Her general health was good, and she had no loin pain, increased micturition, or febrile episodes. Physical examination was unremarkable, and midstream urine at this time was normal to microscopy and culture. An excretion pyelogram showed a horseshoe kidney with hydronephrosis and stones in its left moiety. Cystoscopy under local anesthesia revealed a clean bladder with no tumor, diverticulum, or stone, and the efflux from both kidneys was clear. Further investigations were not undertaken because of her age and minimal symptoms. In 1966 she suffered an epistaxis which required hospital admission, and during the following three years she had occasional hematuria associated with bacteriologically proved urinary infection treated successfully by her physician. In November, 1969, she was admitted to the hospital with a history of persistent hematuria, left loin pain, and difficulty in voiding for three weeks. On examination tenderness was evident in
344
the suprapubic region and the loin. Her blood pressure was 180/110 mm. Hg. Urine was grossly bloodstained and microscopic examination showed 3 white blood cells and 250 red blood cells per high-power field, while culture yielded a scanty growth of Proteus. Hemoglobin was 9.2 Gm. with 100 ml. and the white blood count 9,300 per cubic millimeter, mean hemoglobin concentration was 29 per cent, and a stained blood film showed the changes of iron deficiency anemia. Blood urea was 64 mg. per 100 ml. and serum electrolytes normal. Measurements of serum calcium, phosphate, and uric acid were within the normal ranges. Investigations showed no hematologic cause for the bleeding. Preoperative roentgenogram of the chest was within normal limits, and electrocardiograph showed only minimal changes of ischemic heart disease. Excretion urography showed a horseshoe kidney with hydronephrosis of its left pelvis (Fig. 1A) and calculi in this pelvis (Fig. 1B). Examination under anesthesia showed a partial urethral prolapse, but no abdominal masses were felt. Cystoscopic examination revealed coarse bladder trabeculation with a diverticulum in the right lower quadrant; a bloody efflux was coming from the left ureteric orifice. A left ureteropyelogram confirmed the hydronephrosis and showed a filling defect suggestive of a tumor of the left renal pelvis (Fig. 1C). During the ensuing week heavy hematuria persisted which required repeated transfusion and
UROLOGY
/
SEPTEMBER
1975
I
VOLUME
VI, NUMBER3
j
’
-
.
’
FIGURE 1. (A) Excretion urogram showing horseshoe kidney with hydronephrosis of its leftpelvis. (B) Plain abdominal x-ray film showing calculi in left pelvis of horseshoe kidney. (C) L.eft retrograde ureteropyelogram showing hydronephrosis and filling defect in renal pelvis.
necessitated left heminephrectomy. This was performed through a left loin incision and a combined intra- and extraperitoneal approach. The isthmus was divided and closed with interrupted catgut sutures; no calyx was seen. The ureter was divided outside the bladder, and because of the patient’s age the intramural part of the ureter was not removed. No metastases were seen.
UROLOGY
/ SEPTEMBER1975 / VOLUMEVI, NUMBER3
The patient’s postoperative course was unremarkable for six weeks except for a superficial wound infection which subsided with local treatment. A month after operation her blood urea was 56 mg. per 100 ml., and urine examined microscopically showed no erythrocytes or malignant cells. However, six weeks postoperatively mental confusion supervened, progress was slow, and she died of bronchopneumonia. The excised portion of the kidney (Fig. 2) showed extensive replacement by yellow tumor which filled most of the pelvis and replaced most of the renal tissue. Histologic examination (Fig. 3) showed a poorly differentiated papillary transitional cell carcinoma; the tumor had extensively invaded and destroyed the renal tissue. Examination of the ureter showed only the changes of chronic inflammation. At autopsy there was no residual or metastatic tumor.
345
FIGURE 2. (A and B) L,eft heminephrectomy specimen showing large renal tumor.
Comment The incidence of horseshoe kidney depends on the methods of selecting patients, and it varies from 1 in 200 to 1 in 1,000 of the population. Dajani’ found 1 in 265, Lowsley2 1 in 284, and Davidson (quoted by Lowsley2) 1 in 1,000. These abnormal kidneys are subject to more disease
FIGURE 3. Histology of tumor showing transitional epithelium (hematoxylin and eosin).
346
than normal, but the need for treatment varies from 25 per cent of cases in Dajani’s series to 75 per cent in Kilpatrick’s.3 The case described demonstrates most of the common complications of horseshoe kidney: hydronephrosis, stone formation, infection, and tumor. Hydronephrosis and hydroureter are the most common complications and occurred in 11 of the 20 cases described by KilpatrickV3 The increased incidence of abnormal vessels and pressure on abnormally situated ureters have been held responsible. Calculi form in association with hydronephrosis because of the combination of infection and stasis.4 In 2 of the 20 cases calculi were found within a hydronephrotic horseshoe kidney.3 The first case of malignant tumor in a horseshoe kidney was described in I895,5 and a review of the world literature to the end of 1966 revealed 71 cases.6 These consisted of 34 (47 per cent) adenocarcinomas, 20 (28 per cent) carcinomas of the renal pelvis, 14 (20 per cent) nephroblastomas, and 3 (4 per cent) miscellaneous sarcomas. Further review of the literature to the present shows 13 more cases including the present one (Table I). The patient described herein is the oldest on record, the previous age range for adenocarcinoma being twenty-five to seventyone years and for tumors of the renal pelvis four to seventy-two years. Blackard and Mellinger’ have shown that adenocarcinoma does not occur more frequently in horseshoe kidneys than in normal kidneys; but in contrast there is a higher incidence of carcinoma of the renal pelvis, and this may be related to the more common complications of infection, stasis, and stone. Seventeen of the growths of the renal pelvis were transitional
UROLOGY
I
SEPTEMBER
1975
/
VOLUME
VI, NUMBER
3
TABLE I.
Number Described
Histologic Type .
71
1
1 1 1 1 1 1 1 1 1 1
1 1
.
Incidence
of tumor in horseshoe kidney
Sex
Authors
...
Adenocarcinoma Adenocarcinoma Adenocarcinoma Nephroblastoma Sarcoma Nephrobiastoma
F F M M M M
Adenocarcinoma Adenocarcinoma Adenocarcinoma Adenocarcinoma Adenocarcinoma Adenocarcinoma Renal pelvis
M M M F M M F
-
cell tumors and four squamous. The latter type of tumor frequently occurs when renal calculi are present,’ but in our case renal calculi were associated with a transitional cell tumor. The common factor implicated in their genesis could be urine stasis caused by the hydronephrosis, for calculus formation is the known complication of stasis, and it has been suggested that papillary tumor is similarly related. In our case hydronephrosis was present for at least three years and had not changed significantly while the calculi increased in size and number and the renal tumor had become apparent. Heminephrectomy, as first described by Sorcin (quoted by Lowsley2) in 1883, was performed in this case, using an oblique incision from the tip of the twelfth rib.3 A combined extraperitoneal (Lowsley, 19522) and intraperitoneal approach allowing easy mobilization of the isthmus and laparotomy to exclude distant metastases was utilized. 8 Hammersmith Hospital London W 12, England (DR. CASTRO) ACKNOWLEDGMENT. Thanks to Dr. H. de C. Baker for pathologic assistance and to Mr. Bum for the photography. References 1. DAJANI, A. M.: Horseshoe kidney: a review of 29 cases, Br. J. Ural. 38: 388 (1966). 2. LOWSLEY, J.: Surgery of horseshoe kidney, J. Urol. 67: 565 (1952). 3. KILPATRICK, F. R.: Horseshoe kidneys, Proc. R. Sot. Med. 66: 433 (1967).
UROLOGY
/
SEPTEMBER 1975 /
VOLUME VI. NUMBER 3
Blackard and Mellinger’ Kato, Ishibe, Shiraishi, and Mizoguchi9 Vlasak and Jiran” Dajani’ KrafR” Fazio, Steinberg, and Golombek” Montagnini, Mancia, Crisci, and Emanuelli13 Viville, Katzner, and Gillet14 Schiappapietra15 Czvalinga” Alwasiak and Hajdukiewicz” Itzig, Perry, and McCaughan” Redman” Castro and Green
Year 1968 1966 1966 1968 1968 1968 1969 1969 1969 1969 1971 1972 1973 1974
4. GAMBETTA, G. : Considerations sur la lithiase du rein en fer a cheval, J. Urol. Nephrol. (Paris) 72: 745 (1966). 5. HILDEBRAND, 0.: Beitzag zur Nieren, Deutsche Zschr. Chir. (Leipzig) 40: 96 (1894). 6. BLACKARD, C. E., and MELLINGER, G. T. : Cancer in a horseshoe kidney: a report of two cases, Arch. Surg. 97: 216 (1968). 7. OBERKIRCHER, 0. J., STAUBITZ, W. J., and BLICK, M. S.: Squamous cell carcinoma of renal pelvis, J. Ural. 66: 551 (1951). 8. DAHLEN, C. P., and SCHLUMBERGER, F. C.: Surgery of the diseased horseshoe kidney, Am. J. Surg. 93: 405 (1957). 9. KATO, T., ISHIBE, T., SHIRAISHI, T., and MIZOGUCHI, M. : A case of Grawitz’s tumour originating in a horseshoe kidney, Acta Urol. (Kyoto) 12: 285 (1966). 10. VLASAK, V., and JIRAN, B. : Karcinom v podkovite ledvine, Rozhl. Chir. 45: 323 (1966). 11. KRAFFT, L.: Wilms’ Tumour in Hufeisenniere, Zentralbl. Chir. 93: 1583 (1968). 12. FAZIO, J. M., STEINBERG, S., and GOLOMBEK, M.: Sarcoma litiasis coraliforme en rition en herradura, Rev. Argentina Ural. Nefrol. 37: 222 (1966). 13. MONTAGNINI, R., MANCIA, A., CRISCI, E., and EMANUELLI, M.: Tumore de Wilms su rene a fero di cavallo, Policlinico (Prat.) 76: 305 (1969). 14. VIVILLE, C., KATZNER, M., and GILLET, M.: Tumeur de Grawitz sur rein en fer a cheval, J, Urol. Nephrol. (Paris) 75: 481 (1969). 15. SCHIAPPAPIETRA, J. : Tumour de rinon herradura, Rev. Argentina Urol. Nefrol. 38: 186 (1969). 16. CZVALINGA, I. : A patkovese daganatairol, ON. Hetil. 41: 2383 (1969). 17. ALWASIAK, J., and HAJDUKIEWICZ, Z.: A case of clarocellular carcinoma of the horseshoe kidney, Pol. Przegl. Chir. 43: 1045 (1971). 18. ITZIG, C. B., PERRY, S. R., and MCCAUGHAN, J. J. : Adenocarcinoma in the isthmus of a horseshoe kidney, Am. Surg. 38: 422 (1972). 19. REDMAN, J. F.: Renal cell carcinoma in a horseshoe kidney, South. Med. J. 66: 963 (1973).
347
OF HORSESHOE
J. E. CASTRO,
M.S., F.R.C.S.
N. A. GREEN,
M.S.,
KIDNEY
F.R.C.S.
From the Urological Units, United Norwich Hospitals, Norwich, and Hammer-smith Hospital, London, England
ABSTRACT -A case of horseshoe kidney complicated by hydronephrosis, renal calculi, urinary infection, and tumor of the renal pelvis is reported. The incidence and etiology of the complications are discussed and the frequency of tumors in horseshoe kidneys described.
This case demonstrates four complications associated with horseshoe kidney, namely, hydronephrosis, renal calculi, urinary infection, and tumor of the renal pelvis, It is of particular interest since the occurrence of tumor in a horseshoe kidney is unusual. Eighty-three cases have been described previously, 20 with carcinoma of the renal pelvis. Case Report The patient, a married female, eighty years of age, was seen in 1965 because of two episodes of hematuria and dysuria. Her general health was good, and she had no loin pain, increased micturition, or febrile episodes. Physical examination was unremarkable, and midstream urine at this time was normal to microscopy and culture. An excretion pyelogram showed a horseshoe kidney with hydronephrosis and stones in its left moiety. Cystoscopy under local anesthesia revealed a clean bladder with no tumor, diverticulum, or stone, and the efflux from both kidneys was clear. Further investigations were not undertaken because of her age and minimal symptoms. In 1966 she suffered an epistaxis which required hospital admission, and during the following three years she had occasional hematuria associated with bacteriologically proved urinary infection treated successfully by her physician. In November, 1969, she was admitted to the hospital with a history of persistent hematuria, left loin pain, and difficulty in voiding for three weeks. On examination tenderness was evident in
344
the suprapubic region and the loin. Her blood pressure was 180/110 mm. Hg. Urine was grossly bloodstained and microscopic examination showed 3 white blood cells and 250 red blood cells per high-power field, while culture yielded a scanty growth of Proteus. Hemoglobin was 9.2 Gm. with 100 ml. and the white blood count 9,300 per cubic millimeter, mean hemoglobin concentration was 29 per cent, and a stained blood film showed the changes of iron deficiency anemia. Blood urea was 64 mg. per 100 ml. and serum electrolytes normal. Measurements of serum calcium, phosphate, and uric acid were within the normal ranges. Investigations showed no hematologic cause for the bleeding. Preoperative roentgenogram of the chest was within normal limits, and electrocardiograph showed only minimal changes of ischemic heart disease. Excretion urography showed a horseshoe kidney with hydronephrosis of its left pelvis (Fig. 1A) and calculi in this pelvis (Fig. 1B). Examination under anesthesia showed a partial urethral prolapse, but no abdominal masses were felt. Cystoscopic examination revealed coarse bladder trabeculation with a diverticulum in the right lower quadrant; a bloody efflux was coming from the left ureteric orifice. A left ureteropyelogram confirmed the hydronephrosis and showed a filling defect suggestive of a tumor of the left renal pelvis (Fig. 1C). During the ensuing week heavy hematuria persisted which required repeated transfusion and
UROLOGY
/
SEPTEMBER
1975
I
VOLUME
VI, NUMBER3
j
’
-
.
’
FIGURE 1. (A) Excretion urogram showing horseshoe kidney with hydronephrosis of its leftpelvis. (B) Plain abdominal x-ray film showing calculi in left pelvis of horseshoe kidney. (C) L.eft retrograde ureteropyelogram showing hydronephrosis and filling defect in renal pelvis.
necessitated left heminephrectomy. This was performed through a left loin incision and a combined intra- and extraperitoneal approach. The isthmus was divided and closed with interrupted catgut sutures; no calyx was seen. The ureter was divided outside the bladder, and because of the patient’s age the intramural part of the ureter was not removed. No metastases were seen.
UROLOGY
/ SEPTEMBER1975 / VOLUMEVI, NUMBER3
The patient’s postoperative course was unremarkable for six weeks except for a superficial wound infection which subsided with local treatment. A month after operation her blood urea was 56 mg. per 100 ml., and urine examined microscopically showed no erythrocytes or malignant cells. However, six weeks postoperatively mental confusion supervened, progress was slow, and she died of bronchopneumonia. The excised portion of the kidney (Fig. 2) showed extensive replacement by yellow tumor which filled most of the pelvis and replaced most of the renal tissue. Histologic examination (Fig. 3) showed a poorly differentiated papillary transitional cell carcinoma; the tumor had extensively invaded and destroyed the renal tissue. Examination of the ureter showed only the changes of chronic inflammation. At autopsy there was no residual or metastatic tumor.
345
FIGURE 2. (A and B) L,eft heminephrectomy specimen showing large renal tumor.
Comment The incidence of horseshoe kidney depends on the methods of selecting patients, and it varies from 1 in 200 to 1 in 1,000 of the population. Dajani’ found 1 in 265, Lowsley2 1 in 284, and Davidson (quoted by Lowsley2) 1 in 1,000. These abnormal kidneys are subject to more disease
FIGURE 3. Histology of tumor showing transitional epithelium (hematoxylin and eosin).
346
than normal, but the need for treatment varies from 25 per cent of cases in Dajani’s series to 75 per cent in Kilpatrick’s.3 The case described demonstrates most of the common complications of horseshoe kidney: hydronephrosis, stone formation, infection, and tumor. Hydronephrosis and hydroureter are the most common complications and occurred in 11 of the 20 cases described by KilpatrickV3 The increased incidence of abnormal vessels and pressure on abnormally situated ureters have been held responsible. Calculi form in association with hydronephrosis because of the combination of infection and stasis.4 In 2 of the 20 cases calculi were found within a hydronephrotic horseshoe kidney.3 The first case of malignant tumor in a horseshoe kidney was described in I895,5 and a review of the world literature to the end of 1966 revealed 71 cases.6 These consisted of 34 (47 per cent) adenocarcinomas, 20 (28 per cent) carcinomas of the renal pelvis, 14 (20 per cent) nephroblastomas, and 3 (4 per cent) miscellaneous sarcomas. Further review of the literature to the present shows 13 more cases including the present one (Table I). The patient described herein is the oldest on record, the previous age range for adenocarcinoma being twenty-five to seventyone years and for tumors of the renal pelvis four to seventy-two years. Blackard and Mellinger’ have shown that adenocarcinoma does not occur more frequently in horseshoe kidneys than in normal kidneys; but in contrast there is a higher incidence of carcinoma of the renal pelvis, and this may be related to the more common complications of infection, stasis, and stone. Seventeen of the growths of the renal pelvis were transitional
UROLOGY
I
SEPTEMBER
1975
/
VOLUME
VI, NUMBER
3
TABLE I.
Number Described
Histologic Type .
71
1
1 1 1 1 1 1 1 1 1 1
1 1
.
Incidence
of tumor in horseshoe kidney
Sex
Authors
...
Adenocarcinoma Adenocarcinoma Adenocarcinoma Nephroblastoma Sarcoma Nephrobiastoma
F F M M M M
Adenocarcinoma Adenocarcinoma Adenocarcinoma Adenocarcinoma Adenocarcinoma Adenocarcinoma Renal pelvis
M M M F M M F
-
cell tumors and four squamous. The latter type of tumor frequently occurs when renal calculi are present,’ but in our case renal calculi were associated with a transitional cell tumor. The common factor implicated in their genesis could be urine stasis caused by the hydronephrosis, for calculus formation is the known complication of stasis, and it has been suggested that papillary tumor is similarly related. In our case hydronephrosis was present for at least three years and had not changed significantly while the calculi increased in size and number and the renal tumor had become apparent. Heminephrectomy, as first described by Sorcin (quoted by Lowsley2) in 1883, was performed in this case, using an oblique incision from the tip of the twelfth rib.3 A combined extraperitoneal (Lowsley, 19522) and intraperitoneal approach allowing easy mobilization of the isthmus and laparotomy to exclude distant metastases was utilized. 8 Hammersmith Hospital London W 12, England (DR. CASTRO) ACKNOWLEDGMENT. Thanks to Dr. H. de C. Baker for pathologic assistance and to Mr. Bum for the photography. References 1. DAJANI, A. M.: Horseshoe kidney: a review of 29 cases, Br. J. Ural. 38: 388 (1966). 2. LOWSLEY, J.: Surgery of horseshoe kidney, J. Urol. 67: 565 (1952). 3. KILPATRICK, F. R.: Horseshoe kidneys, Proc. R. Sot. Med. 66: 433 (1967).
UROLOGY
/
SEPTEMBER 1975 /
VOLUME VI. NUMBER 3
Blackard and Mellinger’ Kato, Ishibe, Shiraishi, and Mizoguchi9 Vlasak and Jiran” Dajani’ KrafR” Fazio, Steinberg, and Golombek” Montagnini, Mancia, Crisci, and Emanuelli13 Viville, Katzner, and Gillet14 Schiappapietra15 Czvalinga” Alwasiak and Hajdukiewicz” Itzig, Perry, and McCaughan” Redman” Castro and Green
Year 1968 1966 1966 1968 1968 1968 1969 1969 1969 1969 1971 1972 1973 1974
4. GAMBETTA, G. : Considerations sur la lithiase du rein en fer a cheval, J. Urol. Nephrol. (Paris) 72: 745 (1966). 5. HILDEBRAND, 0.: Beitzag zur Nieren, Deutsche Zschr. Chir. (Leipzig) 40: 96 (1894). 6. BLACKARD, C. E., and MELLINGER, G. T. : Cancer in a horseshoe kidney: a report of two cases, Arch. Surg. 97: 216 (1968). 7. OBERKIRCHER, 0. J., STAUBITZ, W. J., and BLICK, M. S.: Squamous cell carcinoma of renal pelvis, J. Ural. 66: 551 (1951). 8. DAHLEN, C. P., and SCHLUMBERGER, F. C.: Surgery of the diseased horseshoe kidney, Am. J. Surg. 93: 405 (1957). 9. KATO, T., ISHIBE, T., SHIRAISHI, T., and MIZOGUCHI, M. : A case of Grawitz’s tumour originating in a horseshoe kidney, Acta Urol. (Kyoto) 12: 285 (1966). 10. VLASAK, V., and JIRAN, B. : Karcinom v podkovite ledvine, Rozhl. Chir. 45: 323 (1966). 11. KRAFFT, L.: Wilms’ Tumour in Hufeisenniere, Zentralbl. Chir. 93: 1583 (1968). 12. FAZIO, J. M., STEINBERG, S., and GOLOMBEK, M.: Sarcoma litiasis coraliforme en rition en herradura, Rev. Argentina Ural. Nefrol. 37: 222 (1966). 13. MONTAGNINI, R., MANCIA, A., CRISCI, E., and EMANUELLI, M.: Tumore de Wilms su rene a fero di cavallo, Policlinico (Prat.) 76: 305 (1969). 14. VIVILLE, C., KATZNER, M., and GILLET, M.: Tumeur de Grawitz sur rein en fer a cheval, J, Urol. Nephrol. (Paris) 75: 481 (1969). 15. SCHIAPPAPIETRA, J. : Tumour de rinon herradura, Rev. Argentina Urol. Nefrol. 38: 186 (1969). 16. CZVALINGA, I. : A patkovese daganatairol, ON. Hetil. 41: 2383 (1969). 17. ALWASIAK, J., and HAJDUKIEWICZ, Z.: A case of clarocellular carcinoma of the horseshoe kidney, Pol. Przegl. Chir. 43: 1045 (1971). 18. ITZIG, C. B., PERRY, S. R., and MCCAUGHAN, J. J. : Adenocarcinoma in the isthmus of a horseshoe kidney, Am. Surg. 38: 422 (1972). 19. REDMAN, J. F.: Renal cell carcinoma in a horseshoe kidney, South. Med. J. 66: 963 (1973).
347
