CASE REPORTS
MALIGNANCY HORSESHOE
ASSOCIATED
WITH
KIDNEY*
MAJ. DAVID BUNTLEY,
(MC) USA
From the Urology Service, Fitzsimons Medical Cetiter, Denver, Colorado
Army
i \/IS7’Rr,(; 1 - A compilation of the reported cases of cancer in horseshoe kidney from 1895 to 1975 is 1~-cw:n~c rl &ng with a comparison of the incidence of the various types of tumor involved from 1895 to 1963 a’n~;1%6 to 1975. An additional case of hypernephroma is added to the literature for a total of 111 ( ~LSC:; rq lor-tl:dto date. The increased incidence of both carcinoma of the renal pelvis and nephroblastoBOIL is di mused.
‘rEe firs : and most comprehensive review of the *~~rlcL literature regarding the association of renal :letrplasln and horseshoe kidney was that published 1~: Blackard and Mellinger.’ They reported ; t tot,al of 72 cases with the histologic breakdown as 5eer1 in ‘Table I. Dajani2 added 1 case of hyper:lephrorla not included in the Blackard series. In 1969 Cc Ilichaev, Pevzner, and Ivanov3 supplied XI additional case of hypernephroma. U’right4 reviewed the literature from 1966 to 1968 an11 discovered 9 additional cases of hyper:lepbrorla, 2 of Wilms’ tumor and 2 of sarcoma. We added 1 case of hypernephroma. In 1971Ostropolskaya” reported 4 cases of Wilms’ tumor occurring in 38 children with horseshoe kidney. ,Slso during the same year Alwasiak’ contributed I case oj‘hypernephroma discovered at necropsy ;md Esuritert’ reported 2 cases ofhypernephroma. The following year KGllin et al. 8 reported 1 case each of hypernephroma, Wilms’ tumor, and lymphoma with renal involvement. Kotowiczg added 1 case of sarcoma and Segura, Kelalis, and BurkelO reported a single case of lymphoblastic lymphoma. In addition, Itzig, Perry, and McCaughan” reported 1 case ofhypernephroma. Redman and Mobley12 reviewed the literature in 1973 and added 1 case of hypernephroma (Bur-
*The opinions or assertions contained herein are the private vic:ws of the author and are not to be construed as official or as reflecting the views of the Department of the Army or the Department of Defense.
146
kovskii) not reported previously and included an additional case. Kumar, Kumar, and Bhat13 contributed 1 case of hypernephroma in 1974 followed by a review by Shashikumar et al. l4 who added 1 case of Wilms’ tumor not reported previously (Bhatnagar) plus 3 additional cases of Wilms’ tumor. During the same year Choovichian et al. l5 contributed 1 case of transitional cell carcinoma of the renal pelvis. An additional case of hypernephroma in a horseshoe kidney is reported herein. Case Report The patient is a sixty-four-year-old Caucasian male in whom microhematuria with dysuria developed approximately forty-eight hours after a right inguinal hernia repair during November, 1972, at Reynolds Army Hospital, Ft. Sill, Oklahoma. A urologic workup was initiated. Intravenous pyelogram revealed a mass in the left upper pole of a previously undiagnosed horseshoe kidney (Fig. 1A). Nephrotomography and arteriography showed the lesion to be a noncystic, relatively avascular mass (Fig. 1B). Diagnostic puncture of the lesion was not attempted. Findings on chest x-ray film and laboratory values were all within normal limits with the exception of an elevated serum alkaline phosphatase attributed to the patient’s Paget’s disease. On January 24, 1973, the patient was explored through an abdominal transperitoneal approach.
UROLOGY
/ AUGUST
1976/
VOLUME
VIII,
NUMBER
2
review. He concluded that “probably cancer is no more likely to occur in a horseshoe kidney than it is in a normal kidney. “l However, he stated that there was a higher incidence of renal pelvis carcinoma and nephroblastoma. Choovichian and coworkerG also concluded that the incidence of carcinoma which predispose the horseshoe kidney to infection and hydronephrosis, namely, urinary stasis, predispose the renal collecting system to prolonged contact with carcinogenic components of the excreted urine.
There was a firm mass 2.5 cm. in diameter confined to the left upper pole. Left heminephrectomy was accomplished by clamping the vessels supplying the left half of the kidney. Division of a fibrous isthmus was performed at the line of ischemic demarcation. Horizontal mattress sutures were tied over adipose tissue to achieve hemostasis. Cross section of the kidney showed a 2.5-cm. pale yellow tumor, well circumscribed, with focal areas of hemorrhage. There was no grossly apparent involvement of the collecting system or the vessels. Microscopic examination revealed renal cell carcinoma with small neoplastic cells containing rounded hyperchromatic nuclei. The cytoplasm varied from eosinophilic to clear. No vascular invasion was identified (Fig. 2). The postoperative course was marked by a 10 per cent drop in the hematocrit which stabilized with conservative management and transfusion of blood. The patient had no further complications and is alive and well without evidence of recurrence two years after surgery. Comment A total of 111 cases of cancer in a horseshoe kidney have been reported in the world literature since 1895 from which date Blackard initiated his
UROLOGY
/ AUGUST 1976 / VOLUME
VIII,
NUMBER
showing relatively FIGURE 2. Photomicrograph clear cytoplasm with hyperchromutic nuclei of hypernephroma.
2
147
TABLE I.
of incidence of types of carcinoma horseshoe kidney
Comparison
1966
to 1975
No.
of CusesI Percent
r--
I
20
I
Wllm s Tumor I------
1
14
1 20
Renal Pelvis Carcinoma
TOTAL
72
A comparison of the percentages of occurrence of the four main categories of cancer reviewed shows a marked similarity between the Blackard and Mellinger figures with those of this review (Table I). However, an analysis of the 39 cases reviewed since 1966 reveals a significant decrease in occurrence of Wilms’ tumor from 28 to 12.8 per cent. The figure more closely approximates that quoted by Lucke and SchlumbergeP of 5.6 per cent incidence of Wilms’ tumor in 1,659 malignant renal tumors. This figure is halfthe observed incidence seen in horseshoe kidney. The fact that there is an increased incidence of both carcinoma of the renal pelvis and nephroblastoma in a horseshoe kidney should alert the urologist to search for these entities in patients found to have this anomaly. The pediatric patient with a horseshoe kidney should be evaluated and followed adequately not only to discover deteriorating renal disease secondary to the recognized complications of obstruction and infection, but also to interpret early any malignant degeneration. Denver, Colorado 80240 ACKNOWLEDGMENT. To John W. Weigel, M.D., Chief of Urology, Fitzsimons Army Medical Center for editorial assistance. References 1. BLACKARD, C. E., and MELLINGER, T. T. : Cancer in a horsehoe kidney, Arch. Surg. 97: 616 (1968). 2. DAJANI, M. B.: Horseshoe kidney: a review oftwentynine cases, Br. J. Ural. 38: 388 (1966).
148
1895
in
to (975
No. of Cases I PwCent
28
5.1
2
I
occurring
I
.9
2
I.8
III
39
3. GOLICHAEV, Z. K., PEVZNER, P., and IVANOV, A. V.: Tumor ofhorseshoe kidney, Urol. Nefrol. 34: 57 (1969). 4. WRIGHT, R.: A case of carcinoma in a horseshoe kidney and review of the literature, Proc. Kimbrough Urological Sem. 4: 131 (1970). 5. OSTROPOLSKAYA, E. A. : Tumors of horseshoe kidney in children, Urol. Nefrol. 36: 58 (1971). 6. ALWASIAK, J.: Case of clear cell carcinoma of the horseshoe kidney, Pal. Przegl. Chir. 43: 1043 (1971). in a horse7. BURKERT, S. : Two cases of hypemephroma shoe kidney, Fortschr. Geb. Roentgenstr. Nuklearmed. 115: 186 (1971). 8. K~~LLIN, C. P., BOATMAN, D. L., SCHMIDT, J. D., and FLOCKS, R. H.: Horseshoe kidney: a review of 105 patients, J. Ural. 107: 203 (1972). kidney, 9. KOTOWICZ, A.: Malignant tumor ofhorseshoe Acta Ural. Belg. 40: 85 (1972). 10. SEGURA, J.W., KELALIS,P. O., and BURKE, E. C.: Horseshoe kidney in children, J, Urol. 108: 333 (1972). 11. ITZIC,C. B.,PERRY, S. R.,and MCCAUGHAN, J.J.: Adenocarcinoma of the isthmus of a horseshoe kidney: a case report and review of the literature, Am. Surg. 38: 422 (1972). Renal cell carci12. REDMAN, J. F., and MOBLEY, J, E.: noma in a horseshoe kidney, South. Med. J, 66: 963 (1973). 13. KUMAR, M., KUMAR, B. T., and BHAT, H. S.: Diagnosis and treatment of diseased horseshoe kidney, Int. Surg. 59: 159 (1974). 14. SHASHIKUMAR,~.L.,SOMERS,L. A.,PILLING, G. P., and CRESSON, S. L.: Wilms’ tumor in the horseshoe kidney, J. Pediatr. Surg. 9: 185 (1974). 15. CHOOVICHIAN,T., YALLA, S. V., DELIMA, A., and BURROS, H. M.: Transitional cell carcinoma in a horseshoe kidney, South. Med. J. 67: 619 (1974). 16. LUCKE, B., and SCHLUMBERGER, H. G.: Tumors of the kidney, renal pelvis and ureter. Atlas of Tumor Pathology, Armed Forces Institute of Pathology, 1957, section VIII, part 30, p. 13.
UROLOGY
/
AUGUST 1976 /
VOLUME VIII, NUMBER 2
MALIGNANCY HORSESHOE
ASSOCIATED
WITH
KIDNEY*
MAJ. DAVID BUNTLEY,
(MC) USA
From the Urology Service, Fitzsimons Medical Cetiter, Denver, Colorado
Army
i \/IS7’Rr,(; 1 - A compilation of the reported cases of cancer in horseshoe kidney from 1895 to 1975 is 1~-cw:n~c rl &ng with a comparison of the incidence of the various types of tumor involved from 1895 to 1963 a’n~;1%6 to 1975. An additional case of hypernephroma is added to the literature for a total of 111 ( ~LSC:; rq lor-tl:dto date. The increased incidence of both carcinoma of the renal pelvis and nephroblastoBOIL is di mused.
‘rEe firs : and most comprehensive review of the *~~rlcL literature regarding the association of renal :letrplasln and horseshoe kidney was that published 1~: Blackard and Mellinger.’ They reported ; t tot,al of 72 cases with the histologic breakdown as 5eer1 in ‘Table I. Dajani2 added 1 case of hyper:lephrorla not included in the Blackard series. In 1969 Cc Ilichaev, Pevzner, and Ivanov3 supplied XI additional case of hypernephroma. U’right4 reviewed the literature from 1966 to 1968 an11 discovered 9 additional cases of hyper:lepbrorla, 2 of Wilms’ tumor and 2 of sarcoma. We added 1 case of hypernephroma. In 1971Ostropolskaya” reported 4 cases of Wilms’ tumor occurring in 38 children with horseshoe kidney. ,Slso during the same year Alwasiak’ contributed I case oj‘hypernephroma discovered at necropsy ;md Esuritert’ reported 2 cases ofhypernephroma. The following year KGllin et al. 8 reported 1 case each of hypernephroma, Wilms’ tumor, and lymphoma with renal involvement. Kotowiczg added 1 case of sarcoma and Segura, Kelalis, and BurkelO reported a single case of lymphoblastic lymphoma. In addition, Itzig, Perry, and McCaughan” reported 1 case ofhypernephroma. Redman and Mobley12 reviewed the literature in 1973 and added 1 case of hypernephroma (Bur-
*The opinions or assertions contained herein are the private vic:ws of the author and are not to be construed as official or as reflecting the views of the Department of the Army or the Department of Defense.
146
kovskii) not reported previously and included an additional case. Kumar, Kumar, and Bhat13 contributed 1 case of hypernephroma in 1974 followed by a review by Shashikumar et al. l4 who added 1 case of Wilms’ tumor not reported previously (Bhatnagar) plus 3 additional cases of Wilms’ tumor. During the same year Choovichian et al. l5 contributed 1 case of transitional cell carcinoma of the renal pelvis. An additional case of hypernephroma in a horseshoe kidney is reported herein. Case Report The patient is a sixty-four-year-old Caucasian male in whom microhematuria with dysuria developed approximately forty-eight hours after a right inguinal hernia repair during November, 1972, at Reynolds Army Hospital, Ft. Sill, Oklahoma. A urologic workup was initiated. Intravenous pyelogram revealed a mass in the left upper pole of a previously undiagnosed horseshoe kidney (Fig. 1A). Nephrotomography and arteriography showed the lesion to be a noncystic, relatively avascular mass (Fig. 1B). Diagnostic puncture of the lesion was not attempted. Findings on chest x-ray film and laboratory values were all within normal limits with the exception of an elevated serum alkaline phosphatase attributed to the patient’s Paget’s disease. On January 24, 1973, the patient was explored through an abdominal transperitoneal approach.
UROLOGY
/ AUGUST
1976/
VOLUME
VIII,
NUMBER
2
review. He concluded that “probably cancer is no more likely to occur in a horseshoe kidney than it is in a normal kidney. “l However, he stated that there was a higher incidence of renal pelvis carcinoma and nephroblastoma. Choovichian and coworkerG also concluded that the incidence of carcinoma which predispose the horseshoe kidney to infection and hydronephrosis, namely, urinary stasis, predispose the renal collecting system to prolonged contact with carcinogenic components of the excreted urine.
There was a firm mass 2.5 cm. in diameter confined to the left upper pole. Left heminephrectomy was accomplished by clamping the vessels supplying the left half of the kidney. Division of a fibrous isthmus was performed at the line of ischemic demarcation. Horizontal mattress sutures were tied over adipose tissue to achieve hemostasis. Cross section of the kidney showed a 2.5-cm. pale yellow tumor, well circumscribed, with focal areas of hemorrhage. There was no grossly apparent involvement of the collecting system or the vessels. Microscopic examination revealed renal cell carcinoma with small neoplastic cells containing rounded hyperchromatic nuclei. The cytoplasm varied from eosinophilic to clear. No vascular invasion was identified (Fig. 2). The postoperative course was marked by a 10 per cent drop in the hematocrit which stabilized with conservative management and transfusion of blood. The patient had no further complications and is alive and well without evidence of recurrence two years after surgery. Comment A total of 111 cases of cancer in a horseshoe kidney have been reported in the world literature since 1895 from which date Blackard initiated his
UROLOGY
/ AUGUST 1976 / VOLUME
VIII,
NUMBER
showing relatively FIGURE 2. Photomicrograph clear cytoplasm with hyperchromutic nuclei of hypernephroma.
2
147
TABLE I.
of incidence of types of carcinoma horseshoe kidney
Comparison
1966
to 1975
No.
of CusesI Percent
r--
I
20
I
Wllm s Tumor I------
1
14
1 20
Renal Pelvis Carcinoma
TOTAL
72
A comparison of the percentages of occurrence of the four main categories of cancer reviewed shows a marked similarity between the Blackard and Mellinger figures with those of this review (Table I). However, an analysis of the 39 cases reviewed since 1966 reveals a significant decrease in occurrence of Wilms’ tumor from 28 to 12.8 per cent. The figure more closely approximates that quoted by Lucke and SchlumbergeP of 5.6 per cent incidence of Wilms’ tumor in 1,659 malignant renal tumors. This figure is halfthe observed incidence seen in horseshoe kidney. The fact that there is an increased incidence of both carcinoma of the renal pelvis and nephroblastoma in a horseshoe kidney should alert the urologist to search for these entities in patients found to have this anomaly. The pediatric patient with a horseshoe kidney should be evaluated and followed adequately not only to discover deteriorating renal disease secondary to the recognized complications of obstruction and infection, but also to interpret early any malignant degeneration. Denver, Colorado 80240 ACKNOWLEDGMENT. To John W. Weigel, M.D., Chief of Urology, Fitzsimons Army Medical Center for editorial assistance. References 1. BLACKARD, C. E., and MELLINGER, T. T. : Cancer in a horsehoe kidney, Arch. Surg. 97: 616 (1968). 2. DAJANI, M. B.: Horseshoe kidney: a review oftwentynine cases, Br. J. Ural. 38: 388 (1966).
148
1895
in
to (975
No. of Cases I PwCent
28
5.1
2
I
occurring
I
.9
2
I.8
III
39
3. GOLICHAEV, Z. K., PEVZNER, P., and IVANOV, A. V.: Tumor ofhorseshoe kidney, Urol. Nefrol. 34: 57 (1969). 4. WRIGHT, R.: A case of carcinoma in a horseshoe kidney and review of the literature, Proc. Kimbrough Urological Sem. 4: 131 (1970). 5. OSTROPOLSKAYA, E. A. : Tumors of horseshoe kidney in children, Urol. Nefrol. 36: 58 (1971). 6. ALWASIAK, J.: Case of clear cell carcinoma of the horseshoe kidney, Pal. Przegl. Chir. 43: 1043 (1971). in a horse7. BURKERT, S. : Two cases of hypemephroma shoe kidney, Fortschr. Geb. Roentgenstr. Nuklearmed. 115: 186 (1971). 8. K~~LLIN, C. P., BOATMAN, D. L., SCHMIDT, J. D., and FLOCKS, R. H.: Horseshoe kidney: a review of 105 patients, J. Ural. 107: 203 (1972). kidney, 9. KOTOWICZ, A.: Malignant tumor ofhorseshoe Acta Ural. Belg. 40: 85 (1972). 10. SEGURA, J.W., KELALIS,P. O., and BURKE, E. C.: Horseshoe kidney in children, J, Urol. 108: 333 (1972). 11. ITZIC,C. B.,PERRY, S. R.,and MCCAUGHAN, J.J.: Adenocarcinoma of the isthmus of a horseshoe kidney: a case report and review of the literature, Am. Surg. 38: 422 (1972). Renal cell carci12. REDMAN, J. F., and MOBLEY, J, E.: noma in a horseshoe kidney, South. Med. J, 66: 963 (1973). 13. KUMAR, M., KUMAR, B. T., and BHAT, H. S.: Diagnosis and treatment of diseased horseshoe kidney, Int. Surg. 59: 159 (1974). 14. SHASHIKUMAR,~.L.,SOMERS,L. A.,PILLING, G. P., and CRESSON, S. L.: Wilms’ tumor in the horseshoe kidney, J. Pediatr. Surg. 9: 185 (1974). 15. CHOOVICHIAN,T., YALLA, S. V., DELIMA, A., and BURROS, H. M.: Transitional cell carcinoma in a horseshoe kidney, South. Med. J. 67: 619 (1974). 16. LUCKE, B., and SCHLUMBERGER, H. G.: Tumors of the kidney, renal pelvis and ureter. Atlas of Tumor Pathology, Armed Forces Institute of Pathology, 1957, section VIII, part 30, p. 13.
UROLOGY
/
AUGUST 1976 /
VOLUME VIII, NUMBER 2
