Vol. 112, November Printed in U.S.A.
THE JOURNAL OF UROLOGY
Copyright© 1974 by The Williams & Wilkins Co.
FAMILIAL OCCURRENCE OF TESTICULAR NEOPLASMS: A CASE REPORT R. M. GULLEY,* R. KOWALSKI
AND
C. F. NEUHOFF
From the Departments of Family Practice and Pathology, St. Francis Hospital, Peoria, Illinois
Several reports have been made on the occurrence of testicular neoplasms in members of the same family. These reports have been limited to monozygotic and dizygotic twins, 1 -• non-twin brothers 1 - 12 and a father and son. 13 Our report concerns testicular neoplasms in 4 brothers. CASE REPORTS
Case 1. C. E., a 31-year-old white man, was admitted to another hospital in February 1956 with
peritoneal node dissection was done and extensive nodal metastasis was identified. The patient died several months later. Case 2. W. E., a 34-year-old white man, was hospitalized in April 1969 with a 1-year history of progressive, painful enlargement of the right testis, a 65-pound weight loss and a 4-month history of an abdominal mass. Physical examination revealed a large, firm, immobile mass in the right lower quadrant of the abdomen. The right testis was
FIG. 1
a 2-month history of chills, fever and a feeling of heaviness associated with left testicular enlargement. The only abnormality reported on physical examination was an enlarged left testis. Left orchiectomy was done and diagnosis was embryonal cell carcinoma (fig. 1). Two months later radical retro-
reported to be 3 times normal size and partially transmitted light. Right radical orchiectomy was done and diagnosis was mixed em bryonal cell carcinoma (fig. 2, A) and seminoma (fig. 2, B). The patient underwent a course of radiation therapy to
Accepted for publication May 3, 1974. * Current address: 3261 McDermott St., Orlando, Florida 32809. 1 Champlin, H. W.: Similar tumors of testis occurring in identical twins. J.A.M.A., 95: 96, 1930. 2 Wells, H. G.: Personal communication to Twinem, 1937. Cited by Macklin, M. T.: Analysis of tumors in monozygous and dizygous twins, with report of 15 unpublished cases. J. Hered., 31: 277, 1940. •Domrich, H.: Ober Leistenhodencarcinom bei Zwillingen. Arch. f. Klin. Chir., 197: 848, 1940. 'Salm, R. and Adlington, S. R.: Seminoma in identical twins. Brit. Med. J., 2: 964, 1962. 5 Stewart, J. R. and Bagshaw, M. A.: Malignant testicular tumors appearing simultaneously in identical twins: a case report. Cancer, 18: 895, 1965. 6 Villani, U.: Tumore concordante de! testicolo in una 620
coppia di gemelli monozigoit. Acta Genet. Med., 16: 172, 1967. 7 Raven, R. W.: Tumours of the testis in two brothers. Lancet, 2: 870, 1934. • Lownes, J. B. and Leberman, P.: Tumors of the testis in brothers. Urol. & Cutan. Rev., 43: 205, 1939. 'Willis, R. A.: Pathology of Tumours, 2nd ed. London: Butterworth & Co., Ltd., pp. 557-558, 1953. 10 Hutter, A. M., Jr., Lynch, J. J. and Shnider, B. I.: Malignant testicular tumors in brothers. A case report. J.A.M.A., 199: 1009, 1967. 11 Myers, G. H. and Macpherson, B. R.: Histologically similar testicular neoplasms occurring in brothers. J. Urol., 108: 757, 1972. 12 Young, J. A. and Bohne, A. W.: Seminoma in non-twin brothers: a case report. J. Urol., 107: 1000, 1972. 13 Silber, S. J ., Cittan, S. and Friedlander, G.: Testicular neoplasm in father and son. J. Urol., 108: 889, 1972.
FAMILIAL OCCURRENCE OF TESTICULAR NEOPLASMS
621
FIG. 2
the retroperitoneal area, foll.owed by an attempted pelvic exenteration. However, at the second operation, nodal involvement was found proximal to the renal arteries and the exenteration was not performed. He then received a course of radiation therapy consisting of to the abdominal mass, para-aortic, mediastinal and left supraclavicular nodes. The patient followed a progressive downhill course and he died in November 1970. Postmortem examination was refused. Case 3. J. E., a refused induction at a draft in December 1969 because of an enlarged testis. In March 1970 he complained of gross, terminal hernaturia. The right testis was described as twice normal size, hard, smooth and non-tender. Right radical orchiectomy and exploratory ~~ ..~•rnw-., revealed no involvement of the regional nodes. Microscopic diagnosis was pure seminoma (fig. 3). This patient
underwent a course of radiation therapy and was seen one of us in 1973, at which time physical examination and chest x-ray showed him to be free of recurrence. Case 4. M. E., a 40-year-old white man, admitted to the hospital for a disability examination to qualify for public aid. He stated that he had had a double hernia for the last 2 years. He further stated that he had experienced pain in both testes for some time and had been denied 2 occasions during the preceding 2 years ue,c«Lrne the scrotal masses. He had been advised immediate medical attention. He admitted to progressive enlargement of both testes and a 20pound weight loss. Pertinent physical were limited to the abdomen and genitalia. Bilateral, rock hard scrotal masses were present, suring approximately 15 by 10 cm. There was moderate tenderness in both inguinal areas with
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GULLEY, KOWALSKI AND NEUHOFF
FIG. 3
marked induration about the left inguinal ligament. No discrete adenopathy was appreciated. A consulting urologist who examined the patient agreed with the diagnosis of probable bilateral testicular neoplasms. Despite vigorous efforts to persuade him the patient refused diagnostic and therapeutic procedures and was lost to followup until April 1973. At that time, he allowed only limited physical examination, which revealed the presence of a large (20 cm. in diameter) cystic mass occupying the right scrotum, with a large, tender, poorly defined firm mass at the external ring. The left scrotal mass was unchanged. He had lost an additional 20 pounds. Despite his obvious discomfort, he again refused treatment and was lost to followup. FAMILY HISTORY
Family history was obtained through questioning J. E. and M. E., without benefit of review of medical or legal records. The father had been killed in a train wreck when he was 72 years old and "may have had cancer when he died". He was 43 years old when married and his wife was 14 years old. The mother had died of "heart trouble". There were 14 siblings (6 male and 8 female). The uninvolved male siblings include an older brother, 43 years old, who is reported to be "starting to have troubles
with his testicles" and the youngest brother, 25 years old, who was examined and found to have normal genitalia. One female sibling was reported to have had an operation for "cancer of the female organs" when she was 20 years old. Attempts to locate the older brother and female siblings have been unsuccessful. DISCUSSION
This unique occurrence of testicular neoplasms in 4 brothers again raises the question of the role of genetic factors in the development of these neoplasms. The 3 recent reports of the familial occur rence of testicular tumors, 11-• 3 added to our report, emphasize the desirability of adequate case reporting as a tool in further clarifying the role of genetic factors in the development of these uncommon neoplasms. The fact that the histological types were of pure embryonal cell, mixed embryonal cell and seminoma and pure seminoma is also of interest, in view of the germ cell theory of origin of these tumors. Unfortunately, this family did not lend itself well to further epidemiologic or immunologic investigation. 0
SUMMARY
This is the first report of the occurrence of testicular neoplasms in 4 brothers.