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Malignant testicular tumors in non-twin brothers
MALIGNANT TESTICULAR TUMORS IN NON-TWIN BROTHERS NAGUI E . ADEEB, M .D. PASQUALE A . GRECO, M .D . From the Department of Urology, Millard Fillmore Hospital, Buffalo, New York
ABSTRACT -A case report of testicular tumors in non-twin brothers is presented . Review of the literature revealed 5 similar cases . The possibility of a genetic factor influencing the development of germinal testicular tumors is discussed .
The first report of malignant testicular tumors in non-twin brothers was described by Raven in 1934.' Since then four other reports of similar instances have appeared in the literature .` We report the sixth case of testicular neoplasm in nontwin brothers . Case Reports Case 1 A twenty-eight-year-old white man was admitted on November 25, 1965, with painful swelling of the left testicle and pain in the left groin of two weeks' duration . The patient gave a history of having been hit in the scrotum four weeks prior to admission . On physical examination the left testicle was found to be enlarged to twice the size of the right testicle, tender, and of firm consistency . Abdominal examination was negative for masses and organomegaly . Laboratory findings which included a complete blood count, urinalysis and culture, and blood chemistry were within normal limits . Chest x-ray film and excretory urogram were normal . A lymphangiogram was not performed . On November 30, a radical left orchiectomy was performed . Examination of the gross specimen revealed complete replacement of the testicle by grayish white tumor tissue . Microscopically, the tumor consisted of muscle fibers, osseous and cartilagenous tissue with islands of fat cells and undifferentiated epithelial elements, a picture diagnostic of teratocarcinoma (Fig . 1A to C) .
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On December 8, a retroperitoneal lymphadenectomy was attempted but could not be accomplished because of lymph node fixation to the aorta in the region of the renal vessels . Paraaortic node biopsy revealed complete replacement of the nodal tissue by tumor identical with the testicular neoplasm . The patient subsequently received radiation therapy and actinomycin D . He was readmitted in March, 1967, with extensive pulmonary and abdominal metastases, deteriorated rapidly, and died on May 4, 1967, eighteen months after the diagnosis was made . Case 2 A thirty-five-year-old man, the brother of the patient in Case 1, was admitted on February 8, 1966, with a five-month history of nonpainful, nontender swelling of the right testicle . On examination, the testicle was found enlarged, of a firm consistency, and nontender . Abdominal examination was negative for palpable masses . Laboratory studies were within normal range . Chest x-ray film and an excretory urogram were normal . On February 10, the patient underwent right radical orchiectomy. Histologically, the tumor, which replaced the entire testicle, was composed of sheets of pale-staining cells with small round cell infiltrates, a picture diagnostic of seminoma (Fig . 1D). The patient received radiation therapy postoperatively . He is, at present, eight years postorchiectomy and radiation, asymptomatic, with no evidence of metastases .
UROLOGY
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JULY 1975 /
VOLUME VI, NUMBER 1
FIGURE 1 . (A to C) Photomicrographs of mixed tumor in Case 1 : teratomatous elements (A) showing muscle fibers, osseous and cartilagenous tissues ( x 100); undifferentiated epithelial elements (B) in low-power ( x 100) and (C) high-power (x 250) . (D) Seminoma in Case 2 showing sheets of pale-staining cells with small round cell infiltrates (X 2 .50) .
Comment Testicular tumors constitute less than 1 per cent of all malignancies in males . The incidence of these tumors is approximately 2 .3 per 100,000. The tragedy of their occurrence in siblings raises the question of being more than a coincidence . The occurrence of testicular neoplasms in a father and son has also been reported .' The influence of a genetic factor in the UROLOGY
/
JULY 1975 i
VOLUME VI, NUMBER 1
development of these tumors has been suggested on the basis of (1) scarcity in the Negro and oriental races ; (2) occasional appearance in families and case reports of occurrence in twins' ,8 and non-twin brothers ; (3) the frequent presence of a positive chromatin pattern in the cells of the seminomas and mature teratocarcinoma . Statistical evidence of a genetic influence on the development of these neoplasms in siblings has been limited by the relative infrequency of
99
occurrence
of
these
tumors
in the
general
population . 45 Provincetown Lane Orchard Park, New York 14127 (DR . ADEEB) References 1 . RAVEN, R. W . ; Tumours of the testis in two brothers, Lancet 2 : 870 (1934) . 2 . NUTTER, A . M ., JR ., LYNCH, J . L ., and SCIHcIDER, B, I . : Malignant testicular tumours in brothers . A case report, J .A.M .A . 199 : 155 (1967) .
100
3 . LOWNES, J . B., and LEBERMAN, P . : Tumors of the testis in brothers, Urol . Cutan . Rev . 43 : 205 (1939) . 4 . MEYER, G . H ., JR ., and MACPHERSON, B . R . : Histologically similar testicular neoplasms occurring in brothers, J . Urol . 108 : 757 (1972) . 5 . YOUNG, J . A ., and BORNE, A . W . : Seminoma in nontwin brothers . A case report, ibid. 107 : 1000 (1972) . 6 . SILBER, S . J ., GITrAN, S ., and FRIEDLANDER, G . : Testicular neoplasm in father and son, ibid . 108: 889 (1972) . 7 . CHAMPLIN, H . W . : Similar tumours of testis occurring in identical twins, J . A .M .A . 95 : 96 (1930). 8 . STEWART, J . R ., and BACSHAW, M . A . : Malignant testicular tumours appearing simultaneously in identical twins . A case report, Cancer 18 : 895 (1965) .
UROLOGY / JULY 1975
/ VOLUME VI, NUMBER 1
ABSTRACT -A case report of testicular tumors in non-twin brothers is presented . Review of the literature revealed 5 similar cases . The possibility of a genetic factor influencing the development of germinal testicular tumors is discussed .
The first report of malignant testicular tumors in non-twin brothers was described by Raven in 1934.' Since then four other reports of similar instances have appeared in the literature .` We report the sixth case of testicular neoplasm in nontwin brothers . Case Reports Case 1 A twenty-eight-year-old white man was admitted on November 25, 1965, with painful swelling of the left testicle and pain in the left groin of two weeks' duration . The patient gave a history of having been hit in the scrotum four weeks prior to admission . On physical examination the left testicle was found to be enlarged to twice the size of the right testicle, tender, and of firm consistency . Abdominal examination was negative for masses and organomegaly . Laboratory findings which included a complete blood count, urinalysis and culture, and blood chemistry were within normal limits . Chest x-ray film and excretory urogram were normal . A lymphangiogram was not performed . On November 30, a radical left orchiectomy was performed . Examination of the gross specimen revealed complete replacement of the testicle by grayish white tumor tissue . Microscopically, the tumor consisted of muscle fibers, osseous and cartilagenous tissue with islands of fat cells and undifferentiated epithelial elements, a picture diagnostic of teratocarcinoma (Fig . 1A to C) .
98
On December 8, a retroperitoneal lymphadenectomy was attempted but could not be accomplished because of lymph node fixation to the aorta in the region of the renal vessels . Paraaortic node biopsy revealed complete replacement of the nodal tissue by tumor identical with the testicular neoplasm . The patient subsequently received radiation therapy and actinomycin D . He was readmitted in March, 1967, with extensive pulmonary and abdominal metastases, deteriorated rapidly, and died on May 4, 1967, eighteen months after the diagnosis was made . Case 2 A thirty-five-year-old man, the brother of the patient in Case 1, was admitted on February 8, 1966, with a five-month history of nonpainful, nontender swelling of the right testicle . On examination, the testicle was found enlarged, of a firm consistency, and nontender . Abdominal examination was negative for palpable masses . Laboratory studies were within normal range . Chest x-ray film and an excretory urogram were normal . On February 10, the patient underwent right radical orchiectomy. Histologically, the tumor, which replaced the entire testicle, was composed of sheets of pale-staining cells with small round cell infiltrates, a picture diagnostic of seminoma (Fig . 1D). The patient received radiation therapy postoperatively . He is, at present, eight years postorchiectomy and radiation, asymptomatic, with no evidence of metastases .
UROLOGY
/
JULY 1975 /
VOLUME VI, NUMBER 1
FIGURE 1 . (A to C) Photomicrographs of mixed tumor in Case 1 : teratomatous elements (A) showing muscle fibers, osseous and cartilagenous tissues ( x 100); undifferentiated epithelial elements (B) in low-power ( x 100) and (C) high-power (x 250) . (D) Seminoma in Case 2 showing sheets of pale-staining cells with small round cell infiltrates (X 2 .50) .
Comment Testicular tumors constitute less than 1 per cent of all malignancies in males . The incidence of these tumors is approximately 2 .3 per 100,000. The tragedy of their occurrence in siblings raises the question of being more than a coincidence . The occurrence of testicular neoplasms in a father and son has also been reported .' The influence of a genetic factor in the UROLOGY
/
JULY 1975 i
VOLUME VI, NUMBER 1
development of these tumors has been suggested on the basis of (1) scarcity in the Negro and oriental races ; (2) occasional appearance in families and case reports of occurrence in twins' ,8 and non-twin brothers ; (3) the frequent presence of a positive chromatin pattern in the cells of the seminomas and mature teratocarcinoma . Statistical evidence of a genetic influence on the development of these neoplasms in siblings has been limited by the relative infrequency of
99
occurrence
of
these
tumors
in the
general
population . 45 Provincetown Lane Orchard Park, New York 14127 (DR . ADEEB) References 1 . RAVEN, R. W . ; Tumours of the testis in two brothers, Lancet 2 : 870 (1934) . 2 . NUTTER, A . M ., JR ., LYNCH, J . L ., and SCIHcIDER, B, I . : Malignant testicular tumours in brothers . A case report, J .A.M .A . 199 : 155 (1967) .
100
3 . LOWNES, J . B., and LEBERMAN, P . : Tumors of the testis in brothers, Urol . Cutan . Rev . 43 : 205 (1939) . 4 . MEYER, G . H ., JR ., and MACPHERSON, B . R . : Histologically similar testicular neoplasms occurring in brothers, J . Urol . 108 : 757 (1972) . 5 . YOUNG, J . A ., and BORNE, A . W . : Seminoma in nontwin brothers . A case report, ibid. 107 : 1000 (1972) . 6 . SILBER, S . J ., GITrAN, S ., and FRIEDLANDER, G . : Testicular neoplasm in father and son, ibid . 108: 889 (1972) . 7 . CHAMPLIN, H . W . : Similar tumours of testis occurring in identical twins, J . A .M .A . 95 : 96 (1930). 8 . STEWART, J . R ., and BACSHAW, M . A . : Malignant testicular tumours appearing simultaneously in identical twins . A case report, Cancer 18 : 895 (1965) .
UROLOGY / JULY 1975
/ VOLUME VI, NUMBER 1