Bilateral successive testicular cancer of different cell types

BILATERAL SUCCESSIVE DIFFERENT

TESTICULAR

CANCER OF

CELL TYPES

MAJ.

MAX PHILLIP

From

Patrick

LEVINE,

Air Force

Base,

(MC) USAF Florida

ABSTRACT - The fourth case of bilateral successive testicular tumors of different cell types is reported (seminoma and embryonal carcinoma), and the English literature on bilateral testicular tumors from 1965 to 1975 is reviewed. The absence of direct organ extension or demonstrable metastatic lymphatic or vascular involvement points to the primary origin of these lesions.

Testicular neoplasms are a rare occurrence with an incidence of two per 100,000 male subjects.’ The frequency of bilateral testicle tumors is stated to be 2 to 3 per cent of the over-all total in the British literaturezm4 and 1 to 2 per cent in American sources5“ Gilbert and Hamilton* cite a 0.7 per cent incidence ofa second testicular neoplasm in men with one scrotal testicular cancer. Successive tumors are more common than those appearing synchronously. Lymphomas are the most common bilateral lesions and seminomas next in frequency. ‘5’ The very uncommon Leydig cell tumor has a 5 to 9 per cent incidence of bilaterality. lo This report presents a case of bilateral successive histologically different testicular tumors (embryo& cell carcinoma and seminoma) and reviews the English literature on bilateral testicular tumors from 1965 to 1975. Case Report This forty-year-old white male presented at Lackland Air Force Base in 1961with a 2-cm. left testicular mass. The physical examination and laboratory data were essentially normal. Chest x-ray film and electrocardiogram were also normal. A radical orchiectomy and concomitant retroperitoneal node dissection for embryonal cell carcinoma were performed. All nodes were negative for metastases. His postoperative course was uneventful. In August, 1968, he presented at Grissom Air Force Base, Indiana, with a painful l-cm. testicular mass in the right upper pole. Physical exami-

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nation revealed a healthy looking man with an empty left scrotum. There was no significant adenopathy or hepatomegaly. Findings on chest x-ray film and bone scan were normal. An intravenous pyelogram demonstrated medial deviation of the right ureter compatible with a prior retroperitoneal node dissection. Laboratory data were normal. A transcrotal excisional biopsy revealed seminoma. On September 4, 1968, he underwent a radical orchiectomy and iliac node dissection. Pathologic examination confirmed a seminoma confined to the upper pole of the testicle and negative adenopathy. He received 3,000 r to the right ilioinguinal and periaortic regions and 2,000 r to both the mediastinum and right supraclavicular fossa. Follow-up examinations at the Patrick Air Force Base Tumor Board have shown negative bone surveys, chest roentgenograms, and liver scans. He is currently on methyl testosterone (10 mg. daily) for impotence. Comment The incidence of cancer bilaterality in such organs as the ovary and breast may be as high as 35 per cent. l1 Such is not the case with testicular neoplasms. Abeshouse, Tiongson, and Goldfarb5 reviewed the literature in 1955 and found 9 cases of simultaneous bilateral embryonal carcinoma. Dunlap and Oschsner l2 described bilateral successive embryonal carcinomas in a thirty-oneyear-old white male with an interval of ten years between tumors. They were unable to find

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another such case and ascribed this to the dire prognosis of one testicular embryonal carcinoma. In 1972 Willis and Hajdu13 reviewed the literature and collected 26 cases previously reported of histologically confirmed bilateral malignant germ cell tumors of the testes. In a study of315 patients from Memorial Hospital, New York, with primary malignant germ cell tumors they reported 2 additional cases. The first was a thirty-year-old white man who underwent right orchiectomy and cobalt-60 radiotherapy for seminoma in 1964 and reappeared in 1965 with an embryonal carcinoma of the left testicle. He died of’metastatic disease in 1966. The second case involved a twentytwo-year-old white man whose left testicular seminoma was succeeded in six months by a right embryonal carcinoma. Concomitant bilateral orchiectomy and retroperitoneal node dissection were performed. The specimen showed no metastatic disease, and the patient was well at sixmonths follow-up. Farivari, Nasser, and Montiel14 initially treated a forty-five-year-old white male with left embryonal carcinoma with radical orchiectomy, retroperitoneal node dissection, and radiotherapy in 1960, and twelve years later employed contralateral orchiectomy and radiotherapy for seminoma. The patient was without recurrence at one year. Sadoughi et al. l5 described an eighteen-year-old man with bilateral simultaneous embryonal carcinoma with choriocarcinomatous elements. This individual had widespread metastatic disease at laparotomy, and the authors concluded that the bilaterality in this case was metastatic rather than primary. This report documents the fourth case of successive bilateral testicular tumor of different cell type (seminoma and embryonal carcinoma). Seminomas are the second most common bilateral testicular tumors’ with an incidence of approximately 2 per cent in several large series of seminomas. 16-18The English literature in the 1965 to 1975 decade documents 9 additional cases of bilateral testicular seminomas. Vechinski, Jaeschke, and Vermund17 recorded an incidence of 2.7 per cent in 112 cases. Castro and Gonzalez” treated 3 such patients in a series of 96 seminomas (2.5 per cent). Das,lg McLean20 and Barbalias21 each documented an additional case. Rosai, Silber, and Khodadoust22 reviewed 81 cases designated as classic seminoma and reclassified 6 individuals as examples of the more benign and uncommon spermatocytic seminoma. The authors recorded 2 instances of bilaterality in a seventy-three-year-old white man (synchro-

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nously) and a fifty-seven-year-old white man (metachronously). Their review of the literature detailed 26 cases of spermatocytic seminoma and cited an over-all incidence of 6 per cent bilaterality. Bilaterality may be more prevalent in this lesion than in classic seminoma. Eight per cent of classic seminomas arise in undescended testes.16 A higher incidence of bilaterality (24.6 per cent) has also been noted in bilateral ectopy. 23 In up to one third of pseudohermaphrodites a tumor of one or both gonads may develop. 24,25Goel and Samue124 described a twenty-six-year-old married phenotypically normal male with a suprapubic mass whose scrotum was devoid of testes. Surgical removal of the mass disclosed a classic seminoma, an infantile uterus, fallopian tubes, and an atrophic testis with seminoma. The pre- and para-aortic lymphatics contained far advanced metastatic disease. The atrophic testicle with seminoma may represent retrograde lymphatic dissemination rather than a separate primary. Sc~lly~~ reviewed the literature in 1970 and evaluated 74 gonadoblastomas of which 27 were bilateral. He defined the tumor as a “neoplasm containing an intimate mixture of germ cells and elements resembling immature granulosa or Sertoli cells; Leydig cells or lutein type cells may or may not be present.” All patients were sexually abnormal. In the entire series 80 per cent were phenotypically female with virilization or phenotypically male (20 per cent) with cryptorchidism, hypospadias, or female internal secondary sex organs. The literature mentions instances of bilateral testicular neoplasms which are exceedingly rare. In a review of 6,000 testicular tumors the incidence of Leydig cell tumor was 3 per cent.‘O Leydig cell tumor is usually unilateral but 5 to 9 per cent are bilateral. In such cases the diagnosis of adrenogenital syndrome should be entertained.‘0,27 Carney, Kelalis, and Lynn28 documented the second case of bilateral testicular teratoma in a prepubertal child in 1973. Strahlberg and Brown2’ reported the only known case of concomitant bilateral epidermoid cysts of the testes. Hyde Park No. 2 Houston, Texas 77006

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and frequency of testicular tumours, Br. J. Urol. 36: 1 (1964). 3. WHITTLE, R. J. M.: Tumours of the testicle, Br. J. Radiol. 30: 7 (1957). 4. STEPHEN, R. A.: Malignant testicular tumours, Ann. R. Coll. Surg. Engl. 23: 71 (1958). 5. ABESHOUSE, B. S., TIONGSON, A., and COLDFARB, M. : Bilateral tumors of testicles: review of the literature and report of a case of bilateral simultaneous lymphosarcoma, J. Urol. 74: 522 (1955). 6. PA~ON, J. F., SELTZMAN, D. N., and ZONI, R. A.: Diagnosis and treatment of testicular tumors, Am. J. Surg. 99: 525 (1960). of different pathologic 7. SULAK, M. H.: Classification types, J.A.M.A. 213: 91 (1970). 8. GILBERT, J. B., and HAMILTON, J. B.: Studies in malignant testis tumors; incidence and nature of tumors in ectopic testes, Surg. Gynecol. Obstet. 71:731(1940). 9. HAMLIN, J. A., KAGAN, R. A., and FRIEDMAN, N. A.: Lymphomas ofthe testicle, Cancer29: 1352(1972). 10. MOSTOFI, F. K.: Testicular tumors. Epidemiologic, etiologic, and pathologic features, ibid. 32: 1186(1973). Current concepts in the detection and 11. FARROW, J. H.: treatment of the early breast cancers, ibid. 25: 468 (1970). 12. DUNLAP, D. J., and OCHSNER, J. A.: Bilateral successive embryonal carcinoma of the testes, J. Urol. 97: 738 (1967). 13. WILLIS, G. W., and HAJDU, S. I.: Bilateral primary malignant germ cell tumors of the testes: report of two cases, ibid. 107: 279 (1972). 14. FARIVARI, A., NASSER, J., and MONTIEL, M.: Bilateral successive testicular cancer of different cell type, Urology 2: 458 (1973). 15. SADOUGHI, N., et al.: Bilateral germ cell tumors of testes, ibid. 2: 452 (1973). 16. THACKRAY, A. C.: Seminoma. The pathology of tes-

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titular tumours, Br. J. Urol. 36 (suppl): 12 (1964). 17. VECHINSKI, T. O., JAESCHKE, W. H., and VERMUND, H.: Testicular tumors. Analysis of 112 consecutive cases, Am. J. Roentgen. Radium Ther. Nucl. Med. 95: 494 (1965). 18. CASTRO, J. R., and GONZALEZ, M.: Results in treatment of pure seminoma of the testis, ibid. 111: 355 11971L 19. ‘DAs,’ S.: Bilateral testicular tumors, J. Indian Med. Assoc. 50: 526 (1968). 20. MCLEAN, P. A.: Bilateral simultaneous seminoma of the testes. A case report, J. Ir. Med. Assoc. 66: 514 (1973). 21. BARBALIAS, G. A.: Bilateral successive seminomas in scrotal testes, Del. Med. J. 46: 243 (1974). 22. ROSAI, J., SILBER, I., and KHODADOUST, K.: Spermatocytic seminoma. I. Clinicopathologic study of six cases and review of the literature, Cancer 24: 92 (1969). 23. GILBERT, G. B.: Studies in malignant testes tumors, tumors developing after orchidopexy: report of two cases and review of sixty-three, J. Urol. 46: 740 (1941). 24. GOEL, R. G., and SAMUEL, K. C.: Bilateral seminoma in a male pseudohermaphrodite, ibid. 108: 466 (1972). 25. BRECKENRIDGE, R. L., NASH, E., and LITZ, J.: Gonadoblastoma in male pseudohermaphroditism, Exp. Med. Surg. 27: 330 (1969). 26. SCULLY, R. E. : Gonadoblastoma. A review of 74 cases, Cancer 25: 1340 (1970). 27. EARLL, J. M., NEWMAN, S. G., and Dr RAIMONDO, V. C. : Bilateral testicular tumors in untreated congenital adrenocortical hyperplasia, J. A. M. A. 209: 937 (1969). 28. CARNEY, J. A., KELALIS, P. P., and LYNN, H. B.: Bilateral teratoma of testis in an infant, J. Pediatr. Surg. 8: 49 (1973). 29. STRAHLBERG, M., and BROWN, J. S.: Concomitant bilateral epidermoid cysts ofthe testes, J. Urol. 109:434 I197.11 ,__._,,

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