Unusual Differential Diagnosis of Testis Tumor: Intrascrotal Sarcoidosis

Unusual Differential Diagnosis of Testis Tumor: Intrascrotal Sarcoidosis

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0022-534 7/92/14 74-1112$03.00/0 THE JOURNAL OF UROLOGY Copyright© 1992 by AMERICAN UROLOGICAL ASSOCIATION, INC.

Vol. 147, 1112-1114, April 1992

Printed in U.S.A.

UNUSUAL DIFFERENTIAL DIAGNOSIS OF TESTIS TUMOR: INTRASCROTAL SARCOIDOSIS A. J. GROSS, H. HEINZER, V. LOY

AND

K.-P. DIECKMANN

From the Department of Urology, Institute of Pathology, Steglitz Medical Center, Free University of Berlin, Berlin, Federal Republic of Germany

ABSTRACT

We report 2 cases of sarcoidosis with extrapulmonary manifestations in the testis, epididymis and spermatic cord. Each patient presented with an intrascrotal mass of unknown origin that suggested a testicular tumor. Sarcoidosis was confirmed in caseTI:iy radical orchiectomy and further small sarcoid foci were detected in 1 of the lacrimal glands. In case 2 sarcoidosis was confirmed by biopsy and there were no further extrapulmonary indications. Both cases were stage II disease so no specific therapy was applied. The patients were without signs of progression at 2 and 10 years, respectively, after initial diagnosis. KEY WORDS:

testicular neoplasms, sarcoidosis, testis, scrotum

Differential diagnosis of any intrascrotal mass must always take into account the possibility of a testicular tumor. Germ cell tumors constitute 90% of all malignant intrascrotal neoplasms and extragonadal neoplasms rarely metastasize to the scrotal structures. 1 Although sarcoidosis was first described by Boeck as early as 1869 and extrapulmonary symptoms are common, 2 intrascrotal manifestations have been observed in only 16 cases. 3- 17 Sarcoidosis, a systemic granulomatosis, is the most frequent interstitial lung disease. 18 We report 2 cases of sarcoidosis with intrascrotal manifestations. CASE REPORTS

Case 1. A 33-year-old man of Algerian descent had hilar enlargement and pulmonary infiltration diagnosed during a routine screen of the chest (fig. 1). Sarcoidosis was confirmed by bronchial and transbronchial lung biopsy after exclusion of tuberculosis. Further physical examination revealed an indolent enlargement and induration of the right scrotal contents. Results of laboratory measurements, especially of a-fetoprotein and {1-human chorionic gonadotropin, were within the normal range. The blood group was 0. Testicular sonography showed a highly echogenic solid mass associated with the testis and epididymis (fig. 2). During inguinal exposure a solid, white mass affecting the entire epididymis and part of the testis was found (fig. 3). Histological evaluation of the orchiectomy specimen revealed severe epididymitis, periorchitis and orchitis (fig. 4). In the fatty tissue of the spermatic cord a granulomatous reaction extending to the resection margin was found. The granulomas were characterized by epithelioid cell nidi containing individual giant cells. Microorganisms, including acid-fast bacilli, were not detected. The structure of the granulomas in conjunction with the clinical findings was in accordance with sarcoidosis. Gallium scintigraphy and abdominal computerized tomography revealed additional small sarcoid lesions in the lacrimal glands that indicated stage II sarcoidosis, so no specific therapy was indicated. After 2 years of followup the patient had no signs of progression. Case 2. Sarcoidosis confined to the lung was diagnosed in a 26-year-old man who had undergone orchiopexy approximately 15 years ago because of testicular maldescent. The patient presented at the age of 35 years with an induration in the right scrotal contents. A tumor was suspected and inguinal exposure was performed. The spermatic cord was surrounded by scar tissue but the testis did not appear to be tumorous. Only an induration of the tunica albuginea was palpable. After removal Accepted for publication September 6, 1991.

of the scar tissue from the spermatic cord the nodular induration of the tunica albuginea was resected. The lesion was 1.5 cm. in diameter and it was on the side of the tunica. Histological evaluation of the specimen revealed extensive small or medium sized granulomas consisting of epithelioid cells and giant cells. The giant cells contained several unevenly distributed nuclei, some with a horseshoe-like arrangement in the periphery. Langhans' giant cells were also observed. The findings corresponded to granulomas of the sarcoidosis type. Acid-fast bacilli were not detected. No abnormalities were found during further physical examination. In particular, the laboratory values, including complete blood count, transaminases, alkaline phosphatase, serum calcium and electrophoresis, were within normal ranges. The blood group was A. The urine was sterile, tine tuberculin test was negative and no acid-fast bacilli were found in the tine tuberculin culture. Electrocardiography and chest x-ray were appropriate for patient age and revealed no pathological conditions. The stage II lung sarcoidosis diagnosed 9 years earlier was no longer identifiable. No specific therapy had been applied at that time and on this occasion the patient was discharged from the hospital without further treatment. There were no signs of recurrence or progression 10 years later. DISCUSSION

Sarcoidosis is a systemic granulomatous disease of unknown origin that can affect any part of the body. 3 Its prevalence, which is variable, is approximately 1 to 6 cases per 1,000 persons worldwide. 19 In 84 % of the cases the disease is localized in the chest. 20 Manifestations ranging from granulomas to tumors are also found in other organ systems. 21 The urogenital system is affected in 0.2% of all clinically diagnosed cases and in 5% of those diagnosed at autopsy. 14• 22 In most cases with urogenital involvement the condition is confined to the kidneys. The first case of testicular sarcoidosis was described by Nickerson in 1937, 23 and there have been 16 cases described as of 1990. 3 • 10• 15• 17• 23 Thus, when intrascrotal masses are encountered in patients with confirmed sarcoidosis the differential diagnosis must consider the possibility of extrapulmonary manifestations. There are reports of a more than coincidental concurrence of sarcoidosis and testicular tumor. 24 • 25 Sarcoidosis and testicular cancer have their peak incidence in patients in their twenties. 18 In 1 case sarcoidosis followed chemotherapy for a testicular tumor 26 and in 1 a testicular tumor developed in the presence of sarcoidosis. 27 No malignant tumor was found in our 2 cases, although patient age and palpatory findings were suggestive in both.

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FIG. 1. Case 1. Chest x-ray with bilateral hilar enlargement and pulmonary infiltration.

FIG. 3. Case 1. Macroscopic aspect of orchiectomy specimen shows white solid mass replacing entire epididymis with affliction of testis in cranial part.

FIG. 2. Case 1. Duplex sonography of affected testis with highly echogenic solid mass in cranial area of scrotum.

Epidemiological studies have shown that black patients and patients with blood group A are more likely than others to have sarcoidosis. 13 • 19 One of our 2 patients is black, although only 1 % of the German population is black, and 1 has blood group A. Only 1 patient with increased a-fetoprotein in intratesticular sarcoidosis has been reported. 28 Our cases show that intrascrotal masses observed in young men might be secondary to nonneoplastic disease, which should be considered during differential diagnosis, especially in light of the clinical findings. REFERENCES 1. Dieckmann, K.-P., Due, W. and Loy, V.: Intrascrotal metastasis of

renal cell carcinoma. Case reports and review of the literature. Eur. Urol., 15: 297, 1988. 2. Gross, W. L. and Schlaak, W.: Vaskulitis und Granulomatose im

FIG. 4. Case 1. Histological section of orchiectomy specimen with typical epithelioid nidi and individual giant cells with original H & E stain. Reduced from X400. Bereich der Atemwege. Immun. Infekt., 16: 125, 1988. 3. Krauss, L.: Genital sarcoidosis: case report and review of the literature. J. Urol., 80: 367, 1958. 4. Hausfeld, K. F.: Primary sarcoidosis of the scrotum: case report. J. Urol., 86: 269, 1961. 5. McGowan, A. J., Jr. and Smith, E. M.: Urological implications of sarcoidosis. J. Urol., 97: 1090, 1967. 6. Chowdhury, S. D. and Higgins, P. M.: Sarcoid of the testis. Brit. J. Urol., 45: 218, 1973. 7. Opal, S. M., Pittman, D. L. and Hofeldt, F. E.: Testicular sarcoidosis. Amer. J. Med., 67: 147, 1979. 8. Torrington, K. G., Lewis, P. G., Tellis, C. J. and Swedarsky, R.H.: Systemic sarcoidosis presenting with a testicular nodule. South.

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Med. J., 72: 1001, 1979. 9. Wees, S. J.: Testicular sarcoidosis. South. Med. J., 74: 255, 1981. 10. Amenta, P. S., Gonick, P. and Katz, S. M.: Sarcoidosis of testis and epididymis. Urology, 17: 616, 1981. 11. Seaworth, J. F., Davis, S. J. and Donovan, W. N.: Aggressive

delberg: Alfred Hu.thig Verlag, p. 18, 1965. 20. Roos, N., Bick, U., Vassallo, P., Diederich, S., Muller-Miny, H. Auffermann, W., Erlemann, R. and Peters, P. E.: Thorakale Sarkoidose. Radiologe, 30: 581, 1990. 21. Singer, E. P., Hensler, N. M. and Flynn, P. F.: Sarcoidosis: an

diagnostic approach indicated in testicular sarcoidosis. Urology, 21: 396, 1983. McWilliams, W. A., Abramowitz, L. and Tiamson, E. M.: Epididymal sarcoidosis: case report and review. J. Urol., 130: 1201, 1983. Haas, G. P., Badalament, R., Wonnell, D. M. and Miles, B. J.: Testicular sarcoidosis: case report and review of the literature. J. Urol., 135: 1254, 1986. Turk, C. 0., Schacht, M. and Ross, L.: Diagnosis and management of testicular sarcoidosis. J. Urol., 135: 380, 1986. Hackney, R. L., Jackson, A. G. and Worrell, R. G.: Sarcoidosis of report. J. Natl. Med. Ass., 78: 63, 1986. Parr, M. J. A. and Williams, M. V.: Sarcoidosis mimicking metastatic testicular tumour. Brit. J. Rad., 61: 516, 1988. Singer, A. J., Gavrell, G. J., Leidich, R. B. and Quinn, A. D.: Genitourinary involvement of systemic sarcoidosis confined to testicle. Urology, 35: 442, 1990. Barth, J. and Gross, W. L.: Sarkoidose aus der Sicht des Internisten. Med. Welt., 41: 37, 1990. Jorgensen, G.: Untersuchungen zur Genetik der Sarkoidose. Hei-

analysis of forty-five cases in a large military hospital. Amer. J. Med., 26: 364, 1959. Ricker, W. and Clark, M.: Sarcoidosis: a clinico-pathologic review of 300 cases including 22 autopsies. Amer. J. Clin. Path., 19: 725, 1949. Nickerson, D. A.: Boeck's sarcoid: a report of six cases in which autopsies were made. Arch. Path., 24: 19, 1937. Blacher, E. J. and Maynard, J. F.: Seminoma and sarcoidosis: an unusual association. Urology, 26: 288, 1985. Droz, J. P., Ruffie, P., Piot, G., Ghosn, M., Caillaud, J.-M., Elias, D., Perrin, J.-L. and Levasseur, P.: Sarcoidosis and testicular germ cell tumor. Case report. Scand. rtJrol.-Nephrol., 24: 171, 1990. Gefter, W. B., Glick, J. H., Epstein, D. M. and Miller, W. T.: Sarcoidosis: a cause of intrathoracic lymphadenopathy after treatment of testicular carcinoma. AJR, 139: 820, 1982. Colebunders, R.: Sarcoidosis after testicular carcinoma treatment. AJR, 140: 831, 1983. Sieber, P. R. and Duggan, F. E.: Sarcoidosis and testicular tumors. Urology, 31: 140, 1988.

12. 13. 14. 15. 16. 17. 18. 19.

22. 23. 24. 25.

the fesfis:acase

26. 27. 28.

0022-534 7/92/14 7 4-1114$03.00 /0 THE JOURNAL OF UROLOGY Copyright© 1992 by AMERICAN UROLOGICAL ASSOCIATION, INC.

Vol. 147, 1114-1116, April 1992

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EPIDIDYMITIS FROM ENTEROBIUS VERMICULARIS: CASE REPORT G. KOLLIAS, M. KYRIAKOPOULOS

AND

G. TINIAKOS

From the Department of Urology, N.I.M. T.S. Hospital, Athens, Greece

ABSTRACT

A rare case of epididymitis caused by Enterobius vermicularis, a pinworm, is reported. A 52-yearold man underwent resection of the right epididymis and histological examination confirmed the diagnosis of pinworm infection by discovering parts of the body of the oxyurid in various sections. KEY

WORDS: epididymis, epididymitis, Enterobius, parasitic diseases, pinworms

Enterobius vermicularis is distributed worldwide and is the most common cause of helminthic infection. Man is the only host for the parasite. The most common symptom and sign is pruritus of the perianal area, particularly at night. Insomnia, restlessness, enuresis and irritability are also common symptoms especially in children. Anorexia, abdominal pain, nausea and vomiting have also been attributed to this infection. The diagnosis depends upon finding adult worms in the stool on the perianal skin. Worms also migrate, although unusually, into the vagina, uterus, fallopian tubes and so forth. We present a rare case of epididymitis due to E. vermicularis. CASE REPORT

A 52-year-old white man was hospitalized with a swollen right epididymis. Family history was free of any similar findings. He reported no previous serious or chronic diseases, or previous surgical procedure except for the usual childhood diseases. The patient was a sailor by profession and was obliged to stay at various ports of the world, such as in Indochina, Egypt and Thailand. Six years before hospitalization, while he was in South America, the patient reported that he was voiding pinworms in the urine, for which he had taken prescribed medication. At examination the right epididymis was swollen and in places it was of firm consistency. The patient was afebrile and Accepted for publication August 12, 1991.

he was taking antibiotics (tetracycline) for 1 month because of the epididymitis. However, the symptoms were unchanged. There was constant tenderness in the right inguinal region. Routine laboratory studies showed mild eosinophilia, which was considered unimportant at that time. The urine culture was negative and an excretory urogram was free of any abnormalities. With the patient under spinal anesthesia the right epididymis was resected. During the operation inflammatory tissue was noted to cover the area between the skin of the scrotum, the testicular coats and the epididymis. Macroscopically, the epididymis was diffusely inflamed and it was removed completely. Convalescence was uneventful. Histological examination of the excised epididymis showed foci of inflammatory granulomatous tissue in some areas consisting mainly of leukocytes, lymphocytes and histiocytes. The centers of these granulomatous foci appeared to be necrotic and included 1 or several male pinworms on transverse section (diameter 250 to 350 µ., see figure). DISCUSSION

E. vermicularis is a pinworm that measures approximately 8 to 13 mm., is found in all climates and causes an intestinal infection in humans, who are the only hosts of the pinworm. The gravid female and male worms live with the heads attached to the mucosa of the cecum, appendix and adjacent areas of the bowel. The life span is 30 to 45 days. Many infected patients