Testicular gonadal stromal (sertoli cell) tumor

TESTICULAR GONADAL STROMAL (SERTOLI CELL) TUMOR

MATTHIAS I . OKOYE, M .D .

BARRY L . DEWITT, M .D .

WILLYS F. MUELLER, JR ., M .D .

CATHY O . BLIGHT, M .D .

CHENG-YANG CHANG, M .D .

JAMES M . LAU, M .D .

DAVID E . CONGDON, M .D . From the Departments of Pathology and Surgery, Hurley Medical Center, Flint, Michigan

ABSTRACT-Eighty-one cases of testicular gonadal stromal tumor have been reported in the literature . An additional case is herein reported . The pathologic, immunohistologic, and histogenetic aspects are presented .

Primary testicular tumors originate from (1) germ cells, (2) gonadal stroma, (3) ductal system, fibrovascular stroma and capsule . Ninetythree per cent of these tumors arise from germ cells, 6 per cent from primitive gonadal stroma, and only about I per cent from the other elements . Mostofi and Price' classify the gonadal stromal tumors as follows : (1) tumors of specialized gonadal stroma (Leydig cell tumors, Sertoli, granulosa-theca cell tumors, undifferentiated or primitive tumors, and combinations of these), and (2) tumors showing both germ cell and gonadal stromal elements (gonadoblastoma) . Gonadal stromal tumors are more frequent in infants and prepubertal children and comprise about 19 per cent of all tumors in this age group, especially among infants younger than six months of age . Case Report A four-month-old white male was seen in October, 1981, with an enlarged left testicle, first noticed by the mother two weeks prior to admission . There had been no evidence of pain or discomfort . Gynecomastia was absent . Chest and abdominal x-ray films as well as excretory urograms were within normal limits . Hematologic workup and chemical profiles were also

184

normal . A left radical inguinal orchiectomy with high ligation of the spermatic cord was performed . Grossly, the tumor was yellow-tan and firm with tiny cystic and hemorrhagic areas, especially beneath the tunica, and measured 2 .0 cm in greatest diameter . Postoperatively the CT scan of retroperitoneal space and serum testosterone determination were done, and findings were normal . Light Microscopic Features On hematoxylin and eosin stains, the tumor was very cellular and demonstrated a mixed pattern consisting of areas of spindle-type cells as well as areas of cells with round or oval nuclei . The tumor cells did not exhibit distinct cell boundaries . There were also cystic structures, containing a proteinaceous fluid, reminiscent of granulosa cell tumors of the ovary. In other areas the tumor exhibited hemorrhagic cystic degeneration . Numerous mitotic figures were also present in the tumor. The portions of the spermatic cord, epididymis, and rete testis in the specimen were not involved by the neoplasm . Special stains with periodic acid-Schiff (PAS), mucicarmine, and oil-red 0 were negative . The histologic diagnosis of gonadal stromal (Sertoli cell) tumor made, based on the

UROLOGY / FEBRUARY 1985 / VOLUME XXV, NUMBER 2



(A) Low-magnification photomicrograph

FIGURE 1 .

kt shows tumor cells closely packed with microcystic

structures . (B) Another area of tumor shows cystic structure lined by flattened cells and containing eosinophilic material and suggestive of granulosa cell tumor . (C) Higher magnification of tumor shows solid sheets of cells with indistinct cell borders and delicate fibrous stroma . Mitoses were present (arrow) . Note variation in staining of cells . (D) Higher magnification of tumor shows spindle-shaped cells. Some cells exhibit foamy cytoplasm . A similar picture may be seen in gonadoblastoma . (Hernatoxylin and eosin x 50, x 50, x 450, and x450, respectively .)

hematoxylin and eosin and special stains, was confirmed by the AFIP, Washington, D .C . (Fig . 1) . Because of the high mitotic rate in this neoplasm, we believed that this tumor had malignant potential in this patient .

antigen, and estradiol . * The immunoperoxidase staining showed no positive reactions in sections of the tumor. Comment There has been long-standing confusion about the correct nomenclature and histogenesis of testicular gonadal stromal tumors . These tumors have been reported in the literature under various names, including tumors of the specialized gonadal stroma,' androblastoma,s granulosa-cell tumor, 6 Sertoli cell tumor,' and sex-cord stromal tumors . 8 Our case demonstrates a mixed histologic pattern with no evidence that the tumor cells are producing estrogen or any oncofetal antigens, such as

Immunoperoxidase Studies To elucidate specific tumor markers in this neoplasm, we decided to examine 5 ft thick sections of the formalin-fixed, paraffin-embedded tissue using the unlabeled antibody peroxidaseantiperoxidase technique for the histologic demonstration of alpha-fetoprotein, carcinoembryonic antigen (CEA), and estradiol . 2-4 The peroxidase-antiperoxidase method was carried out in the standard fashion utilizing control series prepared in parallel, with rabbit antiserum to alpha-fetoprotein, carcinoembryonic

UROLOGY

/

FEBRUARY

1985

/

VOLUME

XXV

NUMBER

`T nmutok . Inc ., Carpinteria, California 93013 .

2

185

carcinoembryonic antigen or alpha-fetoprotein, as shown by the immunoperoxidase studies . Furthermore, lack of excessive estrogen production, viz ., gynecomastia, was clinically absent . Holtz and Abel19 in their series preferred to restrict the term "granulosa cell tumors" only to lesions that occurred in patients who are genetically female, and the term "specialized gonadal stromal tumors" to most of the other lesions exclusive of those of interstitial cell origin . Mostofi and Price' believed that these tumors actually arise from the primitive gonadal stroma mesenchyme . Our findings support these two views . With regard to the biological behavior, the criteria for determining malignancy in these tumors have not been clearly defined . Polymorphism and increased mitoses are, however, the two most reliable criteria ." These two features are demonstrated by our case . The incidence of metastasis is estimated to be about 10 per cent, and almost all metastasis developed within five months . Our patient had radical orchiectomy and demonstrates no metastatic disease after six months .

186

Flint, Michigan 48502 (DR . OKOYE) ACKNOWLEDGMENT . To Lucia Pacheco for her secretarial assistance and Julio E . Badin, M .D ., of Genesee Memorial Hospital, Flint, Michigan, who assisted with the photomicrographs .

References 1 . Mostofi FK, and Price EB Jr : Tumors of the male genital system, in : Atlas of Tumor Pathology, Washington, D .C ., Armed Forces Institute of Pathology, 1973, series 2, part 8, p 2 . 2 . Taylor CR : Immunoperoxidase techniques, Arch Pathol Lab Med 102 : 113 (1978) . 3 . DeLellis RS, et al : Immunoperoxidase techniques in diagnostic pathology . Am J Clin Pathol 71 : 483 (1979) . 4 . Taylor CR, et al : The potential value of immunohistologic techniques in classification of ovarian and testicular tumors, Human Pathol 9 : 417 (1978) . 5 . Teilum G : Homologous tumors in ovary and testes . Contribution to classification of gonadal tumors, Acta Ohstct Gynccol Scand 24 : 480 (1944) . 6 . Laskowski J : Feminizing tumors of the testes, Endokr Pol 3 : 337 (1942) . 7 . Collins DH, and Symington T : Sertoli cell tumor, Br J Urol 36 : 52 (1964) . 8 . Marshall FF, Kerr WS Jr, Kliman B, and Scully RE : Sex cord-stromal (gonadal stromal) tumors of the testis : a report of 5 cases, j Urol 117 : 180 (1977) . 9 . Holtz F, and Abell MR : Testicular neoplasm in infants and children : tumors of non-germ cell origin, Cancer 16 : 982 (1963) . 10 . Mostofi FK, and Price EB Jr : Op cit,' p 110 .

UROLOGY i FEBRUARY 1985

1

VOLUME XX\; NUMBER 2