Introduction

Testicular cancers are rare clinical conditions and account for approximately 1-2% of all male malignancies (1). In 2020, 9.610 new testicular tumors were estimated to be diagnosed in the United States, with 440 deaths due to this malignancy (2). Therefore, the Sertoli cell testicular tumor is a rare malignancy that constitutes approximately 1% of testicular cancers (3) and is observed at all age intervals. However, clinicians encounter patients with Sertoli cell tumors mostly in the third or fourth decades of their lives. Unilateral testicular mass with a slow growth pattern is the most common symptom of patients who present to urology departments (4). Herein, presented a case of Sertoli cell testicular tumor (not otherwise specified) in a patient who presented with complaints of a slowly growing painless mass in the right testicular region for 2 years.

Case Report

A 28-year-old male patient visited our clinic with a painless mass in the right testicle showing a slow growth pattern for 2 years. The genitourinary system examination revealed a mass of approximately 2.5 cm in a hard and fixed character, with clear boundaries. The patient had no history of scrotal trauma, urological surgery, or chronic disease. Additionally, a detailed general examination of the patient did not yield any pathological indication for systemic diseases, such as gynecomastia, lymphadenopathy, or hyperpigmentation. No abnormal values were detected in routine hematologic and biochemical parameters. Further, testicular tumor markers, such as serum lactate dehydrogenase (31 IU/L), a-fetoprotein (4.14 IU/mL), and b-human chorionic gonadotropin (1.02 mIU/mL), were within the normal limits. B-mode ultrasonography revealed a well-bordered, round, and solid mass lesion with echogenic areas inside the right testicle. The mass was observed in Doppler ultrasonography as a heterogeneous hypoechoic solid lesion in 29´21´20 mm dimensions with vascularization in places inside and in its periphery (Figure 1a, b). The patient underwent right high inguinal orchiectomy. Tissue samples were analyzed by two pathologists in the pathology department. Before the pathologic examination, specimens obtained from the patient were first macroscopically evaluated. Then, tissue samples were formalin-fixed, paraffin-embedded, and 4 µ sections were obtained using a tissue microtome. Sections were stained with hematoxylin and eosin and analyzed under an upright light microscope. Immunohistochemical analyses were performed using a Leica Bond Max immunostainer. All sections were then analyzed under light microscopy, which revealed a tumor infiltration in testicular parenchyma. The infiltrative pattern was both solid and tubular. Tumor cells had relatively uniform nuclei with small nucleoli and a large pale eosinophilic cytoplasm with indistinct borders. They lied in different directions and had a solid, trabecular, and insular architecture. The solid counterpart was predominant; however, slightly elongated tubular structures were also observed in border areas between the tumor and testicular parenchyma. Tubules were lined in a single layer of cuboidal/columnar cells suggesting Sertoli cells. Mitosis counts were >5 in 10 high power fields. Necrosis was absent (Figure 2a, b, c). The testicular tumor was diagnosed as a “Sertoli cell tumor not otherwise specified” type (Figure 3a, b, c). Thoracic and abdominopelvic computed tomography scan revealed no evidence of lymphatic or distant metastases. The follow-up was uneventful in 30 months. This case report was written after obtaining patient consent.

Discussion

Testicular cancer can be derived from any testicular cell type; however, >95% of testicular cancers are germ cell tumors (5). Seminoma is the most frequent germ cell tumor, constituting approximately 60% of all germ cell neoplasms (6). Sex cord-stromal tumors account for approximately 4% of all testicular cancers. Sex cord-stromal tumors (also known as, “androblastoma”) are a spectrum of tumors, which has a differentiation of pure sex cord morphology in one end and pure stromal morphology in the other end, mostly composed of various proportions of both components. Sex cord-stromal tumor includes cells, which show fetal, pubertal, or adult Sertoli cell features (7,8). The pathological analyses categorized them into four groups: Leydig cell tumors, Sertoli cell tumors, granulosa cell tumors, and unclassified. Sertoli cell tumors are rarely observed pathologies and account for 0.4-1.5% of these tumors (9). The most common symptom is a painless and slow-growing testicular mass. Findings may also include gynecomastia and impotence due to hormonal changes (3,9). Studies with large series observed these hormonal changes in an average of 11-25% of cases (10). Concurrently, reports associated these pathologies with genetic syndromes, such as Carney syndrome and Peutz-Jeghers syndrome. This association is more common in bilateral or multiple Sertoli cell tumors (3). Sertoli cell tumors are mostly of benign characters; however, an in-depth literature analysis shows cases with malignant behavior. Generally, the aggressive attitude was estimated to be observed in 10-22% of these tumors (11).Sertoli cell testicular tumors are rarely observed, thus no strict follow-up and treatment protocols are available. The basic treatment approach includes radical inguinal orchiectomy. Currently, many authors report an appropriate evaluation of testicular-sparing surgery among treatment options, as the clinical course is extremely slow in most of these cases. The histopathological tumor features, such as the size of the mass, necrosis, lymphovascular invasion, and pleomorphism, are shown to be very closely related to the clinical course of the disease. Evaluating and analyzing patients who underwent testicular-sparing surgery with testicular ultrasonography is critical in terms of local recurrence and with chest, abdomen, and pelvic imaging to see any distant organ metastases. Grogg et al. (12) reported that 13% of patients underwent testicular-sparing surgery, local recurrence in only 1 patient during the 3-month follow-up, and 1% rate of contralateral recurrence in their meta-analysis of 435 cases. Testicular-sparing surgery was not performed since our patient’s social facilities were extremely limited and he lived in a remote location to health centers, which would make his regular follow-ups difficult. However, various aggressive treatment strategies, such as retroperitoneal lymph node dissection, chemotherapy, and radiation therapy combinations, are applied in cases that show remote organ dissemination (13). Survival rates for 1 and 5 years were estimated as 93 and 77% for Stage 1 Sertoli cell tumors, respectively (11). Contrarily, late metastases were reported in some cases at stages up to 10 years (14).The World Health Organization classified this type of cancer in three categories: Sertoli cell tumor not otherwise specified, large cell calcifying Sertoli cell tumors, and sclerosing Sertoli cell tumors (15), of which the most common type is the Sertoli cell tumor not otherwise specified. Young et al. (8) conducted a large series of 60 cases with Sertoli cell tumor not otherwise specified. The average age of patients was 45 years. All tumors were unilateral and with an average size of 3.6 cm (range 0.3-15 cm). The same study reported the presence of metastatic disease by 6.7% at the time of arrival. Sertoli cell tumor not otherwise specified is located in testicular parenchyma, well-circumscribed, and has lobulated contours. The cut surface is solid, yellowish in color, and may contain foci of hemorrhage but necrosis is rare. Tumor cells are arranged in a solid or tubular fashion, and a reticular pattern of growth may be seen. These cells have uniform round or oval nuclei and pale eosinophilic cytoplasm. Atypia and pleomorphism among tumor cells are rare. A fibrous hyalinized stroma-containing dilated vascular structure may be seen between tumor islands. Mitoses are not frequent (<5 mitoses per 10 HPF) (7,8).

Therefore, considering rare pathologies, such as Sertoli cell tumors, among the pre-diagnoses for effective management of follow-up and treatment strategies for patients with painless testicular mass is crucial.

Acknowledgements

Publication: The results of the study were not published in full or in part in form of abstracts.

Contribution: There is not any contributors who may not be listed as authors.

Conflict of Interest: No conflict of interest was declared by the authors.

Financial Disclosure: The authors declared that this study received no financial support.

Ethics

Informed Consent: This case report was written after obtaining patient consent.

Peer-review: Externally peer-reviewed.

Authorship Contributions

Concept: E.K., M.B., N.U., D.A., Design: E.K., M.B., N.U., D.A., Data Collection or Processing: E.K., F.A.D., F.E., Analysis or Interpretation: F.E., Literature Search: F.A.D., N.U., Writing: E.K.