Large cell calcifying Sertoli cell tumor of the testis in a boy with brucellosis

Abstract

We report the case of a testicular tumor in a boy with recurrence of brucellosis. Although the most probable diagnosis seemed to be genito-urinary complications of brucellosis, the mass proved to be a testicular tumor the large cell calcifying Sertoli cell type, coexisting with the brucellosis. Case reportA 12-year-old boy was admitted to the pediatric department of P and A Kyriakou Children's Hospital, Athens with a 20 day history of pain to the right lower limb and a 14 day history of low-grade fever. Six months prior to his admission, he was treated for brucellosis, with rifampicin and doxycycline for a period of 6 weeks, at the local hospital of his town. Upon admission physical examination revealed infl ammation of the metatarsophalangeal joint of the big toe of the right foot. The whole left testis was moderately swollen and a well-defi ned, fi rm and painless nodule was palpated at its upper pole. Ultrasonography revealed a well-defi ned mass 16 × 12 mm in size with calcifi cations, located in the upper pole of the left testicle ( Fig. 1). The echogenicity of the remainder of the left and the right testicle appeared normal. Computed tomography (CT) of the genitalia and abdomen confi rmed the calcifi cations in the upper pole of the left testicle. The epididymis, as shown on the ultrasonography and the CT scan, was normal without any surrounding fl uid collection. There was no evidence of extratesticular extension. Orchitis/orchiepididymitis due to brucellosis or a testicular tumor were considered. Values of follicle-stimulating hormone , testosterone, ␣ -fetoprotein and ␤ -human chorionic gonadotropin were normal. A relapse of brucellosis was confi rmed by the isolation of Brucella melitensis from blood cultures. Bone scan fi ndings were in keeping with osteomyelitis of the metatarsal bone of the big toe of the right foot as well as of the ipsilateral navicular bone. The patient was treated with streptomycin for 14 days, tetracycline and rifambicin for 6 weeks. Regarding the testicular mass, which remained unchanged, surgical exploration through an inguinal incision and excision of the nodule were performed. The excised nodule was 1.2 cm in greatest dimension. Microscopic examination of the mass revealed clusters of tubules composed of neoplastic cells separated by a connective tissue stroma, which was infi ltrated by a varying number of lymphocytes and plasma cells ( Fig. 2). The tumor cells had uniform round nuclei and abundant eosinophilic cytoplasm containing granules. Between tumor cells there were small laminated deposits of calcifi ed amorphous material ( Fig. 3). Mitoses were rare. The histological fi ndings were consistent with large cell calcifying Sertoli cell tumor (LCCSCT). Neoplastic cells reached the cut surface. Taking into account that the tumor was unilateral and had a fi nite but existing risk of metastasis we proceeded to a radical left orchiectomy with high ligation of the spermatic cord. The testis that was submitted to pathologists for histological examination was 3 cm in diameter wi...

Large Cell Calcifying Sertoli Cell Tumor

“…The LCCSCT, one of the rarest sex cord-stromal tumors of the testis, was initially described by Lange et al6 in 1960 and then formally introduced as a distinct entity by Proppe and Scully in 1980 5. To date, 45 clinically benign,1,5,9,12,25,27,29–45 53 clinically ambiguous,1,16,27,36,38,43,46–78 and 17 clinically malignant1–3,5,23–28 cases have been reported in the English language literature, including this series, according to our comprehensive review (Tables 2, 3, Supplementary Table 1, Supplemental Digital Content 1, http://links.lww.com/PAS/B288).…”

“…The LCCSCT, one of the rarest sex cord-stromal tumors of the testis, was initially described by Lange et al 6 in 1960 and then formally introduced as a distinct entity by Proppe and Scully in 1980. 5 To date, 45 clinically benign, 1,5,9,12,25,27,[29][30][31][32][33][34][35][36][37][38][39][40][41][42][43][44][45] FIGURE 1. There are multiple, solid, tan, encapsulated, and abutting nodules on the cut surface of this testis from a patient with the Carney complex.…”