A Case Report: Ovarian Sertoli-Leydig Cell Tumor With Hyperestrogenism and Endometrial Hyperplasia in a Postmenopausal Woman

Abstract

The patient was a 66-year-old woman, G2P2. The patient presented a chief complaint of irregular postmenopausal bleeding 1 month ago. A transvaginal ultrasonography showed that bilateral ovaries were not enlarged and uterine endometrium was thickened, measuring at 9 mm. As a result of endometrial curettage, the simple endometrial hyperplasia was revealed. A blood examination showed an elevated estradiol level of 67 pg/mL, an elevated level of testosterone 0.64 ng/mL, and a slightly suppressed follicle-stimulating hormone (FSH) level of 34.86 mIU/mL. We conducted laparoscopic hysterectomy and bilateral salpingo-oophorectomy because the patient strongly suggested less invasive surgery. The result of pathological diagnosis was Sertoli-Leydig cell tumor (SLCT) in moderately differentiation. A blood examination after a month postoperatively revealed an elevated FSH level of 85.59 mIU/mL, depressed estradiol level of less than 10 pg/mL, and testosterone level of less than 0.03 ng/mL. There was no evidence of recurrence in the first year of follow-up.

An 88-year-old woman with flushing, alopecia and hirsutism and a Sertoli-Leydig cell tumour

“…Around 50% of SLCT cases come to clinical attention due to the latter manifestations of progressive masculinisation,4 as was seen in our patient. It has been reported that when the value of testosterone, mainly secreted by the Leydig cells of the tumour, exceeds 2 ng/mL, there is obvious evidence of virilisation 7. Our patient presented with initial total testosterone values as high as 1307 ng/mL, thus explaining many of her signs and symptoms of increased androgens.…”

“…These tumours are composed of a mixture of cells that morphologically resemble male Sertoli and Leydig cells and are found to be unilateral in 98% of patients 7 8. They mainly secrete testosterone, which can cause endocrine manifestations—seen in about half of the patients—of virilisation to appear 7. The clinical characteristics of SLCT have been found to be associated with the degree of histological differentiation,5 and these clinical signs and symptoms include that of defeminisation (amenorrhoea, oligomenorrhoea and breast atrophy) followed by that of progressive masculinisation (acne, deepening of the voice, hirsutism, temporal balding and clitoromegaly) 5 9.…”

“…While virilisation and defeminisation symptoms have been described in as high as 77% of cases,7 some reports have suggested signs and symptoms of hyperoestrogenism associated with SLCT, such as endometrial hyperplasia, abnormal uterine bleeding and menorrhagia/metrorrhagia 7 10. These symptoms were not seen in our patient, despite her slightly elevated oestradiol levels for a postmenopausal woman and proliferative endometrium on histology postresection.…”

“…This procedure was deemed sufficient, as pathology did not reveal malignant potential, such as poor differentiation, presence of heterologous elements, high mitotic counts or rupture of the tumour on pathology, and thus did not require further surgical staging 9 11. Furthermore, the choice to proceed with laparoscopy is suggested to be useful in SLCT management and detection of metastasis, as it allows for peritoneal cavity observation and sampling 7. Nonetheless, prognosis of SLCT is generally good and there is typically prompt return of feminine characteristics with excision of the tumour—while disappearance of masculinisation features is frequently slower 9 12.…”

“…In addition to just performing surgery, the gold standard treatment of SLCTs,7 other treatment options include surgery with adjuvant chemotherapy and/or radiotherapy. This more aggressive therapy is indicated for advanced stage tumours and those with poor prognostic features mentioned above 9.…”