A Sertoli-Leydig cell tumor (SLCT) is an extremely rare type of sex cord stromal tumor of the ovary, which mainly secretes testosterone, thus manifestations of hyperandrogenism commonly appear. This paper shall discuss a case of a postmenopausal woman who presented with pelvic organ prolapse, large left ovarian cyst and mild signs of hyperandrogenism. She underwent total abdominal hysterectomy with bilateral salpingo-oophorectomy, which on microscopic examination of the specimens, revealed a Mature cystic teratoma on the left ovary and an incidental finding of a well-differentiated SLCT, on the grossly normal-looking ovary. This histopathologic diagnosis of SLCT explained the patient’s hyperandrogenic characteristics. Authors likewise discussed the proper management of SLCT, including immunostaining and need for adjuvant chemotherapy.
Sertoli-Leydig Cell Tumor

A 51-year-old perimenopausal female patient presented with hirsutism and voice thickening which was started approximately one and a half years ago. Her initial hormone assay revealed elevated plasma testosterone, 5a-dihydrotestosterone, and dehydroepiandrosterone (DHEA) levels and therefore androgen-secreting tumor was first suspected. However, the lesion was inconspicuous on transvaginal sonography, abdominal-pelvic computed tomography (CT) scan, and pelvic magnetic resonance (MRI) imaging. Consequently, 18F-fluorodeoxyglucose (FDG) positron emission tomography-CT was performed, which localized the lesion as a focal FDG uptake within the right adnexa. Total laparoscopic hysterectomy with bilateral salpingo-oophorectomy was performed, and although visible gross mass lesions were not observed intraoperatively, pure Leydig cell tumor was pathologically confirmed within the right ovary. Plasma testosterone, 5a-dihydrotestosterone, and DHEA levels were normalized postoperatively. Clinical signs of virilization were also significantly resolved after 3-months of follow-up.
Dehydroepiandrosterone ; Diagnosis ; Electrons ; Female ; Follow-Up Studies ; Hirsutism ; Humans ; Hysterectomy ; Leydig Cell Tumor ; Middle Aged ; Ovary ; Plasma ; Sertoli-Leydig Cell Tumor ; Testosterone ; Virilism ; Voice

OBJECTIVE: To evaluate the clinical and pathologic characteristics of patients who were diagnosed with ovarian Sertoli-Leydig cell tumors (SLCTs) in a single institution. METHODS: The medical records of 11 patients who were pathologically diagnosed with SLCTs beginning in 1995 in a single institute was reviewed. RESULTS: The median patient age was 31 years (range, 16 to 70 years). Patient International Federation of Gynecology and Obstetrics stages were IA, IC, and IIB in 3 (27.3%), 6 (54.5%), and 2 (18.2%) patients, respectively. Six patients (54.5%) had grade 3 tumors, 3 patients (27.3%) had grade 2 tumors, and 1 patient (9.1%) had a grade 1 tumor. Four patients without children underwent fertility-sparing surgery, and 7 patients had full staging surgery, including a hysterectomy and bilateral salpingo-oophorectomy, with a laparoscopic approach used in 3. Eight patients underwent pelvic lymph node dissection, and 8 patients were administered adjuvant chemotherapy consisting of bleomycin, etoposide, and cisplatin in 6 cases, a modified bleomycin, etoposide, and cisplatin regimen in 1 case, and a combined paclitaxel and cisplatin regimen in 1 case. Two patients died of disease and were re-diagnosed with Sertoli form endometrioid carcinoma. The other patients remain alive without recurrence at the time of reporting. CONCLUSION: Our findings suggest that regardless of tumor stage or grade, ovarian SLCT patients have a good prognosis. Close observation and unilateral salpingo-oophorectomy would be beneficial for women who still wish to have children, while hysterectomy and bilateral salpingo-oophorectomy with adjuvant chemotherapy would be the optimal treatment in other cases. Furthermore, meticulous pathologic diagnosis is needed to develop a precise treatment strategy.
Bleomycin ; Carcinoma, Endometrioid ; Chemotherapy, Adjuvant ; Child ; Cisplatin ; Diagnosis ; Drug Therapy ; Etoposide ; Female ; Gynecology ; Humans ; Hysterectomy ; Lymph Node Excision ; Medical Records ; Obstetrics ; Ovarian Neoplasms ; Paclitaxel ; Prognosis ; Recurrence ; Sertoli-Leydig Cell Tumor*

A 50-year-old peri-menopausal woman presented with hard palpable mass on her lower abdomen and anemia from heavy menstrual bleeding. Ultrasonography showed a 13x12 cm sized hypoechoic solid mass in pelvis and a 2.5x2 cm hypoechoic cystic mass in uterine endometrium. Abdomino-pelvic computed tomography revealed a hypodense pelvic mass without enhancement, suggesting a leiomyoma of intraligamentary type or sex cord tumor of right ovary with submucosal myoma of uterus. Laparoscopy revealed a large Sertoli-Leydig cell tumor of right ovary with a very rare entity of intra-endometrial uterine leiomyoma accompanied by adenomyosis. The final diagnosis of ovarian sex-cord tumor (Sertoli-Leydig cell), stage Ia with intra-endometrial leiomyoma with adenomyosis, was made. Considering the large size of the tumor and poorly differentiated nature, 6 cycles of chemotherapy with Taxol and Carboplatin regimen were administered. There is neither evidence of major complications nor recurrence during 20 months' follow-up.
Adenomyosis/*diagnosis/drug therapy ; Carboplatin/therapeutic use ; Female ; Humans ; Laparoscopy ; Leiomyoma/*diagnosis/drug therapy ; Male ; Menorrhagia ; Middle Aged ; Neoplasm Recurrence, Local ; Paclitaxel/therapeutic use ; Sertoli-Leydig Cell Tumor/*diagnosis/drug therapy ; Treatment Outcome ; Uterine Neoplasms/*diagnosis/drug therapy

OBJECTIVETo investigate the clinicopathological features of testicular malignant Leydig cell tumor (TMLCT) and improve the non-invasive diagnosis of the disease.

METHODSWe retrospectively analyzed the clinicopathological data on a case of TMLCT, detected the circulating tumor cells (CTC) in the peripheral venous blood, and reviewed the related literature.

RESULTSThe patient, a 47-year-old male, underwent radical orchidoepididymectomy under general anesthesia. Postoperative pathology confirmed the lesion to be TMLCT, which was mainly composed of Leydig cells and suspected with vessel carcinoma embolus. Immunohistochemistry showed the tumor cells to be positive for α-inhibin, Ki67, CD30, vimentin, EMA, and PLAP, but negative for CK, CK7, S100, CD10, SMA, Des, AFP, hCG, CEA, CK19, CD117, Oct-4, LCA, CD20, Pax-5, CD3, and CD43. Two CTCs were detected in the peripheral venous blood. The patient received 3 courses of chemotherapy for retroperitoneal multiple lymph nodes metastasis post-operatively. Subsequent CT imaging manifested no obvious reduction of the retroperitoneal lymph nodes and consequently the patient again underwent retroperitoneal lymphadenectomy and cryoablation. At 8 months after treatment, CT examination revealed notably enlarged retroperitoneal lymph nodes with the right adrenal gland evidently invaded.

CONCLUSIONTMLCT is an extremely rare sex-gonad stromal tumor with high malignancy and poor prognosis, and CTCs may be used for its early diagnosis and prognostic prediction.

Biomarkers, Tumor ; metabolism ; Humans ; Immunohistochemistry ; Leydig Cell Tumor ; drug therapy ; pathology ; surgery ; Lymph Node Excision ; Lymphatic Metastasis ; Male ; Middle Aged ; Neoplastic Cells, Circulating ; Prognosis ; Retrospective Studies ; Sex Cord-Gonadal Stromal Tumors ; drug therapy ; pathology ; surgery ; Testicular Neoplasms ; drug therapy ; pathology ; surgery

OBJECTIVETo observe the inhibitory effect of gefitineb on the proliferation and its inducing effect on the apoptosis of mouse I-10 Leydig testicular cancer cells in vitro.

METHODSWe treated I-10 Leydig testicular cancer cells of mice with gefitineb at 0, 1.25, 2.5, 5, 10, 20, and 40 µmol/L. Then we determined the inhibitory effect of gefitineb on the growth of the cells by MTT, detected their early and late apoptosis by Annexin V-FITC/propidium iodide double staining and Hoechst 33258 nuclear staining, respectively, and observed the expressions of apoptosis-related proteins Bcl-2, Bax and caspase 3/9 by Western blot.

RESULTSCompared with the blank control group, gefitineb significantly inhibited the proliferation of the I-10 cells at 10 and 20 µmol/L (P < 0.05). The survival rate of the cells was (32.4 ± 2.8)% (P < 0.01) and their early and late apoptosis rates were (26.7 ± 4.2)% and (59.33 ± 10.2)% in the 40 µmol/L group, significantly different from those in the control (P < 0.05 and P <0.01). In comparison with the blank control group, gefitineb at 10, 20, and 40 µmol/L increased the expression of pro-apoptotic protein Bax by (41.9 ± 7.1), (60.1 ± 9.8), and (69.0 ± 11.3)% (all P < 0.05), decreased that of apoptosis-inhibitory protein Bcl-2 by (50.3 ± 8.9), (63.9 ± 6.9), and (88.7 ± 13.9)% (all P < 0.05), and elevated that of the cleft proteins caspase-3 by (69.0 ± 6.9)% (P < 0.05), (71.5 ± 8.1)% (P < 0.05), and (110.9 ± 14.2)% (P < 0.01) and caspase-9 by (51.8 ± 4.9), (54.7 ± 6.7), and (43.8 ± 11.8)% (all P < 0.05).

CONCLUSIONGefitineb can increase the cytotoxicity of I-10 Leydig testicular cancer cells of mice and induce their apoptosis via the mitochondria-mediated apoptosis signaling pathway.

Animals ; Antineoplastic Agents ; pharmacology ; Apoptosis ; drug effects ; Apoptosis Regulatory Proteins ; metabolism ; Caspase 3 ; metabolism ; Caspase 9 ; metabolism ; Cell Proliferation ; drug effects ; Cell Survival ; Leydig Cell Tumor ; drug therapy ; metabolism ; pathology ; Male ; Mice ; Neoplasm Proteins ; metabolism ; Neoplasms, Germ Cell and Embryonal ; drug therapy ; metabolism ; pathology ; Quinazolines ; pharmacology ; Testicular Neoplasms ; drug therapy ; metabolism ; pathology ; bcl-2-Associated X Protein ; metabolism

Steroid cell tumors account for less than 0.1% of all ovarian tumors. There are three steroid cell tumor subtypes: steroid cell tumor not otherwise specified (NOS), stromal luteoma and Leydig cell tumor. Steroid cell tumor, NOS, is the most common type and has malignant potential. This report describes a case of an ovarian steroid cell tumor, NOS. A 35-year-old woman visited hospital with the complaint of metrorrhagia. Physical examination revealed increased pubic hair. Transvaginal ultrasound indentified a 4.9 x 3.4 cm, well-circumscribed and solid left ovarian tumor. After laparoscopic left oophorectomy, the tumor was revealed as an ovarian steroid cell tumor, NOS. During the laparoscopic surgery, tumor ruptured. Complete surgical staging was performed and no evidence of metastasis was found. Gonadotropin releasing hormone agonist was administered monthly for 6 months. The patient has had no evidence of recurrence for 43 months.
Adult ; Female ; Gonadotropin-Releasing Hormone* ; Gonadotropins ; Hair ; Humans ; Laparoscopy ; Leydig Cell Tumor ; Luteoma ; Metrorrhagia ; Neoplasm Metastasis ; Ovarian Neoplasms ; Ovariectomy ; Ovary ; Physical Examination ; Recurrence ; Sex Cord-Gonadal Stromal Tumors ; Steroids ; Ultrasonography

The aim of this study was to evaluate the effects of low concentrations of DEHP and MEHP on steroidogenesis in a murine Leydig tumor cell line (MLTC-1) in vitro. The result of flow cytometry analysis revealed that the proportion of apoptotic cells was significantly increased after the exposure to DEHP. All three genes (P450scc, P450c17, and 3βHSD) under study showed an increased expression following exposure to DEHP or MEHP, although some insignificant inhibitory effects appeared in the 10 μmol/L treatment group as compared with the controls. It was also found that compared with the controls. It was also found that DEHP or MEHP stimulated INSL3 mRNA and protein especially in the 0.001 μmol/L treatment group. Testosterone secretions were stimulated after the exposure to DEHP or MEHP. Alternations of steroidogenic enzymes and INSL3 in MLTC-1 cells might be involved in the biphasic effects of DEHP/MEHP on androgen production.
Animals ; Apoptosis ; drug effects ; Cell Line, Tumor ; Diethylhexyl Phthalate ; analogs & derivatives ; toxicity ; Leydig Cell Tumor ; metabolism ; pathology ; Mice ; Steroids ; biosynthesis

Adenoma ; metabolism ; pathology ; surgery ; ultrastructure ; Adnexa Uteri ; pathology ; surgery ; Adnexal Diseases ; metabolism ; pathology ; surgery ; Carcinoma, Endometrioid ; metabolism ; pathology ; Diagnosis, Differential ; Female ; Granulosa Cell Tumor ; metabolism ; pathology ; Humans ; Hysterectomy ; Keratins ; metabolism ; Leiomyomatosis ; pathology ; surgery ; Microscopy, Electron ; Middle Aged ; Neoplasms, Multiple Primary ; metabolism ; pathology ; surgery ; ultrastructure ; Sertoli-Leydig Cell Tumor ; metabolism ; pathology ; Uterine Neoplasms ; pathology ; surgery ; Vimentin ; metabolism ; WT1 Proteins ; metabolism

Sertoli-Leydig tumors tend to relapse early and due to their rarity, limited data are available regarding a role of chemotherapy in the management of Sertoli-Leydig cell tumors. We present a case of recurrent ovarian Sertoli-Leydig cell tumor whose salvage treatment was successful with paclitaxel and carboplatin chemotherapy.
Carboplatin ; Female ; Ovary ; Paclitaxel ; Recurrence ; Salvage Therapy ; Sertoli-Leydig Cell Tumor