Cases reported "Virilism"

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1/16. Ovarian steroid cell tumor and a contralateral ovarian thecoma in a postmenopausal woman with severe hyperandrogenism.

    A 49-year-old woman presented with rapidly progressing hirsutism, receding hairline, male-pattern baldness and deepening of voice, which had developed over the past 2 years. Hormonal evaluation showed a markedly elevated serum testosterone level (418 ng/dl) and no evidence of increased production of cortisol, dehydroepiandrosterone, dehydroepiadrosterone-sulfate, androstenedione, or 17-hydroxyprogesterone. Transvaginal ultrasound examination suggested the presence of a small mass within the left ovary, but all other radiological studies, including adrenal and ovarian computed tomography, magnetic resonance imaging, radio-labelled cholesterol scintigraphy and positron emission tomography, were negative. Subsequently, bilateral selective venous sampling showed a marked testosterone gradient in the right ovarian vein. Bilateral salpingo-oophorectomy was performed (the patient had had a previous vaginal hysterectomy), and histopathological examination revealed a 10-mm steroid cell tumor within the right ovary and a 15-mm thecal cell tumor within the left ovary. The postoperative serum testosterone level returned to normal and the patient showed a slow regression of clinical symptoms. The simultaneous occurrence of a virilizing ovarian steroid cell tumor and an apparently non-functioning thecoma within the contralateral ovary emphasizes the potential pitfalls that may exist in the preoperative evaluation of patients with markedly increased testosterone production.
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2/16. Virilizing adrenocortical adenoma with Cushing's syndrome, thyroid papillary carcinoma and hypergastrinemia in a middle-aged woman.

    We report a rare case of virilizing adrenocortical adenoma complicated with Cushing's syndrome, thyroid papillary carcinoma and hypergastrinemia. A 45-year-old woman had a history of amenorrhea for 10 years, hypertension for 8 years, and diabetes mellitus for 3 years. physical examination showed a masculinized woman with severe hirsutism, male-like baldness, deep voice, acne in the precordia, and clitorism. plasma testosterone, DHEA-S and urinary 17-KS were high, and plasma cortisol level was it at the upper limit of the normal range, but it did not show a diurnal rhythm nor was suppressed by 2 and 8 mg of dexamethasone. Abdominal CT scan showed a left adrenal tumor (4.5 cm in size). Adrenal scintigram revealed uptake of the tracer on the left side, and plasma cortisol concentration was high in a blood sample from the left adrenal vein. Left adrenalectomy was performed. Histopathological features of resected adrenal tumor were consistent with those of adrenocortical adenoma, consisting of tumor cells with eosinophilic compact cytoplasm. Immunohistochemical staining for steroidogenic enzymes showed reactivity for P450sec, 3 beta-HSD, P450c17, P450c21 and P450c11. plasma testosterone and cortisol levels decreased to the normal range postoperatively. The patient was also found to have a papillary thyroid carcinoma and hypergastrinemia. Our patient is a rare case of virilizing adrenocortical adenoma associated with Cushing's syndrome, thyroid papillary carcinoma, and hypergastrinemia.
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3/16. Virilisation in a case of transitional cell carcinoma of the ovary.

    A 52 year old woman presented with a five month history of emotional changes, voice changes, and of abdominal discomfort. Clinical, biochemical, and radiological examinations showed evidence of virilisation, raised testosterone, and a complex ovarian mass. microscopy of the resection specimen showed the tumour to be a transitional cell carcinoma of the ovary with luteinisation and hyperplasia of the intervening stromal cells. This is a unique case of virilisation caused by transitional cell carcinoma of the ovary. Theories as to the origin of testosterone production in non-functioning ovarian tumours are discussed.
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4/16. Recurrent severe hyperandrogenism during pregnancy: a case report.

    This report describes the case of a 28 year old woman with virilisation occurring in two successive pregnancies. Recurrent maternal virilisation is rare (seven previous reports) and this case is unique in its severity. Differential diagnoses include ovarian disease and fetal aromatase deficiency. New techniques to exclude a fetal cause were used in this case. This patient presented during the third trimester of her first pregnancy with rapid onset of hirsuitism, increased musculature, and deepening voice. A blood hormone profile revealed significant hyperandrogenism (testosterone, 72.4 nmol/litre; normal range, 0.5-3.0). She delivered a normal boy and maternal androgen concentrations returned rapidly to normal (testosterone, 0.8 nmol/litre). She presented two years later, during her second pregnancy, with similar symptoms and biochemistry (testosterone, 47.5 nmol/litre). Again, she delivered a healthy normal boy and androgens returned immediately to normal (serum testosterone, 2.0 nmol/litre). ultrasonography revealed no evidence of ovarian (or adrenal) masses in either pregnancy. umbilical cord venous blood sampling and placental assays revealed no evidence of fetal aromatase deficiency. Recurrent hyperandrogenism during pregnancy is rare. Ovarian luteoma rarely recurs and hyperreactio luteinalis does not lead to such pronounced androgen concentrations. Therefore, this patient has a unique ovarian condition that could be harmful to offspring and mother.
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5/16. Ovarian vein sampling in rapidly progressing virilization. A case report.

    A woman presented with progressive hirsutism, deepening of the voice, clitoromegaly and increased libido. The preoperative serum testosterone level was 2,042 ng/dL. The intraoperative ovarian venous blood testosterone levels were 56,327 ng/dL on the left and 1,417 on the right. After a bilateral salpingo-oophorectomy, the serum testosterone level was 20.7 ng/dL. Initial pathologic examination of the ovaries revealed no potential source of testosterone, but reexamination revealed a pure lipoid cell tumor. Intraoperative ovarian vein androgen measurements can be helpful in establishing a histopathologic diagnosis in cases of microscopic virilizing ovarian neoplasms. Ovarian vein sampling for androgen was essential in locating this patient's microscopic tumor.
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6/16. Virilizing serous cystadenoma.

    A 71-year-old woman, 26 years post-menopause, presented with virilization. Facial hirsuties, non-hereditary frontal balding, voice changes, male escutcheon, and mild clitorimegaly accompanied a right adnexal mass. blood hormone studies showed testosterone 430 ng/dL, FSH 118 mIU/mL, and LH 210 mIU/mL. By ultrasound examination, the cystic adnexal mass involved the right ovary. An 18-cm, 1300-g, unicameral mass with 1200 mL of clear serous fluid and with smooth inner and outer surfaces was removed from the right broad ligament. Intraoperative testosterone levels were as follows: peripheral vein 285 ng/dL, left ovarian vein 301 ng/dL, and right ovarian vein 1635 ng/dL; tumor cystic fluid was 3032 ng/dL. Peripheral vein testosterone was 15 ng/dL 3 days postoperatively. Histopathologically, the tumor was a serous cystadenoma. No evidence of stromal luteinization, hyperplasia, or inflammation was found, and other virilizing lesions were not encountered in either ovary or in other tissues. epithelial cells constituting the tumor may have been the source of the excess testosterone in this unique case of virilizing serous cystadenoma.
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7/16. Fetal and maternal virilization associated with pregnancy. A case report and review of the literature.

    A masculinized female infant was born to a mother who had virilizing signs dating from the fourth month of pregnancy. serum 17 alpha-hydroxyprogesterone, dehydroepiandrosterone, and testosterone levels were all normal in the infant. Maternal testosterone level was markedly elevated one week post partum. dexamethasone phosphate suppression was normal. Human chorionic gonadotropin stimulation five weeks post partum revealed further elevation of high baseline free and total testosterone levels. Free and total testosterone levels 30 weeks post partum were normal, and all maternal virilizing signs had regressed with the exception of her deepened voice. The child has had no progression of masculinization. The mother is believed to have had a luteoma of pregnancy.
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8/16. Androgenic granulosa cell tumors of the ovary. A clinicopathologic analysis of 17 cases and review of the literature.

    We reviewed the clinicopathologic features of 17 androgenic granulosa cell tumors. The patients, two of whom were pregnant, ranged from 2 to 58 years of age (average age, 22.5 years). Fourteen of the patients were masculinized as evidenced by various combinations of facial acne, temporal recession of hair, clitorimegaly, deepening of the voice, and a male escutcheon. Five of these patients had amenorrhea; three of the patients demonstrated hirsutism only. The plasma testosterone level is known to have been elevated preoperatively in nine patients, including the three patients with hirsutism. Four prepubertal patients also demonstrated hyperestrinism as manifested by sexual pseudoprecocity. At laparotomy, all of the tumors were stage lai. Five of the tumors formed unilocular and two of the tumors formed multilocular thin-walled cysts that ranged from 23 to 36 cm in diameter. Nine of the remaining tumors were solid, and one tumor was solid and cystic; these tumors averaged 7 cm in diameter. Eleven tumors were granulosa cell tumors of the adult type, and six tumors were juvenile granulosa cell tumors. The only clinically malignant tumor in the series was a unilocular, cystic, adult granulosa cell tumor that recurred 18 months postoperatively and was fatal within four years.
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9/16. A 15-year-old girl with pubertal masculinization due to bilateral gonadoblastoma and 45,X/46,X, mar karyotype.

    We report the case of a 15-year-old girl with bilateral gonadoblastoma and 45,X/46,X, mar karyotype. She was short but had no other stigmata of turner syndrome. Her genitalia were completely of the female type until she began to show signs of masculinization at age 13 years. She had breasts of Tanner stage 4, primary amenorrhea, clitoromegaly, hirsutism and low voice. plasma testosterone level was elevated and returned to normal after the successful removal of the bilateral gonadoblastoma. Her karyotype was 45,X/46,X, mar and it was proved that the marker chromosome was derived from the short arm of the y chromosome by Southern blot analysis with a y chromosome specific probe, pDP1007. Even when there are no stigmata of turner syndrome or signs of intrauterine virilization, the possibility of gonadoblastoma should not be ruled out in girls who show postnatal virilization.
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10/16. Late-onset 3 beta-hydroxysteroid dehydrogenase deficiency with virilization induced by a large ovarian cyst.

    A midpubertal girl presented with secondary amenorrhea and a rapidly progressive deepening of her voice as the only signs of virilization. Diagnostic work-up yielded an extremely elevated plasma testosterone (289 ng/dl), low estradiol (29 pg/ml) levels and a large solitary cyst of the right ovary, which was totally removed. Pathohistology was in keeping with a granulosa cyst with mild luteinization. Normalization of testosterone (to 27.3 ng/dl) and estradiol (to 62 pg/ml) and resumption of regular menses after 2 months clearly indicated an autonomous function of the cyst. A malignant tumor was unequivocally excluded. Basal and ACTH stimulated levels of adrenal androgens pointed to a late-onset 3 beta-hydroxysteroid dehydrogenase deficiency, which per se is known to induce polycystic ovarian changes, but to date has never been described to be accompanied with a large and autonomous follicular cyst.
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