Resection of a pure androgen secreting adrenal adenoma in a postmenopausal woman: a case report.

Pure androgen secreting adrenal tumors are exceedingly rare, presenting in higher numbers in women compared with men, and are particularly rare in women of postmenopausal age. Postmenopausal hyperandrogenism is usually ovarian or adrenal in origin, with tumors representing an uncommon cause, which are more frequently ovarian but could also be adrenal. Herein we present a case of a 61-year-old postmenopausal woman, who had suffered multiple reproductive disturbances, presenting with a 10-year history of virilizing symptoms, most bothersome of which was generalized hirsutism, alongside clitoromegaly, irritability, and voice deepening. Work-up of the patient revealed a 1.5 cm left adrenal mass, which was removed through laparoscopic total adrenalectomy. Postoperatively, the patient's androgen levels dropped significantly. An adrenal androgen secreting tumor is a can't miss diagnosis that should always be considered in the evaluation of postmenopausal women with hyperandrogenism, alongside the more common etiologies. Regular hormonal follow-up is essential.

Dexamethasone suppression test versus selective ovarian and adrenal vein catheterization in identifying virilizing tumors in postmenopausal hyperandrogenism - a systematic review and meta-analysis.

OBJECTIVE: The diagnostic accuracy of tests in identifying virilizing tumors in postmenopausal hyperandrogenism is limited. This systematic review compares the dexamethasone suppression test against selective ovarian and adrenal vein sampling of androgens in distinguishing neoplastic from non-neoplastic causes of postmenopausal hyperandrogenism. METHODS: Diagnostic test accuracy studies on these index tests in postmenopausal women were selected based on pre-established criteria. The true positive, false positive, false negative, and true negative values were extracted and meta-analysis was conducted using the hierarchical summary receiver operator characteristics curve method. RESULTS: The summary sensitivity of the dexamethasone suppression test is 100% (95% CI 0-100%) and that for selective venous sampling is 100% (95% CI 0-100%). The summary specificity of the dexamethasone suppression test is 89.2% (95% CI 85.3-92.2%) and that for selective venous sampling is 100% (95% CI 0.3-100%). CONCLUSION: There is limited evidence for the use of dexamethasone suppression test or selective venous sampling in identifying virilizing tumors in postmenopausal hyperandrogenism.

Gonadotropin-Releasing Hormone Analogue Stimulation Test Versus Venous Sampling in Postmenopausal Hyperandrogenism.

Postmenopausal hyperandrogenism can be due to excessive androgen secretion from adrenal or ovarian virilizing tumors or nonneoplastic conditions. The etiology of postmenopausal hyperandrogenism can be difficult to discern because of limited accuracy of current diagnostic tests. This systematic review compares the diagnostic accuracy of the gonadotropin-releasing hormone (GnRH) analogue stimulation test against selective ovarian and adrenal vein sampling of androgens in distinguishing neoplastic from nonneoplastic causes of postmenopausal hyperandrogenism. Diagnostic test accuracy studies on these index tests in postmenopausal women were selected based on preestablished criteria. The true positive, false positive, false negative, and true negative values were extracted and meta-analysis was conducted using the hierarchical summary receiver operator characteristics curve method. The summary sensitivity of the GnRH analogue stimulation test is 10% (95% confidence interval [CI], 1.1%-46.7%) and that for selective venous sampling is 100% (95% CI, 0%-100%). Both tests have 100% specificity. There is limited evidence for the use of either test in identifying virilizing tumors in postmenopausal hyperandrogenism.

Persistent Poor Metabolic Profile in Postmenopausal Women With Ovarian Hyperandrogenism After Testosterone Level Normalization.

CONTEXT: Data on prevalence of metabolic risk factors in hyperandrogenic postmenopausal women are limited. Also, the correlation between metabolic disorders and androgen excess in this scenario is poorly understood. OBJECTIVES: We aimed to assess the prevalence of obesity, hypertension, type 2 diabetes (T2D), and dyslipidemia (DLP) in postmenopausal women with hyperandrogenism of ovarian origin before and after surgical normalization of testosterone (T) levels, as well as the impact of androgen normalization on body mass index (BMI), glucose, and lipid metabolism. DESIGN: Retrospective study. SETTING: Tertiary health center. PARTICIPANTS: Twenty-four Brazilian women with postmenopausal hyperandrogenism who underwent bilateral oophorectomy between 2004 and 2014 and had histologically confirmed virilizing ovarian tumor (VOT) or ovarian hyperthecosis (OH) and T-level normalization after surgery were selected. MAIN OUTCOME MEASURES: FSH, LH, total and calculated free T, BMI, fasting plasma glucose (FPG), glycated hemoglobin (HbA1c), total cholesterol, high-density lipoprotein cholesterol (HDL-C), low-density lipoprotein cholesterol (LDL-C), and triglyceride (TG) serum levels were accessed before (n = 24) and 24 months after (n = 19) bilateral oophorectomy. RESULTS: At baseline, the overall prevalence rates of obesity, T2D, DLP, and hypertension were 58.3%, 83.3%, 66.7%, and 87.5%, respectively. No significant difference in prevalence was found between patients with OH and VOTs. At follow-up, FSH, LH, and total and free T levels had returned to menopausal physiologic levels, but mean BMI and mean FPG, HbA1c, LDL-C, HDL-C, and TG levels did not differ from baseline. CONCLUSIONS: Postmenopausal hyperandrogenism is associated with adverse metabolic risk. Long-term normalization of testosterone levels did not improve BMI, glucose, or lipid metabolism.

Effective GnRH analogue treatment and persistent remission of postmenopausal gonadotropin dependent hyperandrogenism: one case report and literature review / 中华内分泌代谢杂志

A retrospective study was conducted on one patient with postmenopausal hyperandrogenism. The postmenopausal female patient was found with significant hyperandrogenism ( total testosterone 6. 4 nmol/ L) and hirsutism, with normal adrenal androgen ( dehydroepiandrosterone-sulfate ) level. Computed tomography and ultrasound did not detect any ovarian and adrenal mass. Positron emission tomography revealed without high metabolic region, either. Ovarian and adrenal vein catheterization did not reveal any positive result. Obviously, ovary-derived hyperandrogenism was highly suspected, but the patient still refused any surgical exploration. After a trial of a single dose of gonadotropin releasing hormone analogue(GnRHa) triptorelin, increased testosterone level returned to normal along with decreasing level of gonadotropin. Multiple doses were needed for maintenance of testosterone at normal level with the intervals of 2 ～ 12 months. This response to the treatment was consistent with that of gonadotropin dependent hyperandrogenism.

MENOPAUSAL ANDROGEN EXCESS - ASSOCIATED CARDIO-METABOLIC RISK: CLUES FOR OVARIAN LEYDIG CELL TUMOUR (CASE REPORT AND MINI-REVIEW OF LITERATURE).

BACKGROUND: Ovarian Leydig cell tumour is a very rare steroid hormones producing mass, causing clinical and biochemical hyperandrogenism. Even if the level of evidence is based on case studies, many authors (but not all) agree that raised androgens increase the cardio-metabolic risk thus early diagnosis and treatment are necessary On the other hand, the endocrine features pointing an ovarian tumour source of testosterone do not indicate the specific histological finding which needs a post-operative conformation. CASE PRESENTATION: We report a case of a 60-year-old woman with a 4-year history of progressive virilisation in association with hypertension, high number of red blood cells, impaired glucose tolerance and dyslipidemia. Total testosterone was 20 times above normal with suppressed gonadotropins, inadequate for menopause. Trans-vaginal ultrasound and pelvic and abdominal computerized axial tomography imaging revealed a right ovarian solid nodule, and no evidence of alteration in the adrenal glands. Total hysterectomy and bilateral salpingo-oophorectomy were performed. Histopathology and immunohistochemistry confirmed the diagnosis of Leydig cell tumour. After surgery, androgen levels returned to normal and the doses of anti-hypertensive drugs were reduced. CONCLUSIONS: The hyperandrogenic state with elevated plasma testosterone and progressive signs of virilization raises suspicion of an ovarian androgen-secreting tumor. For a postmenopausal patient with hyperandrogenism the diagnosis of Leydig cell tumour should be considered. However, the exact diagnosis is provided by post-operative histological exam. Prolonged exposure to hyperandrogenism may generate cardiovascular abnormalities and metabolic syndrome which after tumor excision and removal of the source of androgen hormones are expected to significantly improve.