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      A benign cause of hyperandrogenism in a postmenopausal woman

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          Abstract

          Summary

          Menopause is a relative hyperandrogenic state but the development of hirsutism or virilizing features should not be regarded as normal. We report the case of a 62-year-old woman with a 9-month history of progressive frontotemporal hair loss and hirsutism, particularly on her back, arms and forearms. Blood tests showed increased total testosterone of 5.20 nmol/L that remained elevated after an overnight dexamethasone suppression test. Free Androgen Index was 13.1 and DHEAS was repeatedly normal. Imaging examinations to study adrenals and ovaries were negative. The biochemical profile and the absence of imaging in favor of an adrenal tumor made us consider the ovarian origin as the most likely hypothesis. After informed consent, bilateral salpingectomy-oophorectomy and total hysterectomy were performed. Gross pathology revealed ovaries of increased volume and histology showed bilateral ovarian stromal hyperplasia. Testosterone levels normalized after surgery and hirsutism had completely subsided 8 months later.

          Learning points
          • Menopause is a relative hyperandrogenic state

          • Hirsutism and/or virilizing features, in a postmenopausal woman, should raise the hypothesis of a malignant cause

          • In the absence of an identifiable ovarian or adrenal tumor, the ovarian origin remains the most likely

          • Peripheral aromatization of excess androgen may conduct to high levels of estrogen increasing the risk of endometrial cancer

          • Bilateral oophorectomy results in significant clinical improvement.

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          Most cited references7

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          Management of postmenopausal virilization.

          Mild clinical signs of hyperandrogenism such as hirsutism may appear during the menopausal transition as part of the normal aging process, but the development of frank virilization suggests a specific source of androgen excess, including androgen-secreting tumors.
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            Androgen suppressive effect of GnRH agonist in ovarian hyperthecosis and virilizing tumours.

            Recent studies have suggested that androgen secretion by ovarian virilizing tumours may be gonadotrophin dependent. The aim of this study was to investigate the suppressive effect of GnRH agonist administration on androgen secretion in women with such tumours. A single i.m. injection of D-Trp-6-GnRH (GnRHa), 3.75 mg, was given to five unrelated patients referred for clinical symptoms of virilization with plasma testosterone (T) levels greater than 7 nmol/l but with normal dehydroepiandrosterone sulphate (DHEAS) levels. Diagnoses of adrenal tumour or a non-classical 21-hydroxylase deficiency were screened for by the dexamethasone suppression test, ACTH stimulation test and adrenal CT scanning, and were ruled out in all patients. The one premenopausal patient received cyproterone acetate in a dose of 50 mg twice daily for 3 weeks, starting 1 week before GnRHa administration. Testosterone, androstenedione (A), DHEAS, 17-hydroxyprogesterone (OHP), LH and FSH plasma concentrations were measured by radioimmunoassay of blood samples taken before and 3 weeks after GnRHa. In each patient, GnRHa suppressed gonadotrophin levels and reduced T and A to the range for normal control women. With these results, and because accurate localization of an ovarian androgen secreting tumour could not be achieved by pelvic ultrasonography and CT scanning, exploratory laparotomy was undertaken. A Sertoli-Leydig cell tumour was found in the premenopausal patient, and granulosa cell tumour, hilus cell tumour and two hyperthecoses in the four post-menopausal patients. After bilateral ovariectomy and hysterectomy in the post-menopausal woman and after unilateral ovariectomy in the premenopausal women, androgen levels were normalized. In virilized women, the findings of increased serum testosterone with normal gonadotrophin levels and GnRHa suppression of gonadotrophins leading to normalization of testosterone levels, suggest that various ovarian androgen-secreting tumours, as well as hyperthecosis, are not autonomous but apparently depend upon continuous gonadotrophin stimulation.
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              Association of ovarian hyperthecosis with endometrial polyp, endometrial hyperplasia, and endometrioid adenocarcinoma in postmenopausal women: a clinicopathological study of 238 cases

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                17 February 2021
                2021
                : 2021
                : 20-0054
                Affiliations
                [1 ]Department of Endocrinology , Hospital de Santa Maria, Lisboa, Portugal
                [2 ]Department of Pathology , Hospital de Santa Maria, Lisboa, Portugal
                [3 ]Department of Obstetrics & Gynecology , Hospital de Santa Maria, Lisboa, Portugal
                [4 ]Faculdade de Medicina da Universidade de Lisboa , Lisboa, Portugal
                Author notes
                Correspondence should be addressed to M J Bugalho; Email: maria.bugalho@ 123456chln.min-saude.pt
                Article
                EDM200054
                10.1530/EDM-20-0054
                7923031
                33597312
                d46f13be-c6c9-49a6-8d53-5c75711bed5d
                © 2021 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                : 17 July 2020
                : 14 January 2021
                Categories
                Adult
                Female
                White
                Portugal
                Ovaries
                Gynaecological Endocrinology
                Gynaecology
                Dermatology
                Oncology
                Error in Diagnosis/Pitfalls and Caveats
                Error in Diagnosis/Pitfalls and Caveats

                adult,female,white,portugal,ovaries,gynaecological endocrinology,gynaecology,dermatology,oncology,error in diagnosis/pitfalls and caveats,february,2021

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