Leydigcelhyperplasie: een zeldzame oorzaak van hyperandrogenisme bij een menopauzale vrouw

Leydig cell hyperplasia: an atypical cause of postmenopausal hyperandrogenism We present a case of a 64-year old postmenopausal patient with new onset hirsutism, acne and clitoromegaly. Her medical history includes a non-secreting adrenal incidentaloma. Biochemical evaluation withheld an elevated testosterone with normal dehydroepiandrosterone sulphate (DHEAS). Other adrenal biochemical tests were normal and adrenal imaging was unaltered. Imaging of the ovaries was unremarkable. Due to the normal DHEAS, ovarian etiology was suspected, for which a bilateral oophorectomy was performed. Pathological examination showed bilateral Leydig cell hyperplasia, a benign though rare cause of postmenopausal hyperandrogenism. Postmenopausal hyperandrogenism is caused by of a group of gynaecological and endocrinological disorders. In case of rapid onset and severe symptoms of hirsutism or virilization an androgen-secreting neoplastic disorder should be suspected. Normal DHEAS is suggestive for an ovarian etiology. Imaging is necessary to differentiate between adrenal and ovarian causes, though normal imaging of the ovaries does not rule out ovarian pathology. Definitive diagnosis is often based on pathological examination after oophorectomy.

SAT-LB2 Elevated Testosterone Secondary to Leydig Cell Hyperplasia in Bilateral Ovaries

Background: Postmenopausal hyperandrogenism can be caused by androgen use, ovarian hyperthecosis, ovarian neoplasms, and adrenal neoplasms. Clinical case: A 64 year old post-menopausal woman presented for evaluation of hirsutism. She had developed generalized hair loss, terminal hairs on face and chest, and new onset acne after discontinuing hormone replacement therapy. During workup for hirsutism, she was found to have elevated testosterone at 119 ng/dL (reference range: 2-45 ng/dL). Other hormonal evaluation came back within normal limits, with DHEA sulfate <15 mcg/dL (reference range <186 mcg/dL), estradiol 25 pg/ml (reference range <12-32 pg/ml), FSH 34.9 mIU/ml (reference range 23-116.3 mIU/ml), and LH 30.1 mIU/ml (reference range 15.9-54 mIU/ml). Transvaginal ultrasound did not reveal any abnormal adnexal masses. CT abdomen showed a 1.0 cm nodule in the left adrenal gland consistent with a lipid rich adenoma. Further work up showed a normal 24 hour urine free cortisol at 25 ug/24h (reference range: 4-50 ug/24h), normal 24 hour urine metanephrines at 709 ug/24h (reference range: 224-832 uh/24h), normal aldosterone at 13 ng/dL (reference range <21 ng/dl) and renin at 0.37 ng/mL/h (reference range 0.25-5.82 ng/mL/h). MRI of the adrenal glands showed a slightly lobular left adrenal gland and no discrete adrenal mass. MRI of the pelvis showed mildly prominent ovaries bilaterally, but no adnexal or ovarian masses. After repeat laboratory testing showed consistently elevated testosterone levels up to 170 ng/dl and symptoms of hyperandrogenism persisted, the patient underwent bilateral salpingo-oophorectomy. Testosterone level post-operatively dropped to 18 ng/dl and remained within normal limits on repeated measurements. Her symptoms of hyperandrogenism resolved over the next several months. Surgical pathology showed endosalpingiosis, benign paratubal cysts, and confluent aggregates of Leydig cell hyperplasia in bilateral ovaries. Conclusion: Leydig cell hyperplasia is a rare cause of hypertestosteronemia that may be considered in patients with negative work up for alternative etiologies for post-menopausal hirsutism.