Case Report: Exceptional Association of Leydig Cell Ovarian Tumor and Primary Hyperparathyroidism in a Postmenopausal Patient

Abstract

Abstract Background: Leydig cell tumors account for less than 0.1% of all ovarian tumors and are usually unilateral and benign. They present mostly in postmenopausal women with symptoms of hyperandrogenism. On the other hand, primary hyperparathyroidism (PHPT) is a relatively common endocrine disorder, with a prevalence of one to seven cases per 1,000 adults. It most commonly presents as an isolated, sporadic clinical entity, but in approximately 10% of the cases, it presents with a clear hereditary pattern and sometimes in association with other hormone producing tumors. Clinical Case: a 55-year-old nulliparous postmenopausal woman with history of breast cancer, Type 2 Diabetes Mellitus, hypothyroidism, hypercalcemia and hypertension who presented with progressive hirsutism and male pattern hair loss. Total testosterone levels were found to be significantly elevated in the 800s ng/dL (normal range 14–76 ng/dL). Additionally, patient was found to have mild hypercalcemia with inappropriately normal PTH and hypercalciuria, consistent with primary hyperparathyroidism (PHPT). A hypoechoic lesion in the left ovary was evident on pelvic ultrasound after 1 year of repeat imaging studies, including MRI. Bone density was within normal limits. The patient underwent bilateral Hysterosalpingo-oophorectomy and pathology report was consistent with Ovarian Leydig cell tumor. During the postoperative period, the patient reported improvement of hyperandrogenism symptoms. Discussion: We are presenting this case because of the rare association of ovarian Leydig cell tumor and PHPT in a postmenopausal female patient with virilizing symptoms. Thus, this would be the second case reported in literature by this time. This could be a rare coincidence or part of an inherited or sporadic mutation yet to be discovered. This case restates the concept that in the presence of hirsutism, rare etiologies should be sought. Virilizing tumors of the ovary should be considered despite negative initial imaging studies. PHPT should be considered in patients with asymptomatic hypercalcemia with inappropriately normal PTH and hypercalciuria. Conclusion: Careful identification and report of cases with unusual clinical presentations is essential to better understand the pathophysiology of uncommon diseases and ultimately be able to guide their management.