Ovarian Leydig cell tumor, a sub-type of ovarian steroid cell tumor, accounts for less than 0.1% of all ovarian tumors. It can affect women of any age group but is most common in postmenopausal women. We here report a case of virilizing ovarian Leydig cell tumor with multiple non-functional endocrine neoplasias (pituitary and adrenal adenomas) in a 48-year-old woman. She first presented with sub-abdominal pain and hirsutism since menopause three years ago. Subsequently, she had slight facial acne, voice deepening, breast atrophy, and a prominent Adam’s apple. Her hormone profile showed an elevated level of testosterone, high free androgen index, low levels of luteinizing hormone and follicle stimulating hormone, and normal levels of random cortisol, androstenedione, 17-hydroxyprogesterone and dehydroepiandrosterone sulfate. A pelvic enhanced magnetic resonance imaging (MRI) scan showed nodules in the right ovary, and a pituitary enhanced MRI revealed a microadenoma. An enhanced computerized tomography scan of the adrenal gland revealed left adrenal nodules, possibly adenomas. After a right cystectomy and right fallopian tube resection, her testosterone level declined to 0.38 nmol/L and the symptoms associated with hyperandrogenism improved. This is a rare case of virilizing ovarian Leydig cell tumor with multiple non-functional endocrine neoplasias. We believe our findings will be helpful in the clinical diagnosis and treatment of hyperandrogenism.
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