Objective. Oncocytomas of the adrenal cortex are usually benign and nonfunctional. They are rarely seen as the cause of hirsutism. Therefore, we aimed to report a case of adrenocortical oncocytoma presenting with hirsutism. Methods. We report a testosterone- and cortisol-secreting adrenal oncocytoma in a 23-year-old female patient presenting with hirsutism. Results. The patient had the complaint of hirsutism for the last year. Laboratory tests revealed total testosterone level of 4.2？ng/mL, free testosterone of >100？pg/mL, and DHEAS level of 574？μg/dL. There was no suppression in cortisol levels with 2？mg dexamethasone suppression test (5.4？μg/dL). Adrenal MRI revealed a ？mm isointense solid mass lesion in the left adrenal gland and the patient underwent laparoscopic left adrenalectomy. Pathological examination confirmed the diagnosis of benign adrenocortical oncoyctoma. Conclusion. This well-characterized case describes a testosterone- and cortisol-secreting adrenocortical oncocytoma as a possible cause of hirsutism. To our knowledge, this is the second report in the literature. Adrenal oncocytomas should always be considered in the differential diagnosis of hirsutism. 1. Introduction Hirsutism, defined as excessive male-pattern hair growth, affects between 5 and 10% of women of reproductive age and most women with hirsutism have polycystic ovary syndrome [1, 2]. Androgen-secreting tumors are rarely seen as the cause of hirsutism. In an epidemiological study, the frequency of androgen-secreting tumors was 0.2% in 950 hirsute women . Most testosterone-secreting tumors arise from the ovary and rarely origins from the adrenal gland. Oncocytic neoplasms or oncocytomas usually arise in the kidneys or thyroid, parathyroid, salivary, or pituitary glands . Oncocytomas of the adrenal cortex are extremely rare and usually detected incidentally . Adrenal oncocytomas are usually benign and nonfunctional in most of cases. Herein, we report a testosterone- and cortisol-secreting adrenal oncocytoma in a 23-year-old female patient presenting with hirsutism. 2. Case Report A 23-year-old female patient admitted to endocrinology outpatient clinic with the complaint of hirsutism for the last year. Excessive hair growth was identified to originate from facial and mandibular areas initially and then to spread to abdominal and thoracic regions. She had regular menstrual cycles since her first period by the age of 12. Medical background and family history were unremarkable. Physical examination revealed that body temperature was 37°C, pulse rate was 80 beats/min,
E. Carmina, F. Rosato, A. Jannì, M. Rizzo, and R. A. Longo, “Relative prevalence of different androgen excess disorders in 950 women referred because of clinical hyperandrogenism,” The Journal of Clinical Endocrinology & Metabolism, vol. 91, no. 1, pp. 2–6, 2006.
M. Bisceglia, O. Ludovico, A. di Mattia et al., “Adrenocortical oncocytic tumors: report of 10 cases and review of the literature,” International Journal of Surgical Pathology, vol. 12, no. 3, pp. 231–243, 2004.
B. Geramizadeh, B. Norouzzadeh, S. Bolandparvaz, and S. Sefidbakht, “Functioning adrenocortical oncocytoma: a case report and review of literature,” Indian Journal of Pathology & Microbiology, vol. 51, no. 2, pp. 237–239, 2008.