Endocrine Abstracts

Introduction: Adrenocortical oncocytomas, made up of specific epithelial-origin cells, are rare neoplasms, generally without hormonal activity. Most of them are considered to be benign tumors, with the size of generally 4-8 cm and lack of pathognomonic radiological features.

Case Report: This is a 2 year follow up of a 43-year-old woman, who first referred to the endocrinologist in 2021 due to weight gain, resistant arterial hypertension and menstrual irregularities for several years. After 10-years of infertility 7-years ago she delivered a preterm healthy baby-girl. Physical examination revealed cushingoid characteristic features: dorsal cervical fat pad, moon facies, fragile skin and thin extremities. Cushing’s syndrome was confirmed by an overnight 1-mg dexamethasone-suppression-test, an elevated free cortisol on a 24-hour urine collection, suppressed ACTH level and low DHEA-S level. Primary hyperaldosteronism was excluded by an oral salt loading test. Urinary metanephrines were normal. HBA1C-6.0%. Abdominal CT and MRI revealed left-sided adrenal heterogeneous lesion measuring 2.3×2.8×2.2 cm with a calcification focus in the periphery. She was also revealed small liver hemangiomas and NAFL-disease. In July 2021 the patient underwent laparoscopic left adrenalectomy. To prevent postoperative adrenal insufficiency, the patient was medicated with hydrocortisone. The operative adrenal gland measured 6×2.5×2.5 cm, with a nodule of 2.5×2.2×2.0 cm of well-defined limits, yellowish. The histological/immunohistochemical analysis revealed the diagnosis of adrenocortical neoplasm with predominance of oncocytic cells in solid-trabecular-structures (adrenocortical oncocytoma), having uncertain malignant potential. The doses of hydrocortisone were tapered slowly and stopped 3 months later. 6,12 and 24 months after surgery her cortisol, DHEA-S, electrolyte levels, HBA1C were normal, without any radiologic evidence of recurrence. In March 2023 she delivered a healthy baby-girl through C-section, without any gynecological or intraoperative complications. She is currently in follow up.

Discussion: Hormonally active adrenocortical oncocytomas are extremely rare tumors, about 17.0% of adrenal oncocytomas may have hormonal activity. By origin oncocytomas are epithelial-tumors composed of large eosinophilic cells, having mitochondria-rich cytoplasm and large nucleoli. The frequency of non-functioning oncocytomas is higher in females. The diagnosis of these neoplasms is mostly based on histological/immunohistochemical analysis. The Lin-Weiss-Bisceglia risk system is used to distinguish malignant oncocytic tumors, with major and minor criteria for differentiation. Small oncocytomas are commonly benign tumors, but in our clinical case oncocytoma expressed with Cushing’s syndrome, with its two minor criteria (necrosis and capsular invasion), was classified as having uncertain malignant potential, thus suggesting important prognosis. Two-years close follow-up did not reveal any abnormalities.