Abstract
Adrenal oncocytoma (AO) is an extremely rare adrenocortical neoplasm and little is known about its malignant potential, secretory properties, and hereditary origin. We present the case of a benign AO with concomitant incidentally found papillary thyroid cancer (PTC) and review similar cases in the literature. Immunohistochemistry and next-generation sequencing (NGS) were performed. A 66-year-old women was incidentally found to have a large, androgen-secreting right adrenal mass. 18F-Fluorodeoxyglucose positron emission tomography showed intense uptake (SUVmax 88.7) of this mass and found a hypermetabolic right thyroid mass. Open adrenalectomy was performed for this highly suspicious adrenal mass. Histopathology revealed benign AO that was BRAFV600E negative, with low Ki-67, and no somatic mutation found on NGS. Thyroidectomy revealed invasive, BRAFV600E-positive PTC. At 6 months follow-up, androgen levels returned to normal, and no recurrence was seen on imaging. To our knowledge, this is the first report of an androgen-secreting AO with concomitant PTC. Possibly the simultaneous discovery of two independent neoplasms was observed. In conclusion, this case highlights that care should be given to exclude concomitant neoplasms. Long-term and regular imaging with biochemical follow-up is warranted, since the outcome and clinical behavior of AO remains uncertain.
Highlights
Oncocytic neoplasms occur in various organs such as the kidneys, thyroid, parathyroid, or salivary glands
Computed tomography (CT) scan or magnetic resonance imaging is weak in defining the nature of large adrenal masses, and the role of 18F-FDG positron emission tomography (PET) remains uncertain
We describe a female patient with a highly suspicious adrenal mass that reveals to be a benign lesion based on the pathological criteria, without somatic mutations, and who presents with a simultaneous endocrine malignancy of the thyroid gland
Summary
Oncocytic neoplasms occur in various organs such as the kidneys, thyroid, parathyroid, or salivary glands. They have been considered benign non-functional neoplasms; in about 20% of cases, they show malignant potential, and in 15% of cases, adrenocortical hormone secretion was detected [1] In most cases, these adrenal masses were detected incidentally at a large size and were suspicious for adrenocortical carcinoma (ACC) [1]. A 18F-FDG PET was performed, revealing a highly metabolic 7.3 cm × 5.6 cm × 8.1 cm right adrenal mass (SUVmax 88.7) with a mass effect on the liver (Figures 1A–C and 2A). Hypermetabolic activity, and androgen secretion, malignancy of the adrenal mass was suspected, and an open right adrenalectomy was performed. At 6-month followup, the androgen levels stayed normal (1.7 μmol/l), and no recurrence was seen on CT scan imaging. Regular imaging and biochemical follow-up at 12 and 18 months for a minimum of 5 years are planned
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