Testosterone-producing Adrenocortical Carcinoma Identified by Amenorrhea—A Case Report—

Abstract

A 28-year-old woman was referred to our hospital because of an elevated serum testosterone level and amenorrhea. No features suggesting Cushing's syndrome were observed. Her clitoris was slightly swollen. Her serum levels of testosterone and dehydroepiandrosterone sulfate (DHEA-S) were abnormally elevated. No abnormality in pituitary function was identified. A 9-cm, well-bordered, large, round, left adrenal tumor with heterogeneous enhancement was revealed using contrast-enhanced abdominal computed tomography. No lymph node swelling or lung or liver metastasis was observed. A diagnosis of testosterone-producing left adrenocortical carcinoma was confirmed. Laparotomy revealed a clear-bordered, hypervascular tumor originating from the left adrenal gland without invasion into the surrounding organs. A 380-g tumor could be excised easily with the left adrenal gland. On the cut surface, the tumor was reddish-brown with a dark-red central portion. Histopathological examination revealed atypical tumor cells with palisade arrangement and large irregular nuclei. The tumor fulfilled 8 of the 9 Weiss criteria, and a diagnosis of adrenocortical carcinoma (T2N0M0) was confirmed. The postoperative course was uneventful, and she was discharged on the seventh postoperative day. Her testosterone and DHEA-S decreased to normal levels, and menstruation resumed. No sign of recurrence was observed and adjuvant therapy was not required for 16 months.