Steroid Profiles in Estrogen Producing Adenoma

Yuta Tezuka, Yuto Yamazaki, Yoshikiyo Ono, Ryo Morimoto, Kei Omata, Kazumasa Seiji, Kei Takase, Yoshihide Kawasaki, Akihiro Ito, Yasuhiro Nakamura, Hideo Harigae, Hironobu Sasano, Fumitoshi Satoh
Journal of the Endocrine Society, Volume 4, Issue 2, February 2020, bvaa004
https://doi.org/10.1210/jendso/bvaa004

Abstract

Because of its rarity, our understanding of steroidogenesis in estrogen-producing adrenocortical adenoma, including the response to adrenocorticotropic hormone (ACTH) stimulation, remains limited. A 65-year-old man was referred to us because of primary aldosteronism and a right adrenal tumor. Endocrinological evaluations revealed secondary hypogonadism due to hyperestrogenemia. Adrenal venous sampling (AVS) and subsequent liquid chromatography–tandem mass spectrometry (LC-MS/MS) indicated bilateral hyperaldosteronism and a right estrogen-producing adrenocortical tumor. He subsequently underwent right unilateral adrenalectomy, which resulted in clinical remission of hypogonadism. Subsequent histopathological analysis identified a right estrogen-producing adrenocortical adenoma and multiple, concomitant adrenocortical micronodules. Sequential evaluation of steroid profiles using LC-MS/MS revealed unique hormone production, including adrenal androgens, and less responsiveness to ACTH in the right estrogen-producing adrenocortical adenoma as compared to the nonneoplastic adrenal cortex. This case study revealed unique profiles of steroid production in estrogen-producing adrenocortical adenoma associated with concomitant primary aldosteronism. Sequential steroid profiling analysis using LC-MS/MS in combination with AVS can contribute to the diagnosis of various adrenal disorders.

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