Ikki Sakuma, Jun Saito, Yoko Matsuzawa, Masao Omura, Seiji Matsui, Koshiro Nishimoto, Kuniaki Mukai and Tetsuo Nishikawa
A 52-year old woman was admitted to our hospital for evaluation of left adrenal incidenataloma. Endocrinological examination showed Cushing’s syndrome (CS) complicated with masked primary aldosteronism (PA). On the other hand, multiple sampling from the central veins and one or two tributaries of the adrenal veins before and after ACTH-stimulation (multiple AVS) clearly revealed bilateral hyperaldosteronism with excess cortisol secretion from the left adrenal. Thus, we diagnosed this case as CS due to left adrenal tumor with bilateral hyperaldosteronism, and left adrenalectomy was done. Immunohistochemical analysis of the removed left adrenal showed cortisol-producing adenoma and multiple aldosterone-producing cell clusters (APCCs) expressing CYP11B2 within the attached adrenal. Bilateral PA is mostly diagnosed as idiopathic hyperaldosteronism (IHA). IHA has not been examined enough pathologically. We first describe here a possible involvement of APCCs inducing hyperaldosteronism in a case of bilateral PA with a cortisol-producing-adenoma.