title: Cushing's syndrome by left adrenocortical adenoma synchronously associated with primary aldosteronism by right adrenocortical adenoma: report of a case.
abstract: Synchronous associations of Cushing's syndrome (CS) and primary aldosteronism (PA) with multiple adrenocortical adenomas secreting each hormone independently have rarely been reported. Herein, we describe a unique case of PA associated with CS with detailed clinical and pathological investigations. Bilateral adrenal masses with clinical symptoms of CS and PA were found in a 43-year-old woman. Venous sampling demonstrated excess secretion of cortisol, and aldosterone from right, and left tumor, respectively. A bilateral laparoscopic partial adrenalectomy was undergone. The right adrenal tumor (3 cm) was yellow in color with abundant lipofuscin granules, and was composed of both eosinophilic compact cells and clear cells. In situ hybridization showed that both mRNAs for HSD3B2 and CYP17A1 were strongly expressed in the tumor, suggesting cortisol synthesis. Left adrenal tumor (2.4 cm) was golden-yellow in color, and composed of clear cells only. Expression of HSD3B2 and CYP11B mRNAs were observed in the tumor compatible with the aldosterone synthesis. Furthermore, minute nodules were found at the surface of normal-appearing cortex on both sides of the adrenal glands, and the expression of HSD3B2 and CYP11B mRNAs was clearly demonstrated within the nodules, indicating aldosterone synthesis. We diagnosed that the present case had 1) cortisol-producing right adrenocortical adenoma, 2) aldosterone producing left adrenocortical adenoma, and 3) cortical minute nodules with aldosterone production in both adrenal glands compatible with idiopathic adrenal hyperplasia. We reviewed the cases reported, and discussed the significance of the minute nodules in the adrenal cortex, often found in association with the adrenocortical adenoma.
authors.affiliation: Department of Surgical Oncology, Osaka City University Graduate School of Medicine, Osaka, Japan.
authors.forename: Naoyoshi
authors.initials: N
authors.lastname: Onoda
authors.forename: Tetsuro
authors.initials: T
authors.lastname: Ishikawa
authors.forename: Kohei
authors.initials: K
authors.lastname: Nishio
authors.forename: Hideki
authors.initials: H
authors.lastname: Tahara
authors.forename: Masaaki
authors.initials: M
authors.lastname: Inaba
authors.forename: Kenichi
authors.initials: K
authors.lastname: Wakasa
authors.forename: Tomohiko
authors.initials: T
authors.lastname: Sumi
authors.forename: Takanori
authors.initials: T
authors.lastname: Yamazaki
authors.forename: Kazuto
authors.initials: K
authors.lastname: Shigematsu
authors.forename: Kosei
authors.initials: K
authors.lastname: Hirakawa
mesh_terms: D000306:Adrenal Cortex Neoplasms / Q000150:complications* / Q000175:diagnosis / Q000601:surgery; D000315:Adrenalectomy; D018246:Adrenocortical Adenoma / Q000150:complications* / Q000175:diagnosis / Q000601:surgery; D000328:Adult; D003480:Cushing Syndrome / Q000175:diagnosis / Q000209:etiology* / Q000601:surgery; D005260:Female; D006801:Humans; D006929:Hyperaldosteronism / Q000175:diagnosis / Q000209:etiology* / Q000601:surgery; D011375:Progesterone Reductase / Q000378:metabolism; D013252:Steroid 11-beta-Hydroxylase / Q000378:metabolism
chemical_list: C533068:3 beta-hydroxysteroid dehydrogenase type II; D011375:Progesterone Reductase; D013252:Steroid 11-beta-Hydroxylase