A case of Adrenocoricotrophic hormone-independent bilateral adrenocortical macronodular hyperplasia concomitant with primary aldosteronism

被引：9

作者：

Tokumoto, Mao ^{[1
]}

Onoda, Naoyoshi ^{[1
]}

Tauchi, Yukie ^{[1
]}

Kashiwagi, Shinichiro ^{[1
]}

Noda, Satoru ^{[1
]}

Toi, Norikazu ^{[2
]}

Kurajoh, Masahumi ^{[2
]}

Ohsawa, Masahiko ^{[3
]}

Yamazaki, Yuto ^{[4
]}

Sasano, Hironobu ^{[4
]}

Hirakawa, Kosei ^{[1
]}

Ohira, Masaichi ^{[1
]}

机构：

[1] Osaka City Univ, Grad Sch Med, Dept Surg Oncol, Abeno Ku, 1-4-3 Asahi Machi, Osaka 5458585, Japan

[2] Osaka City Univ, Grad Sch Med, Dept Metab Endocrinol & Mol Med, Osaka, Japan

[3] Osaka City Univ, Grad Sch Med, Dept Diagnost Pathol, Osaka, Japan

[4] Tohoku Univ, Grad Sch Med, Dept Anat Pathol, Sendai, Miyagi, Japan

来源：

BMC SURGERY | 2017年 / 17卷

基金：

日本学术振兴会;

关键词：

ACTH - Independent bilateral adrenocortical macronodular hyperplasia; Primary aldosteronism; Laparoscopic unilateral adrenolectomy; Preclinical Cushing's syndrome; CUSHINGS-SYNDROME; ADRENAL-HYPERPLASIA; UNILATERAL ADRENALECTOMY; RECEPTORS; MINERALOCORTICOIDS; THERAPY;

D O I：

10.1186/s12893-017-0293-z

中图分类号：

R61 [外科手术学];

学科分类号：

摘要：

Background: Adrenocoricotrophic hormone (ACTH) -independent bilateral adrenocortical macronodular hyperplasia (AIMAH) is a rare cause of Cushing's syndrome, and is characterized by bilateral adrenal hyperplasia. However, Primary aldosteronism (PA) is a relatively common adrenal disease. Case presentation: A 56-year-old man who has been treated hypertension and diabetes mellitus was detected low plasma potassium level with an elevated level of plasma aldosterone concentration and bilateral adrenal swelling. Endocrinological examinations showed autonomous secretion of cortisol and aldosterone, with suppression of plasma ACTH level and renin activity. A selective adrenal venous sampling demonstrated that left adrenal gland was responsible for aldosterone hypersecretion. He was diagnosed preclinical Cushing's syndrome due to ACTH-independent bilateral adrenocortical macronodular hyperplasia (AIMAH) associated with aldosterone producing adenoma of the left adrenal gland. A laparoscopic left adrenalectomy was performed. Conclusion: The resected adrenal specimen histologically consisted with a diagnosis of AIMAH. Moreover, tiny cell clusters positive immunostaining for aldosterone synthase was revealed. This is a rare case of AIMAH accompanied by preclinical Cushing's syndrome and primary aldosteronism.

引用

页数：5