Cushing syndrome caused by ectopic adrenal adenoma: two-case report and literature review

Abstract

Objective To analyze the clinical characteristics and treatment of Cushing syndrome caused by ectopic adrenal adenoma.Methods The diagnosis and treatment data of 2 cases of ectopic adrenal adenoma were reported retrospectively.Results One male case (36 years old) and another female case (51 years old) were reported.The duration of the disease was 2 and 12 years respectively.Both patients had typical appearance of Cushing syndrome,with hypertension,hyperglycemia,hypokalemia,and thoracolumbar vertebral compression fractures.Serum and 24 h urinary free cortisol level were elevated,circadian rhythm was disappeared,and serum ACTH was suppressed.The cortisol level could not be suppressed in both low-and high-dose dexamethasone suppression tests.Imaging studies revealed atrophy of bilateral adrenal glands and a big nodule near the left renal hilum in each of both patients.The clinical manifestations were relieved after successful laparoscopic wedge resection of the adenoma near the left renal hilum.Blood pressure and glucose levels were also significantly improved after appropriate hormone replacement.Conclusions When the diagnosis of corticotrophin-independent Cushing syndrome was definite and imaging studies showed that bilateral adrenal glands were normal or atrophic,the possibility of ectopic adrenal adenoma should be considered. Key words: Adrenal adenoma; Cushing syndrome