Abstract
A 39-year-old man was admitted to the hospital because of hypertension and obesity. On physical examination, he had a moon-shaped appearance and purple striae on the lower abdomen. Abdominal CT scan revealed situs inversus totalis and a right adrenal tumor. His plasma cortisol level was elevated without showing circadian rhythm and was not suppressed with dexamethasone suppression test. Plasma ACTH level was suppressed, below 5pg/ml (normal: 9-52pg/ml), and urinary level of 17-OHCS was elevated to 33.0mg/day (normal: 3.4-12.0mg/day). 131I adsterol scintigram showed hot nodule only at the right adrenal area. Under the diagnosis of Cushing syndrome due to the right adrenal tumor, the patient underwent right adrenalectomy. Cut surface of the tumor was tan in color, and the tumor was 40×30mm in diameter and 45g in weight. Histological examination showed benign adrenocortical adenoma. Cushing syndrome associated with situs inversus totalis is very rare, and to the best of our knowledge, this is the first case in the English and Japanese literature.