Assessment of long-term efficacy and safety of metyrapone monotherapy in a patient with ACTH-independent macronodular adrenal hyperplasia
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To the Editor,
ACTH-independent macronodular adrenocortical hyperplasia (AIMAH) is a rare form of Cushing’s syndrome (CS), characterized by macronodular enlargement of the bilateral adrenal glands. Although an 11β-hydroxylase inhibitor, metyrapone, has been used in inoperable CS cases, its efficacy and safety for AIMAH are still unclear. The curative therapy for AIMAH is bilateral adrenalectomy; however, because AIMAH usually occurs in elderly persons, patients often have high operation risks.
A 69-year-old woman, with apparent cushingoid features, was referred to us for hypokalemia (2.1 mEq/l) after coronary artery bypass graft surgery for triple-vessel disease. She received insulin therapy for diabetes (HbA1c, 8.8%). Abdominal computed tomography revealed macronodular enlargement of the adrenal glands (right, 48 × 31 mm; left, 55 × 33 mm). Morning plasma ACTH and cortisol levels were <5.0 pg/ml (reference 7.4–54.7) and 19.0 μg/dl (reference 4.0–18.3), respectively. The cortisol level...
KeywordsCortisol Level Pioglitazone Metyrapone Adrenal Lesion Urinary Free Cortisol Level
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