Resolution of Cushing's disease followed by secondary adrenal insufficiency after anticoagulant-associated pituitary hemorrhage: report of a case and review of the literature

Abstract

Objective: To describe the 12th well-documented case of spontaneous resolution of pituitary Cushing's disease due to pituitary hemorrhage and to review data on the previous 11 such patients described in the literature.

Methods: We present the longitudinal clinical, endocrinologic, and radiographic data in a 41-year-old woman with Cushing's disease before and after pituitary hemorrhage and summarize similar data in 11 previous reports of patients who convincingly appear to demonstrate the same syndrome.

Results: A 41-year-old woman with classic features of Cushing's disease had an overnight dexamethasone suppressed serum cortisol level of 23 mg/dL. Five months later, symptomatic pituitary hemorrhage developed in conjunction with characteristic pituitary magnetic resonance imaging findings and a serum cortisol value of 2.2 mg/dL. During the ensuing 8 months, she lost her Cushing's habitus, demonstrated improvement in her secondary adrenal insufficiency, and developed an empty right sella turcica, which remained unchanged on 1-year follow-up magnetic resonance imaging of the pituitary. An overnight metyrapone test 3 months later yielded normal results.

Conclusion: Spontaneous remission in pituitary Cushing's disease, with or without later recurrence, has now been well documented in 12 patients. These findings (1) compel a reassessment of whether previously described patients experiencing spontaneous remission in association with medical therapy may have actually sustained asymptomatic pituitary hemorrhage and (2) raise the question of whether, in selected patents with microadenomas, medical treatment of Cushing's disease should be considered more often.