title: Periodic secretion of adrenocorticotropin in a patient with Cushing's disease manifested during pregnancy.
abstract: We report the case of 19-year-old woman with cyclical Cushing's disease, in whom plasma adrenocorticotropin (ACTH) was secreted periodically after her first pregnancy. Since the 33rd week of pregnancy, hypertension and proteinuria became clinically remarkable. She gave normal birth at 36th week of pregnancy; however she continued to gain body weight even after delivery and developed typical Cushingoid features. Her ACTH secretion lacked normal daily fluctuation but exhibited periodic change during 1-year observation, showing 119 pg/ml, 34.6 pg/ml and 115 pg/ml at the 4th, 7th and 13th months after delivery. Plasma ACTH levels were increased by corticotropin releasing hormone and metyrapone, while low-dose dexamethasone suppressed cortisol secretion. Gel filtration analysis of the patient's plasma detected big ACTH molecules being eluted with a peak of authentic 1-39 ACTH. Cranial magnetic resonance imaging revealed a 1-cm pituitary mass in right cavernous sinus. The pituitary tumor was removed by transsphenoidal surgery at 13th month after delivery and was pathologically compatible with ACTH-producing pituitary adenoma by immunohistochemistry. This case includes clinically rare subsets of Cushing's syndrome showing periodic ACTH secretion and aberrant ACTH molecules.
authors: Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Japan.
authors: Tomoko
authors: T
authors: Miyoshi
authors: Fumio
authors: F
authors: Otsuka
authors: Jiro
authors: J
authors: Suzuki
authors: Kenichi
authors: K
authors: Inagaki
authors: Masaya
authors: M
authors: Takeda
authors: Yoshihiro
authors: Y
authors: Kano
authors: Tetsuji
authors: T
authors: Yamashita
authors: Toshio
authors: T
authors: Ogura
authors: Isao
authors: I
authors: Date
authors: Yasushi
authors: Y
authors: Tanaka
authors: Kozo
authors: K
authors: Hashimoto
authors: Hirofumi
authors: H
authors: Makino
mesh_terms: D000236:Adenoma / Q000097:blood / Q000378:metabolism* / Q000601:surgery; D000324:Adrenocorticotropic Hormone / Q000097:blood / Q000378:metabolism*; D000328:Adult; D003346:Corticotropin-Releasing Hormone / Q000494:pharmacology; D005260:Female; D006801:Humans; D006854:Hydrocortisone / Q000097:blood / Q000378:metabolism; D008797:Metyrapone; D047748:Pituitary ACTH Hypersecretion / Q000097:blood* / Q000150:complications / Q000175:diagnosis; D010911:Pituitary Neoplasms / Q000097:blood / Q000378:metabolism* / Q000601:surgery; D011247:Pregnancy; D011252:Pregnancy Complications, Neoplastic / Q000097:blood*
chemical_list: D000324:Adrenocorticotropic Hormone; D003346:Corticotropin-Releasing Hormone; D006854:Hydrocortisone; D008797:Metyrapone