Periodic Secretion of Adrenocorticotropin in a Patient with Cushing's Disease Manifested during Pregnancy

Tomoko MIYOSHI; Fumio OTSUKA; Jiro SUZUKI; Kenichi INAGAKI; Masaya TAKEDA; Yoshihiro KANO; Tetsuji YAMASHITA; Toshio OGURA; Isao DATE; Yasushi TANAKA; Kozo HASHIMOTO; Hirofumi MAKINO

doi:10.1507/endocrj.52.287

ORIGINALS

Periodic Secretion of Adrenocorticotropin in a Patient with Cushing's Disease Manifested during Pregnancy

Tomoko MIYOSHI, Fumio OTSUKA, Jiro SUZUKI, Kenichi INAGAKI, Masaya TAKEDA, Yoshihiro KANO, Tetsuji YAMASHITA, Toshio OGURA, Isao DATE, Yasushi TANAKA, Kozo HASHIMOTO, Hirofumi MAKINO

Author information

Tomoko MIYOSHI
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Fumio OTSUKA
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Jiro SUZUKI
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Kenichi INAGAKI
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Masaya TAKEDA
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Yoshihiro KANO
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Tetsuji YAMASHITA
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Toshio OGURA
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Isao DATE
Department of Neurosurgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Yasushi TANAKA
Department of Endocrinology, Metabolism and Nephrology, Kochi Medical School, Kochi University
Kozo HASHIMOTO
Department of Endocrinology, Metabolism and Nephrology, Kochi Medical School, Kochi University
Hirofumi MAKINO
Department of Medicine and Clinical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences

Keywords: Adrenocorticotropin (ACTH), Big ACTH, Cyclical Cushing's syndrome, Gel filtration analysis, Pituitary adenoma, Pregnancy

JOURNAL FREE ACCESS

2005 Volume 52 Issue 3 Pages 287-292

DOI https://doi.org/10.1507/endocrj.52.287

Details

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.

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