title: Isolated adrenocorticotrophin deficiency associated with anti-pituitary antibodies, pituitary cyst, sphenoidal cyst and pineal tumor.
abstract: This paper reports a rare case of isolated ACTH deficiency associated with anti-pituitary antibodies, pituitary cyst, sphenoidal cyst and pineal tumor. A 68-year-old man consulted our clinic for general fatigue. Laboratory data showed low plasma adrenocorticotrophin (ACTH) and cortisol levels with blunted responses to insulin-induced hypoglycemia and corticotrophin releasing factor (CRF). Urinary 17-OHCS was low but responded to ACTH-Z administration. No other pituitary functions were impaired. Antibodies to the cytoplasm of rat pituitary and the surface of GH3 cells were detected in the serum. The magnetic resonance imaging (MRI) showed a high signal intensity mass in the anterior pituitary and in the sphenoidal sinus in both T1 and T2 weighted images as well as a low signal intensity mass in a T1 weighted image of the pineal region. Transsphenoidal surgery was performed to resect the mass in the sphenoid sinus and in the pituitary. Pathological studies showed a benign cyst in the sphenoid sinus, and fibrous degeneration and decreased basophils in the pituitary. No infiltrative mononuclear cells were detected in the pituitary. Immunohistochemical studies revealed a decrease in the number of ACTH-producing cells in the pituitary. The patient was well maintained by glucocorticoid replacement without any growth of a possibly benign pineal tumor.
authors.affiliation: Department of Medicine, Shimane Medical University, Izumo, Japan.
authors.forename: K
authors.initials: K
authors.lastname: Notsu
authors.forename: N
authors.initials: N
authors.lastname: Oka
authors.forename: M
authors.initials: M
authors.lastname: Sohmiya
authors.forename: T
authors.initials: T
authors.lastname: Sato
authors.forename: S
authors.initials: S
authors.lastname: Ando
authors.forename: K
authors.initials: K
authors.lastname: Moritake
authors.forename: K
authors.initials: K
authors.lastname: Inada
authors.forename: Y
authors.initials: Y
authors.lastname: Osamura
authors.forename: Y
authors.initials: Y
authors.lastname: Kato
mesh_terms: D000324:Adrenocorticotropic Hormone / Q000172:deficiency*; D000368:Aged; D001323:Autoantibodies / Q000097:blood*; D001324:Autoantigens / Q000276:immunology; D001932:Brain Neoplasms / Q000276:immunology / Q000473:pathology; D003346:Corticotropin-Releasing Hormone; D003560:Cysts / Q000276:immunology* / Q000473:pathology; D005455:Fluorescent Antibody Technique; D005938:Glucocorticoids / Q000627:therapeutic use; D007987:Gonadotropin-Releasing Hormone; D006801:Humans; D007124:Immunoenzyme Techniques; D008279:Magnetic Resonance Imaging; D008297:Male; D010870:Pineal Gland / Q000473:pathology; D010900:Pituitary Diseases / Q000276:immunology / Q000473:pathology; D010902:Pituitary Gland / Q000276:immunology* / Q000473:pathology; D013101:Sphenoid Sinus; D013973:Thyrotropin-Releasing Hormone
chemical_list: D001323:Autoantibodies; D001324:Autoantigens; D005938:Glucocorticoids; D007987:Gonadotropin-Releasing Hormone; D013973:Thyrotropin-Releasing Hormone; D000324:Adrenocorticotropic Hormone; D003346:Corticotropin-Releasing Hormone