title: FSH-producing macroadenoma associated in a patient with Cushing's disease.
abstract: OBJECTIVE
  We encountered a Cushing's disease patient whose surgically removed pituitary macroadenoma was not an ACTH-producing, but rather a gonadotroph adenoma. Cure was obtained only after a tiny microadenoma, overlooked on preoperative studies, was removed by a 2nd operation from a compressed thin normal anterior pituitary gland.
  
  
  METHODS
  This 45-year-old woman with Cushing syndrome presented with diabetes mellitus and steroid psychosis. Endocrinological examinations suggested Cushing's disease and MRI disclosed an invasive macroadenoma (22 mm in diameter) with suprasellar extension.
  
  
  METHODS
  Despite total removal of the invasive macroadenoma by transsphenoidal surgery, her elevated serum cortisol- and ACTH levels failed to decrease. Histologic study of the surgical specimen disclosed that the tumor was a silent FSH-producing, rather than an ACTH-producing adenoma. Detailed re-evaluation of pre- and postoperative MRI suggested the presence of a 3-mm microadenoma on the left side of a thin compressed normal gland. Venous sampling of the cavernous sinus confirmed this suspicion. In a 2nd operation an ACTH-producing microadenoma was removed from inside the thin remaining compressed normal pituitary gland and endocrinological cure of Cushing's disease was achieved.
  
  
  CONCLUSIONS
  Although double adenomas, being a non-ACTH producing macroadenoma associated with an ACTH producing tiny microadenoma, are extremely rare in patients with Cushing's disease, detailed preoperative MRI evaluation is necessary to avoid missing tiny adenomas hidden in a compressed normal pituitary gland which is the cause of Cushing's disease, especially when a macroadenoma is found in patient with Cushing's disease.
authors.affiliation: Department of Hypothalamic and Pituitary Surgery, Toranomon Hospital, Tokyo, and Division of Diabetes & Endocrinology, Saitama Medical Center, Saitama Medical University, Japan. ko-neuro@nms.ac.jp
authors.forename: Ken-Ichi
authors.initials: K
authors.lastname: Oyama
authors.forename: Shozo
authors.initials: S
authors.lastname: Yamada
authors.forename: Noriaki
authors.initials: N
authors.lastname: Hukuhara
authors.forename: Rikako
authors.initials: R
authors.lastname: Hiramatsu
authors.forename: Manabu
authors.initials: M
authors.lastname: Taguchi
authors.forename: Masako
authors.initials: M
authors.lastname: Yazawa
authors.forename: Akira
authors.initials: A
authors.lastname: Matsuda
authors.forename: Eiji
authors.initials: E
authors.lastname: Ohmura
authors.forename: Yasuo
authors.initials: Y
authors.lastname: Imai
mesh_terms: D000324:Adrenocorticotropic Hormone / Q000097:blood; D005260:Female; D005640:Follicle Stimulating Hormone / Q000378:metabolism*; D052681:Gonadotrophs / Q000378:metabolism* / Q000473:pathology; D006801:Humans; D006854:Hydrocortisone / Q000097:blood; D008279:Magnetic Resonance Imaging; D008875:Middle Aged; D009378:Neoplasms, Multiple Primary / Q000473:pathology* / Q000601:surgery; D047748:Pituitary ACTH Hypersecretion / Q000097:blood* / Q000473:pathology / Q000601:surgery; D010911:Pituitary Neoplasms / Q000378:metabolism* / Q000473:pathology* / Q000601:surgery
chemical_list: D000324:Adrenocorticotropic Hormone; D005640:Follicle Stimulating Hormone; D006854:Hydrocortisone