Pituitary, 2015, Vol.18(2), p.279(4)
Byline: J. Flitsch (1), S. M. Schmid (2), C. Bernreuther (3), B. Winterberg (4), M. M. Ritter (5), H. Lehnert (2), T. Burkhardt (1) Keywords: Cushing's disease; Ectopic pituitary adenoma; Transsphenoidal surgery Abstract: Purpose To show a rare case of Cushing's disease and possible cause of failed transsphenoidal surgery. Method We report on a 50-year-old woman suffering from ACTH-dependent Cushing's syndrome. Endocrinological work-up including low-dose/high-dose dexamethasone test (Liddle-test) and CRH test were clearly compatible with pituitary origin. Although an MRI showed no pituitary tumor, CRH-stimulated petrosal sinus sampling revealed a significant central-peripheral gradient in ACTH concentrations, rendering Cushing's disease very likely. The patient underwent transsphenoidal surgery with negative exploration of the pituitary gland. After intraoperative re-evaluation of the preoperative MRI, a "polyp" at the bottom of the sphenoid sinus was identified. The intraoperative microscopic aspect as well as instantaneous sections and cytology of a biopsy confirmed an adenoma, which was then removed. Histological analysis demonstrated an ACTH-producing pituitary adenoma adjacent to respiratory mucous membrane consisting of ciliated epithelium with submucous connective tissue. Postoperatively, ACTH concentrations were decreased and intermittent hydrocortisone substitution treatment was initiated. At the 3-month follow up, Cushing's stigmata were found to be alleviated and the hydrocortisone dosage could be reduced. Conclusion Ectopic pituitary adenoma tissue causing Cushing's disease is extremely rare but a potential cause for surgical failure or re-evaluation. Author Affiliation: (1) Department of Neurosurgery, Bereich Hypophysenchirurgie, Neurochirurgische Klinik, University Medical Center Hamburg-Eppendorf (UKE), Martinistra[sz]e 52, 20246, Hamburg, Germany (2) Department of Internal Medicine I - Endocrinology, University Hospital Schleswig-Holstein, Campus Luebeck, Luebeck, Germany (3) Institute of Neuropathology, University Medical Center Hamburg-Eppendorf (UKE), Hamburg, Germany (4) Marienhospital, Emsdetten, Germany (5) Helios Berlin-Buch, Berlin, Germany Article History: Registration Date: 11/08/2014 Online Date: 17/08/2014
Dexamethasone – Analysis ; Adenoma – Diagnosis ; Adenoma – Analysis ; Corticotropin – Analysis ; Pituitary Tumors – Diagnosis ; Pituitary Tumors – Analysis ; Hydrocortisone – Analysis ; Diagnostic Imaging – Analysis
Cengage Learning, Inc.