Tags

Type your tag names separated by a space and hit enter

A pitfall in diagnosing Cushing's disease: ectopic ACTH-producing pituitary adenoma in the sphenoid sinus.
Pituitary. 2015 Apr; 18(2):279-82.P

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.

Authors+Show Affiliations

Department of Neurosurgery, Bereich Hypophysenchirurgie, Neurochirurgische Klinik, University Medical Center Hamburg-Eppendorf (UKE), Martinistraβe 52, 20246, Hamburg, Germany, flitsch@uke.de.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

25129688

Citation

Flitsch, J, et al. "A Pitfall in Diagnosing Cushing's Disease: Ectopic ACTH-producing Pituitary Adenoma in the Sphenoid Sinus." Pituitary, vol. 18, no. 2, 2015, pp. 279-82.
Flitsch J, Schmid SM, Bernreuther C, et al. A pitfall in diagnosing Cushing's disease: ectopic ACTH-producing pituitary adenoma in the sphenoid sinus. Pituitary. 2015;18(2):279-82.
Flitsch, J., Schmid, S. M., Bernreuther, C., Winterberg, B., Ritter, M. M., Lehnert, H., & Burkhardt, T. (2015). A pitfall in diagnosing Cushing's disease: ectopic ACTH-producing pituitary adenoma in the sphenoid sinus. Pituitary, 18(2), 279-82. https://doi.org/10.1007/s11102-014-0591-8
Flitsch J, et al. A Pitfall in Diagnosing Cushing's Disease: Ectopic ACTH-producing Pituitary Adenoma in the Sphenoid Sinus. Pituitary. 2015;18(2):279-82. PubMed PMID: 25129688.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - A pitfall in diagnosing Cushing's disease: ectopic ACTH-producing pituitary adenoma in the sphenoid sinus. AU - Flitsch,J, AU - Schmid,S M, AU - Bernreuther,C, AU - Winterberg,B, AU - Ritter,M M, AU - Lehnert,H, AU - Burkhardt,T, PY - 2014/8/18/entrez PY - 2014/8/19/pubmed PY - 2015/12/25/medline SP - 279 EP - 82 JF - Pituitary JO - Pituitary VL - 18 IS - 2 N2 - 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. SN - 1573-7403 UR - https://www.unboundmedicine.com/medline/citation/25129688/A_pitfall_in_diagnosing_Cushing's_disease:_ectopic_ACTH_producing_pituitary_adenoma_in_the_sphenoid_sinus_ L2 - https://doi.org/10.1007/s11102-014-0591-8 DB - PRIME DP - Unbound Medicine ER -