, Volume 13, Issue 4, pp 574–578 | Cite as

Rare case of Cushing’s disease due to double ACTH-producing adenomas, one located in the pituitary gland and one into the stalk

  • Marco Mendola
  • Alessia Dolci
  • Lanfranco Piscopello
  • Giustino Tomei
  • Dario Bauer
  • Sabrina Corbetta
  • Bruno AmbrosiEmail author
Case report


We describe a patient affected by Cushing’s disease due to the presence of double pituitary adenomas, one located within the anterior pituitary and the other in the infundibulum associated with a remnant of Rakthe’s pouch. Cure was achieved only after the infundibulum lesion was surgically removed. CASE REPORT: A 38-year-old female presented with unexplained weight gain, hirsutism, amenorrhea, asthenia, recurrent cutaneous micotic infections and alopecia. Hormonal studies indicated Cushing’s disease and MRI showed an enlarged pituitary gland with a marked and homogeneous enhancement after injection of gadolinium and an enlarged infundibulum with a maximum diameter of 8 mm. As a venous sampling of the inferior petrosal sinus after 10 µg iv desmopressin stimulation revealed a central to peripheral ACTH ratio consistent with a pituitary ACTH-secreting tumor, transphenoidal explorative surgery was performed and a 4-mm pituitary adenoma immunopositive for ACTH was disclosed and removed. Since postoperative hormonal evaluation showed persistent hypercortisolism, confirmed by dynamic tests, the patient again underwent surgery by transcranial access and the infundibulum mass was removed. Histology and immunochemistry were consistent with an ACTH-secreting adenoma. A few months after the second operation, cushingoid features were significantly reverted and symptoms improved. CONCLUSION Although Cushing’s patients bearing multiple adenomas have already been documented, the presence of two adenomas both immunohistochemically positive for ACTH is a very rare cause of Cushing’s disease and this is the first report of a case of double ACTH-producing adenomas, one located in the pituitary gland and one attached to the stalk.

Key Words

Cushing’s disease Double ACTH-producing adenomas Magnetic resonance imaging Pituitary stalk 


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Copyright information

© Hellenic Endocrine Society 2014

Authors and Affiliations

  • Marco Mendola
    • 1
  • Alessia Dolci
    • 1
  • Lanfranco Piscopello
    • 2
  • Giustino Tomei
    • 3
  • Dario Bauer
    • 4
  • Sabrina Corbetta
    • 1
  • Bruno Ambrosi
    • 1
    Email author
  1. 1.Endocrinology and Diabetology Unit, Department of Biomedical Sciences for HealthUniversity of Milan, IRCCS Policlinico San DonatoMilanoItaly
  2. 2.Endocrinology UnitOspedale Maggiore TriesteTriesteItaly
  3. 3.Neurosurgical Unit, Department of BiotechnologyUniversity of InsubriaVareseItaly
  4. 4.Pathology Unit, Department of Health SciencesUniversity of Milan, San Paolo HospitalMilanoItaly

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