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Hormones

, 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 Ambrosi
Case report

Abstract

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 

References

  1. 1.
    Kontogeorgos G, Scheithauer BW, Horvath E, et al, 1992 Double adenomas of the pituitary: a clinicopathological study of 11 tumors. Neurosurgery 31: 840–849.CrossRefGoogle Scholar
  2. 2.
    Iacovazzo D, Bianchi A, Lugli F, et al, 2013 Double pituitary adenomas. Endocrine 43: 452–457.CrossRefGoogle Scholar
  3. 3.
    Magri F, Villa C, Locatelli D, et al, 2010 Prevalence of double pituitary adenomas in a surgical series: clinical, histological and genetic features. J Endocrinol Invest 33: 325–331.CrossRefGoogle Scholar
  4. 4.
    Kontogeorgos G, Kovacs K, Horvath E, Scheithauer BW, 1991 Multiple adenomas of the human pituitary. A retrospective autopsy study with clinical implication. J Neurosurg 74: 243–247.CrossRefGoogle Scholar
  5. 5.
    Kim K, Yamada S, Usui M, Sano T, 2004 Preoperative identification of clearly separated double pituitary adenomas. Clin Endocrinol 61: 26–30.CrossRefGoogle Scholar
  6. 6.
    Ratcliff JK, Oldfield EH, 2000 Multiple pituitary adenomas in Cushing’s disease. J Neurosurg 93: 753–761CrossRefGoogle Scholar
  7. 7.
    Sano T, Horiguchi H, Xu B, et al, 1999 Double pituitary adenomas: six surgical cases. Pituitary 1: 243–250.CrossRefGoogle Scholar
  8. 8.
    Koutourousiou M, Kontogeorgos G, Wesseling P, Grotenhuis AJ, Seretis A, 2010 Collision sellar lesions: experience with eight cases and review of the literature. Pituitary 13: 8–17.CrossRefGoogle Scholar
  9. 9.
    Syro LV, Horvath E, Kovacs K, 2000 Double adenoma of the pituitary: a somatotroph adenoma colliding with a gonadotroph adenoma. J Endocrinol Invest 23: 37–41.CrossRefGoogle Scholar
  10. 10.
    Tomita T, Gates E, 1999 Pituitary adenomas and granular cells tumors. Incidence, cell type and location in 100 pituitary glands at autopsy. Am J Clin Pathol 111: 817–825.CrossRefGoogle Scholar
  11. 11.
    Tosaka M, Kohga H, Kobayashi S, et al, 2000 Double pituitary adenomas detected on preoperative magnetic resonance images. Case illustration. J Neurosurg 92: 361.CrossRefGoogle Scholar
  12. 12.
    McKelvie PA, McNeill P, 2002 Double pituitary adenomas: a series of three patients. Pathology 34: 57–60.CrossRefGoogle Scholar
  13. 13.
    Shimizu C, Koike T, Sawamura Y, 2004 Double pituitary adenomas with distinct histological features and immunophenotypes. J Neurol Neurosurg Psychiatry 75: 140.PubMedPubMedCentralGoogle Scholar
  14. 14.
    Mohammed S, Cusimano MD, Scheithauer BW, Rotondo F, Horvath E, Kovacs K, 2010 O-Methylguanine-DNA methyltransferase immunoexpression in a double pituitary adenoma: case report. Neurosurgery 66: E421–E422.CrossRefGoogle Scholar
  15. 15.
    Cannavò S, Curto L, Lania A, Saccomanno K, Salpietro FM, Trimarchi F, 1999 Unusual MRI finding of multiple pituitary adenomas in the gland: a case report and review of the literature. Magn Reson Imaging 17: 633–636.CrossRefGoogle Scholar
  16. 16.
    Meij BP, Lopes MBS, Vance ML, Thorner MO, Laes ER Jr, 2001 Double pituitary lesions in three patients with Cushing’s disease. Pituitary 3: 159–168.CrossRefGoogle Scholar
  17. 17.
    Pantelia E, Kontogeorgos G, Piaditis G, Rologis D, 1998 Triple adenoma in Cushing’s disease: case report. Acta Neurochir 140: 190–193.CrossRefGoogle Scholar
  18. 18.
    Oyama K, Yamada S, Hukuhara N, Hiramatsu R, Taguchi M, Imai Y, 2006 FSH-producing macroadenoma associated in a patient with Cushing’s disease. Neuroendocrinol Lett 27: 733–736.PubMedGoogle Scholar
  19. 19.
    Andrioli M, Pecori Giraldi F, Losa M, Terreni M, Invitti C, Cavagnini F, 2010 Cushing’s disease due to double pituitary ACTH-secreting adenomas: the first case report. Endocr J 57: 833–837.CrossRefGoogle Scholar
  20. 20.
    Melmed S, 2003 Mechanism for pituitary tumorigenesis: the plastic pituitary. J Clin Invest 112: 1603–1618.CrossRefPubMedPubMedCentralGoogle Scholar
  21. 21.
    Hori A, Schmidt D, Kuebber S. 1999 Immunohistochemical survey of migration of human anterior pituitary cells in developmental, pathological, and clinical aspects: a review. Microscopy Research and Technique 46: 59–68.CrossRefGoogle Scholar
  22. 22.
    Kuebber S, Ropte S, Hori A, 1990 Proliferation of adenohypophyseal cells into posterior lobe. Their normal anatomical condition and possible neoplastic potentiality. Acta Neurochir 104: 21–26.CrossRefGoogle Scholar
  23. 23.
    Mason RB, Nieman LK, Doppman JL, Oldfield EH, 1997 Selective excision of adenomas originating in or extending into the pituitary stalk with preservation of pituitary function. J Neurosurg 87: 343–351.CrossRefGoogle Scholar

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
  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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