Abstract
Double pituitary adenomas are rare in surgical specimens and the most common clinical feature in reported patients has been acromegaly. We report 3 cases of double pituitary lesions in patients who presented with Cushing's disease. In a 22-year-old man (case 1) with delayed puberty and low testosterone levels, mild hyperprolactinemia was diagnosed and treated with dopamine agonist therapy that reduced the prolactin (PRL) levels to normal. Over a 1-year period Cushing's disease developed gradually and was confirmed with classical endocrine testing. In a 27-year-old woman (case 2) who initially presented with severe depression and morbid obesity there was a gradual onset of Cushing's disease; initially she had minimally elevated serum PRL. In a 33-year-old woman (case 3) there was a 2-year history of Cushing's disease characterized by hirsutism, hypertension and weight gain; serum PRL was normal. Magnetic resonance imaging in all 3 patients revealed a microadenoma that was successfully removed by transsphenoidal pituitary surgery. Histology and immunocytochemistry in case 1 and case 3 revealed a corticotroph cell adenoma and a PRL cell adenoma in separate areas of the pituitary. In case 3 the PRL cell adenoma was “silent” but in case 1 the PRL cell adenoma may have been the cause of the mild hyperprolactinemia. In case 2 nodular corticotroph hyperplasia was the cause of Cushing's disease and a “silent” PRL cell adenoma was also identified.
We conclude from these cases and a literature review that double pituitary lesions may occur in patients with Cushing's disease. The corticotroph part of the double lesion may consist of a corticotroph cell adenoma or, as reported in this study, of corticotroph nodular hyperplasia. The counterpart of the double lesion may consist either of a “silent” PRL cell adenoma or a functional PRL cell adenoma causing hyperprolactinemia.
Similar content being viewed by others
References
Kontogeorgos G, Scheithauer BW, Horvath E, Kovacs K, Lloyd RV, Smyth HS, Rologis D. Double adenomas of the pituitary: a clinicopathological study of 11 tumors. Neurosurgery 1992;31:840–849.
Kontogeorgos G, Kovacs K, Horvath E, Scheithauer BW. Multiple adenomas of the human pituitary. A retrospective autopsy study with clinical implications. J Neurosurg 1991;74:243–247.
Tomita T, Kuziez M, Watanabe I. Double tumors of anterior and posterior pituitary gland. Acta Neuropathol (Berl) 1981;54:161–164.
Tomita T, Gates E. Pituitary adenomas and granular cell tumors. Incidence, cell type, and location in 100 pituitary glands at autopsy. Am J Clin Pathol 1999;111:817–825.
Sano T, Horiguchi H, Xu B, Li C, Hino A, Sakaki M, Kannuki S, Yamada S. Double pituitary adenomas: six surgical cases. Pituitary 1999;1:243–250.
Scheithauer BW, Horvath E, Kovacs K, Laws ER Jr, Randall RV, Ryan N. Plurihormonal pituitary adenomas. Semin Diagn Pathol 1986;3:69–82.
Kannuki S, Matsumoto K, Sano T, Shintani Y, Bando H, Saito S. Double pituitary adenoma—two case reports. Neurol Med Chir (Tokyo) 1996;36:818–821.
Wynne AG, Scheithauer BW, Young WF Jr, Kovacs K, Ebersold MJ, Horvath E. Coexisting corticotroph and lactotroph adenomas: case report with reference to the relationship of corticotropin and prolactin excess. Neurosurgery 1992;30:919–923.
Blevins LS, Jr, Hall GS, Madoff DG, Laws ER, Jr, Wand CS. Case report: Acromegaly and Cushing's disease in a patient with synchronous pituitary adenomas. Am J Med Sci 1992;304:294–297.
Booth GL, Redelmeier DA, Grosman H, Kovacs K, Smyth HS, Ezzat S. Improved diagnostic accuracy of inferior petrosal sinus sampling over imaging for localizing pituitary pathology in patients with Cushing's disease. J Clin Edocrinol Metab 1998;83:2291–2295.
Pantelia E, Kontogeorgos G, Piaditis G, Rologis D. Triple adenoma in Cushing's disease: case report. Acta Neurochir (Wien) 1998;140:190–193.
Schulte HM, Petersenn S. Cushing's disease: clinical findings and edocrinology. In: Landolt AM, Vance ML, Reilly PL, eds. Pituitary Adenomas. New York: Churchill Livingstone, 1996:101–110.
Graham KE, Samuels MH. Cushing's disease. In: Krisht AF, Tindall GT, eds. Pituitary Disorders. Comprehensive Management. Baltimore: Lippincott Williams & Wilkins, 1999:209–224.
Friend KE, Chiou YK, Lopes MB, Laws ER Jr, Hughes KM, Shupnik MA. Estrogen receptor expression in human pituitary: correlation with immunohistochemistry in normal tissue, and immunohistochemistry and morphology in macroadenomas. J Clin Endocrinol Metab 1994;78:1497–1504.
Thorner MO. Hyperprolactinaemia. In: Besser GM, Thorner MO, eds. Clinical Endocrinology. London: Mosby-Wolfe, 2nd Edition, 1994:4.1–4.14.
Arafah BM, Kailani SH, Nekl KE, Gold RS, Selman WR. Immediate recovery of pituitary function after transsphenodial resection of pituitary macroadenomas. J Clin Endocrinol Metab 1994;79:348–354.
Tosaka M, Kohga H, Kobayashi S, Zama A, Tamura M, Murakami M, Sasaki T. Double pituitary adenomas detected on preoperative magnetic resonance images. Case illustration. J Neurosurg 2000;92:361.
Cannavo S, Curto L, Lania A, Saccomanno K, Salpietro FM, Trimarchi F. Unusual MRI finding of multiple adenomas in the pituitary gland: a case report and review of the literature. Magnetic Resonance Imaging 1999;17:633–636.
Salpietro FM, Alafaci C, Grasso G, Lucerna S, Passalacqua M, Tomasello F. Transsphenoidal microsurgical selective removal of multiple (triple) adenomas of the pituitary gland. Acta Neurochir (Wien) 1999;141:425–428.
Mahler CH, Verhelst J, Klaes R. Cushing's disease and hyperprolactinemia due to a mixed ACTH-and prolactin-secreting pituitary macroadenoma. Pathol Res Pract 1991;187:598–602.
Sherry SH, Guay AT, Lee AK, Hedley-Whyte ET, Felderman M, Freidberg SR, Woolf PD. Concurrent production of adrenocorticotropin and prolactin from two distinct cell lines in a single pituitary adenoma: a detailed immunohistochemical analysis. J Clin Endocrinol Metab 1982;55:947–955.
Horvath E, Kovacs K, Scheithauer BW. Pituitary hyperplasia. Pituitary 1999;1:169–180.
Asa SL, Ezzat S. Molecular determinants of pituitary cytodifferentiation. Pituitary 1999;1:159–168.
Author information
Authors and Affiliations
Rights and permissions
About this article
Cite this article
Meij, B.P., Lopes, M.B.S., Vance, M.L. et al. Double Pituitary Lesions in Three Patients with Cushing's Disease. Pituitary 3, 159–168 (2000). https://doi.org/10.1023/A:1011499609096
Issue Date:
DOI: https://doi.org/10.1023/A:1011499609096