Abstract
Infarction of prolactin-secreting or growth hormone-secreting pituitary adenomas is not unusual. However, Infarction of ACTH-secreting adenomas has rarely been reported. Cyclical course of Cushing's syndrome alternating with adrenal insufficiency due to recurrent infarction of an ACTH-secreting pituitary adenoma has not been reported. We report here a 20-year-old lady who presented with florid signs of Cushing's syndrome but was found to have adrenal insufficiency on biochemical evaluation. Magnetic resonance imaging (MRI) of the pituitary gland showed that she had infarction of an ACTH-secreting macroadenoma. Over the next 6 years, her disease ran a cyclical course characterized by periods of hypercortisolism alternating with adrenal insufficiency due to repeated episodes of infarctions of the ACTH-secreting pituitary macroadenoma with corresponding changes in the pituitary adenoma on serial MRIs. The case alerts clinicians to this possibility when a patient presents with clinical picture of Cushing's syndrome but has adrenal insufficiency on biochemical testing. It also suggests that silent or subclinical infarction of pituitary adenomas is not uncommon and is probably under diagnosed.
Similar content being viewed by others
References
Mohr G, Hardy J. Hemorrhage, necrosis, and apoplexy in pituitary adenomas. Surg Neurol 1982;18:181–189.
Wakai S, Fukushima T, Teramoto A, Sano K. Pituitary apoplexy: Its incidence and clinical significance. J Neurosurg 1981;55:187–193.
Randeva HS, Schoebel J, Byrne J, Esiri M, Adams CB, Wass JA. Classical pituitary apoplexy: Clinical features, management and outcome. Clin Endocrinol 1999;51:181–188.
Rovit RL, Fein JM. Pituitary apoplexy: A review and reappraisal. J Neurosurg 1972;37:280–288.
Dickstein G, Arad E, Shechner C. Late complications in remission from Gushing disease. Recurrence of tumor with reinfarction or transformation into a silent adenoma. Arch Intern Med 1997;157:2377–2380.
Onesti ST, Wisniewski T, Post KD. Clinical versus subclinical pituitary apoplexy: Presentation, surgical management, and outcome in 21 patients. Neurosurgery 1990;26:980–986.
Biousse V, Newman NJ, Oyesiku NM. Precipitating factors in pituitary apoplexy. J Neurol Neurosurg Psychiatry 2001;71:542–545.
Kamoi K, Toyama M, Sudo N. A case of Cushing's disease revealed six years after postpartum hypopituitarism. J Clin Endocrinol Metab 1999;84:2718–2723.
Mercado-Asis LB, Oldfield EH, Cutler GB Jr. Pituitary tumor hemorrhage in Gushing disease. Ann Intern Med 1995;122:189–190.
Le Nestour E, Abecassis JP, Bertagna X, Bonnin A, Luton JP. Silent necrosis of a pituitary corticotroph adenoma revealed by timely magnetic resonance imaging: A cause of spontaneous remission of Cushing's disease. Eur J Endocrinol 1994;130:469–471.
Findling JW, Tyrrell JB, Aron DC, Fitzgerald PA, Wilson CB, Forsham PH. Silent pituitary apoplexy: Subclinical infarction of an adrenocorticotropin-producing pituitary adenoma. J Clin Endocrinol Metab 1981;52:95–97.
Vella A, Young W. Pituitary Apoplexy. The Endocrinologist 2001;11:282–288.
Rotman-Pikielny P, Patronas N, Papanicolaou DA. Pituitary apoplexy induced by corticotrophin-releasing hormone in a patient with Cushing's disease [see comment]. Clin Endocrinol 2003;58:545–549.
Reid RL, Quigley ME, Yen SS. Pituitary apoplexy. A review. Arch Neurol 1985;42:712–719.
Pelkonen R, Kuusisto A, Salmi J, Eistola P, Raitta C, Karonen SL, et al. Pituitary function after pituitary apoplexy. Am J Med 1978;65:773–778.
Veldhuis JD, Hammond JM. Endocrine function after spontaneous infarction of the human pituitary: Report, review, and reappraisal. Endocr Rev 1980;1:100–107.
Arafah BM, Harrington JF, Madhoun ZT, Selman WR. Improvement of pituitary function after surgical decompression for pituitary tumor apoplexy. J Clin Endocrinol Metab 1990;71:323–328.
Orth DN. Differential diagnosis of Cushing's syndrome [comment]. N Engl J Med 1991;325:957–959.
Cizza G, Nieman LK, Doppman JL, Passaro MD, Czerwiec FS, Chrousos GP, et al. Factitious Cushing syndrome. J Clin Endocrinol Metab 1996;81:3573–3577.
Lin CL, Wu TJ, Machacek DA, Jiang NS, Kao PC. Urinary free cortisol and cortisone determined by high performance liquid chromatography in the diagnosis of Cushing's syndrome. J Clin Endocrinol Metab 1997;82:151–155.
Lacroix A, Bolte E, Tremblay J, Dupre J, Poitras P, Fournier H, et al. Gastric inhibitory polypeptide-dependent cortisol hypersecretion—A new cause of Cushing's syndrome [see comment]. N Engl J Med 1992;327:974–980.
Reznik Y, Allali-Zerah V, Chayvialle JA, Leroyer R, Leymarie P, Travert G, et al. Food-dependent Cushing's syndrome mediated by aberrant adrenal sensitivity to gastric inhibitory polypeptide [see comment]. N Engl J Med 1992;327:981–986.
Lacroix A, Ndiaye N, Tremblay J, Hamet P. Ectopic and abnormal hormone receptors in adrenal Cushing's syndrome. Endocr Rev 2001;22:75–110.
Atkinson AB, Kennedy AL, Carson DJ, Madden DR, Weaver JA, Sheridan B. Five cases of cyclical Cushing's syndrome. Br Med J (Clin Res Ed) 1985;291:1453–1457.
Mercado-Asis LB, Oldfield EH, Cutler GB Jr. Pituitary tumor hemorrhage in Gushing disease. Ann Intern Med 1995;122:189–190.
Kamiya Y, Jin-No Y, Tomita K, Suzuki T, Ban K, Sugiyama N, et al. Recurrence of Cushing's disease after long-term remission due to pituitary apoplexy. Endocr J 2000;47:793–797.
Sasaki M, Funayama H, Asano T, Kasono K, Namai K, Tamemoto H, et al. Full-blown Cushing's disease after an episode of pituitary apoplexy. Endocr J 2003;50:501–506.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Alarifi, A., Alzahrani, A.S., Salam, S.A. et al. Repeated Remissions of Cushing's Disease Due to Recurrent Infarctions of an ACTH-Producing Pituitary Macroadenoma. Pituitary 8, 81–87 (2005). https://doi.org/10.1007/s11102-005-2961-8
Published:
Issue Date:
DOI: https://doi.org/10.1007/s11102-005-2961-8