Abstract
Herein, we report a unique case of an adult male with a corticotrophic pituitary adenoma of silent subtype 1 exhibiting conspicuous idiopathic tumoral noncaseating granulomatous inflammation. The lesion was unassociated with clinical or laboratory evidence of either systemic sarcoidosis or infection. Histochemical and polymerase chain reaction (PCR) studies revealed neither fungi nor tubercle bacilli. We suggest that tumoral production of an as yet uncharacterized antigen may have induced the granulomatous inflammatory reaction.
Similar content being viewed by others
References
Goodman RH, Post KD, Molitch ME, Adelman LS, Altemus LR, Johnston H. Eosinophilic granuloma mimicking a pituitary tumor. Neurosurgery 5 6:723–5, 1979.
Kaltsas GA, Powles TB, Evanson J, Plowman PN, Drinkwater JE, Jenkins PJ, et al. Hypothalamo–pituitary abnormalities in adult patients with langerhans cell histiocytosis: clinical, endocrinological, and radiological features and response to treatment. J Clin Endocrinol Metab 85 4:1370–6, 2000.
Scolozzi P, Lombardi T, Monnier P, Jaques B. Multisystem Langerhans’ cell histiocytosis (Hand–Schuller–Christian disease) in an adult: a case report and review of the literature. Eur Arch Otorhinolaryngol 261 6:326–30, 2004.
Flanagan DE, Ibrahim AE, Ellison DW, Armitage M, Gawne-Cain M, Lees PD. Inflammatory hypophysitis—the spectrum of disease. Acta Neurochir (Wien) 144 1:47–56, 2002.
Leung GK, Lopes MB, Thorner MO, Vance ML, Laws ER Jr. Primary hypophysitis: a single-center experience in 16 cases. J Neurosurg 101 2:262–71, 2004.
Murakami K, Muraishi K, Ikeda H, Yoshimoto T. Plasma cell granuloma of the pituitary gland. Case report. Surg Neurol 56 4:247–51, 2001.
Chapelon C, Ziza JM, Piette JC, Levy Y, Raguin G, Wechsler B, et al. Neurosarcoidosis: signs, course and treatment in 35 confirmed cases. Medicine (Baltimore) 69 5:261–76, 1990.
Murialdo G, Tamagno G. Endocrine aspects of neurosarcoidosis. J Endocrinol Invest 25 7:650–62, 2002.
Tabuena RP, Nagai S, Handa T, Shigematsu M, Hamada K, Ito I, et al. Diabetes insipidus from neurosarcoidosis: long-term follow-up for more than eight years. Intern Med 43 10:960–6, 2004.
Holck S, Laursen H. Prolactinoma coexistent with granulomatous hypophysitis. Acta Neuropathol (Berl) 61 3–4:253–7, 1983.
Desai KI, Nadkarni TD, Goel A. Tuberculomas of the hypophysis cerebri: report of five cases. J Clin Neurosci 10 5:562–6, 2003.
Florakis D, Kontogeorgos G, Anapliotou M, Mazarakis N, Richter E, Bruck W, et al. Isolated pituitary granuloma by atypical Mycobacterium in a nonimmunosuppressed woman. Clin Endocrinol (Oxf) 56 1:123–6, 2002.
Bhadada S, Bhansali A, Nahar U, Reddy KS, Pathak A, Dutta P, et al. An unusual association of acromegaly and pituitary tuberculosis. Pituitary 7 2:103–6, 2004.
Rubin MR, Bruce JN, Khandji AG, Freda PU. Sarcoidosis within a pituitary adenoma. Pituitary 4 3:195–202, 2001.
Kovacs K, Giannini C, Scheithauer BW, Stefaneanu L, Lloyd RV, Horvath E. Pituitary changes in ataxia–telangiectasia syndrome: an immunocytochemical, in situ hybridization, and DNA cytometric study of three cases. Endocr Pathol 8 3:195–203, 1997.
Kovacs K, Stefaneanu L, Ezzat S, Smyth HS. Prolactin-producing pituitary adenoma in a male-to-female transsexual patient with protracted estrogen administration. A morphologic study. Arch Pathol Lab Med 118 5:562–5, 1994.
Buckwalter SP, Wengenack NL. Detection of myocobacteria in paraffin-embedded tissue using LighyCycler PCR. New Orleans, LA, Abstracts of the Annual Meeting of the American Society for Microbiology, 104th General Meeting, 2004, May 23–27.
Honegger J, Fahlbusch R, Bornemann A, Hensen J, Buchfelder M, Muller M, et al. Lymphocytic and granulomatous hypophysitis: experience with nine cases. Neurosurgery 40 4:713–22, discussion 722–3, 1997.
Paulus W, Honegger J, Keyvani K, Fahlbusch R. Xanthogranuloma of the sellar region: a clinicopathological entity different from adamantinomatous craniopharyngioma. Acta Neuropathol (Berl) 97 4:377–82, 1999.
Yokoyama S, Sano T, Tajitsu K, Kusumoto K. Xanthogranulomatous hypophysitis mimicking a pituitary neoplasm. Endocr Pathol 15 4:351–7, 2004.
Bhansali A, Velayutham P, Radotra BD, Pathak A. Idiopathic granulomatous hypophysitis presenting as non-functioning pituitary adenoma: description of six cases and review of literature. Br J Neurosurg 18 5:489–94, 2004.
Vittaz L, Ramanoelina J, Mahr A, Cohen R, Cohen P, Reach G, et al. [Pituitary involvement in Wegener’s granulomatosis. Two cases]. Presse Med 33 22:1585–90, 2004.
Roberts GA, Eren E, Sinclair H, Pelling M, Burns A, Bradford R, et al. Two cases of Wegener’s granulomatosis involving the pituitary. Clin Endocrinol (Oxf) 42 3:323–8, 1995.
Woywodt A, Knoblauch H, Kettritz R, Schneider W, Gobel U. Sudden death and Wegener’s granulomatosis of the pituitary. Scand J Rheumatol 29 4:264–6, 2000.
Toth M, Szabo P, Racz K, Szende B, Balogh I, Czirjak S, et al. Granulomatous hypophysitis associated with Takayasu’s disease. Clin Endocrinol (Oxf) 45 4:499–503, 1996.
Oweity T, Scheithauer BW, Ching HS, Lei C, Wong KP. Multiple system Erdheim–Chester disease with massive hypothalamic-sellar involvement and hypopituitarism. J Neurosurg 96 2:344–51, 2002.
Reithmeier T, Trost HA, Wolf S, Stolzle A, Feiden W, Lumenta CB. Xanthogranuloma of the Erdheim–Chester type within the sellar region: case report. Clin Neuropathol 21 1:24–8, 2002.
Deodhare SS, Bilbao JM, Kovacs K, Horvath E, Nomikos P, Buchfelder M, et al. Xanthomatous hypophysitis: a novel entity of obscure etiology. Endocr Pathol 10 3:237–41, 1999.
Chanson P, Timsit J, Kujas M, Violante A, Guillausseau PJ, Derome PJ, et al. Pituitary granuloma and pyoderma gangrenosum. J Endocrinol Invest 13 8:677–81, 1990.
Arunkumar MJ, Rajshekhar V. Intrasellar tuberculoma presenting as pituitary apoplexy. Neurol India 49 4:407–10, 2001.
Gazioglu N, Ak H, Oz B, Seckin MS, Kuday C, Sarioglu AC. Silent pituitary tuberculoma associated with pituitary adenoma. Acta Neurochir (Wien) 141 7:785–6, 1999.
Sharma MC, Vaish S, Arora R, Gaikwad S, Sarkar C. Composite pituitary adenoma and intrasellar tuberculoma: report of a rare case. Pathol Oncol Res 7 1:74–6, 2001.
Roncaroli F, Bacci A, Frank G, Calbucci F. Granulomatous hypophysitis caused by a ruptured intrasellar Rathke’s cleft cyst: report of a case and review of the literature. Neurosurgery 43 1:146–9, 1998.
Hama S, Arita K, Nishisaka T, Fukuhara T, Tominaga A, Sugiyama K, et al. Changes in the epithelium of Rathke cleft cyst associated with inflammation. J Neurosurg 96 2:209–16, 2002.
Komoribayashi N, Arai H, Kubo Y, Beppu T, Ogasawara K, Sugai T, et al. [Granulomatous change of the pituitary stalk caused by Rathke’s cleft cyst: a case report]. No Shinkei Geka 31 11:1193–6, 2003.
Kovacs J, Varga A, Bessenyei M, Gomba S. Renal cell cancer associated with sarcoid-like reaction. Pathol Oncol Res 10 3:169–71, 2004.
Coyne JD. Colonic carcinoma with granulomatous (sarcoid) reaction. J Clin Pathol 55 9:708–9, 2002.
Kashiwabara K, Toyonaga M, Yamaguchi Y, Nakamura H, Hirayama S, Kurano R. Sarcoid reaction in primary tumor of bronchogenic large cell carcinoma accompanied with massive necrosis. Intern Med 40 2:127–30, 2001.
Saeger W, Hofmann BM, Buslei R, Buchfelder M. Silent ACTH cell adenoma in coincidence with granulomatous hypophysitis—a case report. Pathol Res Pract 203 4:221–5, 2007.
Agostini C, Basso U, Semenzato G. Cells and molecules involved in the development of sarcoid granuloma. J Clin Immunol 18 3:184–92, 1998.
Co DO, Hogan LH, Il-Kim S, Sandor M. T cell contributions to the different phases of granuloma formation. Immunol Lett 92 1–2:135–42, 2004.
Acknowledgment
The authors express their appreciation to Seanne P. Buckwalter, M.S. and Dr. Nancy L. Wengenack of the Division of Microbiology, Mayo Clinic, for performance of PCR reactions and to Mr. and Mrs. Jarislowsky and the Lloyd Carr-Harris Foundations for their generous financial support. We also acknowledge the expert secretarial support of Mrs. Denise Chase.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Scheithauer, B.W., Silva, A.I., Atkinson, J.L.D. et al. Pituitary Adenoma with Tumoral Granulomatous Reaction. Endocr Pathol 18, 86–90 (2007). https://doi.org/10.1007/s12022-007-0017-5
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12022-007-0017-5