Abstract
On histology, IgG4-related hypophysitis is essentially similar to lymphocytic hypophysitis besides massive IgG4-positive plasmacyte infiltration. This immunohistochemical study was performed to investigate the presence of IgG4-positive plasmacytes in 14 various inflammatory lesions. Five cases of lymphocytic hypophysitis and a case of granulomatous hypophysitis were either negative or showed only a few IgG4-postive cells. A case of hypophysitis associated with pachymeningitis and a case of pituitary invasion of lymphoma were IgG4 negative. On the other hand, some IgG4-positive cells that tended to accumulate were observed in every six cases of secondary inflammation. In conclusion, immunohistochemistry for IgG4 can be a clue for differentiating IgG4-related hypophysitis from lymphocytic hypophysitis. However, clusters of IgG4-positive plasmacytes could be observed in secondary inflammation. Diagnosing hypophysitis with small biopsy specimen still remains troublesome.
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Bateman AC, Deheragoda MG. IgG4-related systemic sclerosing disease—an emerging and under-diagnosed condition. Histopathol 55: 373–378, 2009
Masaki Y, Dong L, Kurose N, Kitagawa K, Morikawa Y, Yamamoto M, Takahashi H, Shinomura Y, Imai K, Saeki T, Azumi A, Nakada S, Sugiyama E, Matsui S, Origuchi T, Nishiyama S, Nishimori I, Nojima T, Yamada K, Kawano M, Zen Y, Kaneko M, Miyazaki K, Tsubota K, Eguchi K, Tomoda K, Sawaki T, Kawanami T, Tanaka M, Fukushima T, Sugai S & Umehara H. Proposal of a new clinical entity, IgG4-positive multiorgan lymphoproliferative syndrome: analysis of 64 cases of IgG4-related disorders. Ann Rheum Dis 68: 1310–1315, 2009
Shimatsu A, Oki Y, Fujisawa I, Sano T. Pituitary and stalk lesions (infundibulo-neurohypophysitis) associated with immunoglobulin G4-related systemic disease: an emerging clinical entity. Endocr J 56: 1033–1041, 2009
Yamamoto M, Takahashi H, Ohara M, Suzuki C, Naishiro Y, Yamamoto H, Shinomura Y, Imai K. A new conseptualization for Mikulicz’s disease as an IgG4-related plasmacytic disease. Mod Rheumatol 16: 335–340, 2006
Fukuda W, Kimura M, Akaogi T, Sako M, Ohiwa K, Yamamoto Y, Kato G, Hayashi H, Yoshikawa T. Multifocal fibrosclerosis: retroperitoneal fibrosis associated with a suprasellar tumor and pachymeningitis. Inter Med 42: 1006–1010, 2003
Isaka Y, Yoshioka K, Nishio M, Yamagami K, Konishi Y, Inoue T, Hirano A, Hosoi M, Imanishi M. A case of IgG4-related multifocal fibrosclerosis complicated by central diabetes insipidus. Endocr J 55: 723–728, 2008
Osawa S, Ogawa Y, Watanabe M, Tominaga T. Hypophysitis presenting with atypical rapid deterioration: with special reference to immunoglobulin G4-related disease. Case report. Neurol Med-Chir (Tokyo) 49: 622–625, 2009
Tanabe T, Tsushima K, Yasuo M, Urushihata K, Hanaoka M, Koizumi T, Fujimoto K, Kubo K, Uehara T, Shigematsu S, Hamano H, Kawa S. IgG4-related multifocal systemic fibrosis complicating sclerosing sialadenitis, hypophysitis, and retroperitoneal fibrosis, but lacking pancreatic involvement. Intern Med 45: 1243–1247, 2006
van der Vliet HJJ, Perenboom RM. Multiple pseudotumors in IgG4-associated multifocal systemic fibrosis. Ann Intern Med 141: 896–897, 2004
Wong S, Lam WY, Wong WK, Lee KC. Hypophysitis presented as inflammatory pseudotumor in immunoglobulin G4-related systemic disease. Hum Pathol 38: 1720–1723, 2007
Yamamoto M, Takahashi H, Ohara M, Suzuki C, Naishiro Y, Yamamoto H, Shinomura Y, Imai K. A case of Mikulicz’s disease (IgG4-related plasmacytic disease) complicated by autoimmune hypophysitis. Scand J Rheumatol 35: 410–411, 2006
Gutenberg A, Buslei R, Fahrbusch R, Buchfelder M, Bruck W. Immunopathology of primary hypophysitis: implications for pathogenesis. Am J Surg Pathol 29: 329–338, 2005
Nishioka H, Ito H, Miki T, Wada J, Sano T. Lymphocytic adenohypophysitis associated with Rathke’s cleft cyst. Endocr Pathol 6: 337–343, 1995
Nishioka H, Ito H, Miki T, Akada K. A case of lymphocytic hypophysitis with massive fibrosis and the role of surgical intervention. Surg Neurol 42: 74–78, 1994
Nishioka H, Ito H, Sano T, Ito Y. Two cases of lymphocytic hypophysitis presenting with diabetes insipidus: a variant of lymphocytic infundibulo-neurohypophysitis. Surg Neurol 46: 285–291, 1997
Nishioka H, Ito H, Fukushima C. Recurrent lymphocytic hypophysitis: case report. Neurosurgery 41: 684–687, 1997
Murakami M, Nishioka H, Izawa H, Ikeda Y, Haraoka J. Granulomatous hypophysitis associated with Rathke’s cleft cyst; a case report. Minim Inv Neurosurg 51: 169–172, 2008
Nishioka H, Ito H, Haraoka J, Takahashi M, Shinmura F. Idiopathic hypertrophic cranial pachymeningitis of the cavernous sinus mimicking lymphocytic hypophysitis—case report. Neurol Med-Chir (Tokyo) 38: 377–382, 1998
Cheung CC, Ezzat S, Smyth HS, Asa SL. The spectrum and significance of primary hypophysitis. J Clin Endocrinol Metab 86: 1048–1053, 2001
Tashiro T, Sano T, Xu B, Wakatsuki S, Kagawa N, Nishioka H, Yamada S, Kovacs K. Spectrum of different type of hypophysitis: a clinicopathologic study of hypophysitis in 31 cases. Endocr Pathol 18: 183–195, 2002
Choi EK, Kim M-H, Lee TY, Kwon S, Oh H-C, Hwang CY, Seo DW, Lee SS, Lee SK. The sensitivity and specificity of serum immunoglobulin G and immunoglobulin G4 levels in the diagnosis of autoimmune chronic pancreatitis: Korean experience. Pancreas 35: 156–161, 2007
Burt MG, Morey AL, Turner JJ, Pell M, Sheehy JP, Ho KKY. Xanthomatous pituitary lesions: a report of two cases and review of the literature. Pituitary 6: 161–168, 2003
Nishioka H, Haraoka J, Izawa H, Ikeda Y. Magnetic resonance imaging, clinical manifestations, and management of Rathke’s cleft cyst. Clin Endocrinol 64: 184–188, 2006
Nishioka H, Shibuya M, Ohtsuka K, Ikeda Y, Haraoka J. Endocrinological and MRI features of pituitary adenomas with marked xanthogranulomatous reaction. Neuroradiol 2010 doi:10.1007/s00234-010-0675-8
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We declare that we have no conflict of interest that could be perceived as prejudicing the impartiality of the research reported. We did not receive any specific grant from any funding agency in the public, commercial, or not-for-profit sector. We are grateful to Prof. J. Patrick Barron of the Department of International Medical Communications of Tokyo Medical University for the review of this manuscript.
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Nishioka, H., Shibuya, M. & Haraoka, J. Immunohistochemical Study for IgG4-positive Plasmacytes in Pituitary Inflammatory Lesions. Endocr Pathol 21, 236–241 (2010). https://doi.org/10.1007/s12022-010-9128-5
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DOI: https://doi.org/10.1007/s12022-010-9128-5