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      Thickened Pituitary Stalk Associated with a Mass in the Sphenoidal Sinus: An Alarm to Suspect Hypophysitis by Immunoglobulin G4?

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          Abstract

          Introduction Hypophysitis is a chronic inflammation of the pituitary gland of complex and still incompletely defined pathogenesis. It belongs to the group of non-hormone-secreting sellar masses, sharing with them comparable clinical presentation and radiographic appearance.

          Objectives Describe the case of immunoglobulin G4 (IgG4)-related hypophysitis presenting as a mass in the sphenoid sinus.

          Resumed Report A 40-year-old Brazilian man had a diagnosis of central diabetes insipidus since 2001 associated with pituitary insufficiency. Pituitary magnetic resonance imaging revealed a centered pituitary stalk with focal nodular thickening and the presence of heterogeneous materials inside the sphenoid sinus. The patient was treated with testosterone replacement therapy. Laboratory results revealed increased IgG4 serum.

          Conclusion IgG4-related hypophysitis should be considered in patients with pituitary insufficiency associated with sellar mass and/or thickened pituitary stalk. IgG4 serum measurement for early diagnosis of IgG4-related hypophysitis should be performed.

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          A new clinicopathological entity of IgG4-related autoimmune disease.

          Terumi Kamisawa, Nobuaki Funata, Yukiko Hayashi … (2003)
          Autoimmune pancreatitis (AIP) is occasionally associated with other autoimmune diseases. To investigate the pathophysiology of AIP, we immunohistochemically examined the pancreas and other organs in eight patients with AIP, and in controls, using anti-CD4-T and CD8-T cell subsets, as well as IgG4 antibodies. In AIP patients, severe or moderate infiltration of IgG4-positive plasma cells associated with CD4- or CD8-positive T lymphocytes was detected in the peripancreatic tissue (6/6), bile duct (8/8), gallbladder (8/8), portal area of the liver (3/3), gastric mucosa (5/7), colonic mucosa (2/2), salivary glands (1/2), lymph nodes (6/6), and bone marrow (2/2), as well as in the pancreas (8/8). There were few IgG4-positive plasma cells at the same sites in controls. These results suggest that AIP is not simply pancreatitis but that it is a pancreatic lesion involved in IgG4-related systemic disease with extensive organ involvement. We propose a new clinicopathological entity, of a systemic IgG4-related autoimmune disease in which AIP and its associated diseases might be involved. Autoimmune pancreatitis (AIP) is occasionally associated with other autoimmune diseases.
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            Are Classification Criteria for IgG4-RD Now Possible? The Concept of IgG4-Related Disease and Proposal of Comprehensive Diagnostic Criteria in Japan

            Kazuichi Okazaki, Hisanori Umehara (2012)
            Recent studies suggest simultaneous or metachronous lesions in multiorgans characterized by elevated serum levels of IgG4 and abundant infiltration of IgG4-positive plasma cells with various degrees of fibrosis. Two Japanese research committees for IgG4-RD, one from fibrosclerosis (Okazaki team) and the other from lymph proliferation (Umehara team) supported by the “Research Program for Intractable Disease” of the Ministry of Health, Labor, and Welfare of Japan, have agreed with the unified nomenclature as “IgG4-RD” and proposed the comprehensive diagnostic criteria (CDC) for IgG4-RD. Validation of the CDC demonstrated satisfactory sensitivity for the practical use of general physicians and nonspecialists but low sensitivity in the organs to be difficult in taking biopsy specimens such as type1 autoimmune pancreatitis (IgG4-related AIP), compared with IgG4-related sialadenitis/dacryoadenitis (Mikulicz's disease) and IgG4-related kidney disease. Although the diagnostic criteria covering all IgG4-RD are hard to be established, combination with the CDC and organ-specific diagnostic criteria should improve sensitivity.
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              Hypophysitis presented as inflammatory pseudotumor in immunoglobulin G4-related systemic disease.

              Ricky Wong, Clement Lee, Vicky W. Y. Lam … (2007)
              Immunoglobulin (Ig) G4-related systemic disease is a recently characterized entity. The best-known manifestation is pancreatitis. Other systemic involvements are also described. Three cases of this disease with hypophyseal involvement have been reported in the literature, all diagnosed clinically. We herein present the first case of IgG4-related hypophysitis diagnosed histopathologically. The patient is a 77-year-old Chinese man with a pituitary tumor. Histologic examination of the resected tumor showed hypophysitis with features of inflammatory pseudotumor. Clinical review showed history of pancreatitis and cholecystitis 4 years ago. The pancreatic biopsy and gall bladder specimens obtained previously had the same pathologic features of inflammatory pseudotumor. Immunohistochemistry highlighted abundant IgG4-positive plasma cells in all 3 specimens. Serum IgG4 level was also elevated. A diagnosis of IgG4-related systemic disease was confirmed. This is the first case of intracranial inflammatory pseudotumor shown to be associated with IgG4-related systemic disease.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Int Arch Otorhinolaryngol
                Journal ID (iso-abbrev): Int Arch Otorhinolaryngol
                Journal ID (doi): 10.1055/s-00025477
                Title: International Archives of Otorhinolaryngology
                Publisher: Thieme Publicações Ltda (Rio de Janeiro, Brazil )
                ISSN (Print): 1809-9777
                ISSN (Electronic): 1809-4864
                Publication date (Electronic): 05 March 2015
                Publication date (Print): July 2015
                Volume: 19
                Issue: 3
                Pages: 273-276
                Affiliations
                [1 ]Neurosurgical Unit, Universidade de São Paulo, São Paulo, SP, Brazil
                [2 ]Funcional Neurosurgery Unit, Universidade de São Paulo, São Paulo, SP, Brazil
                Author notes
                Address for correspondence Rafael Loch Batista, MD Neurosurgical Unit, USP Alameda Joauime Eugênio de Lima, 1058/102, Sao Paulo, Sao Paulo 01403002Brazil r-loch@ 123456uol.com.br rafael.loch@ 123456hc.fm.usp.br
                Article
                Publisher ID: 0178
                DOI: 10.1055/s-0034-1397333
                PMC ID: 4490927
                SO-VID: fb768b07-4e17-41ab-ae73-de15a80e1e9e
                Copyright © © Thieme Medical Publishers
                History
                Date received : 03 October 2014
                Date accepted : 24 November 2014
                Categories
                Subject: Article

                Keywords: pituitary diseases,hypopituitarism,sphenoid sinus
                Data availability:
                Keywords: pituitary diseases, hypopituitarism, sphenoid sinus

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