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      Spontaneous Resolution of IgG4-Related Hepatic Inflammatory Pseudotumor Mimicking Malignancy

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          Abstract

          Hepatic inflammatory pseudotumor (IPT) is characterized by a well-circumscribed benign tumor mimicking or often mistaken for a malignant lesion. A 48-year-old male presented to the hospital with complaints of epigastric pain, with initial laboratory findings showing mildly elevated alkaline phosphatase (140 U/L) with normal AST, ALT, bilirubin, and lipase, a CD4 count of 384, and an HIV viral load of > 10 million copies. The total IgG level was elevated to 2,228 mg/dL (normal IgG4 level 114 mg/dL). Contrast-enhanced MRI of the abdomen showed heterogeneous mass-like infiltration in the right lobe of the liver measuring 9.6 cm. The liver mass was biopsied which showed dense collagenous fibrosis with abundant lymphoplasmacytic infiltrates with 18 IgG4-positive plasma cells per high-power field. The patient was not given any treatment for this IPT. For more than 1 year of follow-up triple-phase CT scan of the liver was repeated, which showed no liver mass. As radiological images of hepatic IPTs, including IgG4-related hepatic IPT, mimic liver malignancy, histological analysis of the biopsy remains the cornerstone for the diagnosis. Symptomatic patients with IgG4-related hepatic IPT have shown improvement with corticosteroid use; however, spontaneous resolution has also been reported like in the present case.

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          Most cited references17

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          Comprehensive diagnostic criteria for IgG4-related disease (IgG4-RD), 2011

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            IgG4-related sclerosing disease.

            Based on histological and immunohistochemical examination of various organs of patients with autoimmune pancreatitis (AIP), a novel clinicopathological entity of IgG4-related sclerosing disease has been proposed. This is a systemic disease that is characterized by extensive IgG4-positive plasma cells and T-lymphocyte infiltration of various organs. Clinical manifestations are apparent in the pancreas, bile duct, gallbladder, salivary gland, retroperitoneum, kidney, lung, and prostate, in which tissue fibrosis with obliterative phlebitis is pathologically induced. AIP is not simply pancreatitis but, in fact, is a pancreatic disease indicative of IgG4-related sclerosing diseases. This disease includes AIP, sclerosing cholangitis, cholecystitis, sialadenitis, retroperitoneal fibrosis, tubulointerstitial nephritis, interstitial pneumonia, prostatitis, inflammatory pseudotumor and lymphadenopathy, all IgG4-related. Most IgG4-related sclerosing diseases have been found to be associated with AIP, but also those without pancreatic involvement have been reported. In some cases, only one or two organs are clinically involved, while in others, three or four organs are affected. The disease occurs predominantly in older men and responds well to steroid therapy. Serum IgG4 levels and immunostaining with anti-IgG4 antibody are useful in making the diagnosis. Since malignant tumors are frequently suspected on initial presentation, IgG4-related sclerosing disease should be considered in the differential diagnosis to avoid unnecessary surgery.
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              Pathological classification of hepatic inflammatory pseudotumor with respect to IgG4-related disease.

              Recently, much attention has focused on IgG4-related disease, which is characterized by abundant IgG4-positive plasma cell infiltration and high serum IgG4 levels. IgG4-related disease sometimes manifests as tumorous lesions, and its relationship to inflammatory pseudotumor has been suggested. In this study, we examined clinicopathological features of a total of 16 cases of hepatic inflammatory pseudotumor (11 men and 5 women with an average age of 67 years) with respect to IgG4-related disease. The tumors could be pathologically classified into two types: fibrohistiocytic (10 cases) and lymphoplasmacytic (6 cases). Fibrohistiocytic inflammatory pseudotumors were characterized by xanthogranulomatous inflammation, multinucleated giant cells, and neutrophilic infiltration, and mostly occurred in the peripheral hepatic parenchyma as mass-forming lesions. In contrast, lymphoplasmacytic inflammatory pseudotumors showed diffuse lymphoplasmacytic infiltration and prominent eosinophilic infiltration, and were all found around the hepatic hilum. In addition, venous occlusion with little inflammation and cholangitis without periductal fibrosis were frequently observed in the fibrohistiocytic type, whereas obliterative phlebitis and cholangitis with periductal fibrosis were common features of the lymphoplasmacytic type. Interestingly, IgG4-positive plasma cells were significantly more numerous in the lymphoplasmacytic than fibrohistiocytic type. However, two of the fibrohistiocytic inflammatory pseudotumors had relatively many IgG4-positive plasma cells. In conclusion, hepatic inflammatory pseudotumor could be classified into two types based on clinicopathological characteristics. The lymphoplasmacytic type is unique, and could belong to the so-called IgG4-related diseases. In contrast, the fibrohistiocytic type might still be a heterogeneous group of disorders. This latter type seems pathologically different from IgG4-related disease, although cases with relatively abundant IgG4-positive plasma cells should be differentiated from IgG4-related disease with secondary histopathologic modifications.
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                Author and article information

                Journal
                CRG
                CRG
                10.1159/issn.1662-0631
                Case Reports in Gastroenterology
                S. Karger AG
                1662-0631
                2018
                May – August 2018
                22 June 2018
                : 12
                : 2
                : 311-316
                Affiliations
                [_a] aDepartment of Gastroenterology and Hepatology, St. Joseph’s University Medical Center, Paterson, New Jersey, USA
                [_b] bDepartment of Internal Medicine, St. Joseph’s University Medical Center, Paterson, New Jersey, USA
                Author notes
                *Sushant Nanavati, Department of Internal Medicine, St. Joseph’s University Medical Center, Xavier 5, 703 Main Street, Paterson, NJ 07503 (USA), E-Mail nanavats@sjhmc.org
                Article
                490100 PMC6047548 Case Rep Gastroenterol 2018;12:311–316
                10.1159/000490100
                PMC6047548
                30022921
                6aad6003-27ad-43ff-a154-1b333f2d9f2a
                © 2018 The Author(s). Published by S. Karger AG, Basel

                This article is licensed under the Creative Commons Attribution-NonCommercial 4.0 International License (CC BY-NC). Usage and distribution for commercial purposes requires written permission. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

                History
                : 27 April 2018
                : 07 May 2018
                Page count
                Figures: 2, Tables: 1, Pages: 6
                Categories
                Single Case

                Oncology & Radiotherapy,Gastroenterology & Hepatology,Surgery,Nutrition & Dietetics,Internal medicine
                HIV-positive patient,Storiform fibrosis,Inflammatory pseudotumor,IgG4-related diseases,Lymphoplasmacytic infiltrate

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