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      Autoimmune hepatitis in a patient with immunoglobulin A nephropathy: A case report

      case-report

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          Abstract

          BACKGROUND

          Immunoglobulin A nephropathy (IgAN) is the most commonly encountered glomerular disease in Asian countries. It has a broad clinical presentation, and it is frequently associated with other conditions. Chronic liver disease is well recognized as the leading cause of secondary IgAN. However, cases of IgAN associated with autoimmune hepatitis (AIH) have seldom been reported.

          CASE SUMMARY

          A 63-year-old Korean woman was admitted to Pusan National University Hospital for an evaluation of abdominal pain and elevated liver enzymes. Two weeks prior, she had presented to our hospital with proteinuria of approximately 1350 mg/d and hematuria and was diagnosed with IgAN. Autoimmune profiles were highly positive for antinuclear antibodies, and symptoms related to portal hypertension including ascites and peripheral edema were present. A diagnosis of AIH was made according to the simplified scoring system of the International Autoimmune Hepatitis Group. Despite immunosuppression with prednisolone and azathioprine, rapid deterioration of liver function led to end-stage liver disease. After a living-donor liver transplantation, liver function gradually improved, and she had maintained stable liver and kidney function at the six months follow-up.

          CONCLUSION

          Cases of secondary IgAN with chronic liver disease have been frequently reported in the literature but are rarely associated with AIH. We encountered an IgAN patient with concurrent progressive liver failure due to AIH.

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          Most cited references13

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          Autoimmune hepatitis--Update 2015.

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            Receptor-mediated binding and uptake of immunoglobulin A by human liver.

            We have studied the molecular mechanisms of the binding and uptake of secretory and serum immunoglobulin A (IgA) of both subclasses (1 and 2) and molecular forms (monomer and polymer) by the particulate fraction of human liver homogenate and by a human hepatoma cell line (HepG2). Inhibition by asialoorosomucoid and the requirement for the presence of calcium indicated that the binding of secretory IgA and polymeric IgA1 was mediated by the asialoglycoprotein receptor. Secretory component, which functions as a receptor for polymeric IgA in several animal species, was detected in the epithelial cells of bile ducts, but not in hepatocytes. Secretory IgA and all molecular forms and subclasses of serum IgA were bound by HepG2 cells, which do not express secretory component. The requirement for the presence of calcium, the presence of a terminal galactose residue in IgA, and the molecular weight of the major plasma membrane protein responsible for binding (41,700 daltons) indicated the involvement of asialoglycoprotein receptor. Immunoglobulin A proteins bound by HepG2 cells were endocytosed and catabolized.
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              Occurrence of mesangial IgA and IgM deposits in a control necropsy population.

              R. Sinniah (1983)
              Kidney sections were obtained from 200 consecutive control necropsies of patients who died of traumatic injuries, with no clinical history of renal disease or other organic disease discovered at necropsy. Mesangial IgA as the predominant immunoglobulin was found in 8/200 (4%) cases, with accompanying IgM in two of them; and IgM alone in two (1%) subjects. Deposits of C3 alone in blood vessels was observed in nine (4.5%) cases. The glomerular morphology was essentially normal or minor change only, with one case showing diffuse mesangial hypercellularity. The high incidence of mesangial IgA deposits in the local apparently healthy population may reflect some common feature of the antigen(s) or complex involved. They may be of environmental, dietary or infectious origin. It is possible that many of these "spontaneous" deposits in the glomerular mesangium may result from the clearance of circulating non-nephritogenic immune complexes.
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                Author and article information

                Contributors
                Journal
                World J Clin Cases
                WJCC
                World Journal of Clinical Cases
                Baishideng Publishing Group Inc
                2307-8960
                6 September 2020
                6 September 2020
                : 8
                : 17
                : 3828-3834
                Affiliations
                Department of Internal Medicine, Pusan National University School of Medicine, Busan 49241, South Korea
                Department of Internal Medicine, Pusan National University School of Medicine, Busan 49241, South Korea
                Department of Pathology, Pusan National University Hospital, Busan 49241, South Korea
                Division of Gastroenterology, Department of Internal Medicine, Biomedical Research Institute, Pusan National University Hospital, Busan 49241, South Korea
                Division of Nephrology, Department of Internal Medicine, Biomedical Research Institute, Pusan National University Hospital, Busan 49241, South Korea
                Division of Nephrology, Department of Internal Medicine, Biomedical Research Institute, Pusan National University Hospital, Busan 49241, South Korea
                Division of Nephrology, Department of Internal Medicine, Pusan National University Yangsan Hospital, Yangsan 626-770, South Korea
                Division of Nephrology, Department of Internal Medicine, Pusan National University Yangsan Hospital, Yangsan 626-770, South Korea
                Division of Nephrology, Department of Internal Medicine, Biomedical Research Institute, Pusan National University Hospital, Busan 49241, South Korea
                Division of Nephrology, Department of Internal Medicine, Pusan National University Yangsan Hospital, Yangsan 626-770, South Korea
                Division of Nephrology, Department of Internal Medicine, Biomedical Research Institute, Pusan National University Hospital, Busan 49241, South Korea. sey-0220@ 123456hanmail.net
                Author notes

                Author contributions: Jeon YH collected clinical data and contributed manuscript drafting; Kim DW collected clinical data and drafted table and figures; Lee SJ performed the histological analyses and interpretation; Park YJ was physician who provided treatment and intellectual content; Kim HJ, Han M, Kim IY and Lee DW reviewed the manuscript and provided intellectual content; Song SH, Lee SB and Seong EY reviewed the literature and revised the manuscript; all authors approved the final version of the manuscript.

                Supported by Pusan National University Hospital Education and Research Team, No 219.

                Corresponding author: Eun Young Seong, MD, PhD, Associate Professor, Division of Nephrology, Department of Internal Medicine, Biomedical Research Institute, Pusan National University Hospital, 179 Gudeok-ro, Seo-gu, Busan 49241, South Korea. sey-0220@ 123456hanmail.net

                Article
                jWJG.v8.i17.pg3828
                10.12998/wjcc.v8.i17.3828
                7479551
                b2c9b25e-d50a-47dd-a57f-64df5c24c913
                ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.

                This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial.

                History
                : 29 June 2020
                : 5 August 2020
                : 20 August 2020
                Categories
                Case Report

                immunoglobulin a nephropathy,secondary immunoglobulin a nephropathy,autoimmune hepatitis,liver transplantation,case report

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