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A Case of Cholestatic Autoimmune Hepatitis and Acute Liver Failure

Source: jkms.kams.or.kr
Topic: Autoimmune hepatitis
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Sort Desciption: an Unusual Hepatic Manifestation of Mixed Connective Tissue Disease and Sjogrens Syndrome. Autoimmune hepatitis (AIH) is characterized by chronic necro-inflammation of ...

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A Case of Cholestatic Autoimmune Hepatitis and Acute Liver Failure: an Unusual Hepatic Manifestation of Mixed Connective Tissue Disease and Sjogrens Syndrome INTRODUCTION Autoimmune hepatitis (AIH) is characterized by chronic necro-inflammation of the liver of unknown cause, in which immune reactions against host antigens are found to be the major pathologic mechanism. Autoimmune hepatitis can be associated with a number of other autoimmune diseases (1). Liver involvement, except hepatomegaly, is rare in patients with mixed connective tissue disease (MCTD) or Sjogren s syndrome (SS). Only two cases of AIH associated with MCTD and SS have been reported in the literature (2, 3). The clinical features of autoimmune hepatitis vary from asymptomatic, in which the disease is discovered when abnormal serum enzyme levels are detected during a screening examination, to a severe, acute, and even fulminant hepatitis (4). Cholestatic liver dysfunction is a rare clinical manifestation of AIH. Unlike primary biliary cirrhosis and sclerosing cholangitis, AIH responds favorably to steroid therapy (5). We describe a patient with MCTD and SS presenting with severe cholestatic AIH and acute liver failure, whose liver function abnormalities resolved after steroid therapy. CASE REPORT A 40-yr-old woman was admitted to the hospital because of progressive jaundice over two months. Two years before admission, the patient was diagnosed to have an overlap syndrome of MCTD and SS. The diagnosis of MCTD was made by the presence of high titer of anti-ribonucleoprotein antibodies, edema of both hands, synovitis, Raynaud s phenomenon, and acrosclerosis. Sjogren s syndrome was diagnosed by the presence of dry mouth, dry eyes, rheumatoid factor, anti-Ro antibodies, positive results of a Schirmer s test, salivary scintigraphy, and labial salivary gland biopsy. She had been intermittently treated with prednisolone (10 mg/day), but had not taken any medications during the previous three months. ...

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