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Successful Treatment of a Korean Infant with Giant Cell Hepatitis with Autoimmune Hemolytic Anemia Using Rituximab

김영호, 김주휘, 이은주, 강경훈, 강형진,
소속 상세정보
김영호 ( Kim Young-Ho ) - Seoul National University Hospital Department of Pediatrics
김주휘 ( Kim Ju-Whi ) - Seoul National University Hospital Department of Pediatrics
이은주 ( Lee Eun-Joo ) - Seoul National University Hospital Department of Pediatrics
강경훈 ( Kang Gyeong-Hoon ) - Seoul National University Hospital Department of Pathology
강형진 ( Kang Hyoung-Jin ) - Seoul National University Children’s Hospital Department of Pediatrics

Abstract


Giant cell hepatitis with autoimmune hemolytic anemia (AHA) is a rare disease of infancy characterized by the presence of both Coombs-positive hemolytic anemia and progressive liver disease with giant cell transformation of hepatocytes. Here, we report a case involving a seven-month-old male infant who presented with AHA followed by cholestatic hepatitis. The clinical features included jaundice, pallor, and red urine. Physical examination showed generalized icterus and splenomegaly. The laboratory findings suggested warm-type AHA with cholestatic hepatitis. Liver biopsy revealed giant cell transformation of hepatocytes and moderate lobular inflammation. The patient was successfully treated with four doses of rituximab. Early relapse of hemolytic anemia and hepatitis was observed, which prompted the use of an additional salvage dose of rituximab. He is currently in clinical remission.

키워드

Hepatitis; Autoimmune hemolytic anemia; Rituximab

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