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

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...

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Autores principales: Kim, Young Ho, Kim, Ju Whi, Lee, Eun Joo, Kang, Gyeong Hoon, Kang, Hyoung Jin, Moon, Jin Soo, Ko, Jae Sung
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7073370/
https://www.ncbi.nlm.nih.gov/pubmed/32206631
http://dx.doi.org/10.5223/pghn.2020.23.2.180
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author Kim, Young Ho
Kim, Ju Whi
Lee, Eun Joo
Kang, Gyeong Hoon
Kang, Hyoung Jin
Moon, Jin Soo
Ko, Jae Sung
author_facet Kim, Young Ho
Kim, Ju Whi
Lee, Eun Joo
Kang, Gyeong Hoon
Kang, Hyoung Jin
Moon, Jin Soo
Ko, Jae Sung
author_sort Kim, Young Ho
collection PubMed
description 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.
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spelling pubmed-70733702020-03-23 Successful Treatment of a Korean Infant with Giant Cell Hepatitis with Autoimmune Hemolytic Anemia Using Rituximab Kim, Young Ho Kim, Ju Whi Lee, Eun Joo Kang, Gyeong Hoon Kang, Hyoung Jin Moon, Jin Soo Ko, Jae Sung Pediatr Gastroenterol Hepatol Nutr Case Report 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. The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition 2020-03 2020-03-04 /pmc/articles/PMC7073370/ /pubmed/32206631 http://dx.doi.org/10.5223/pghn.2020.23.2.180 Text en Copyright © 2020 by The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition https://creativecommons.org/licenses/by-nc/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (https://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Kim, Young Ho
Kim, Ju Whi
Lee, Eun Joo
Kang, Gyeong Hoon
Kang, Hyoung Jin
Moon, Jin Soo
Ko, Jae Sung
Successful Treatment of a Korean Infant with Giant Cell Hepatitis with Autoimmune Hemolytic Anemia Using Rituximab
title Successful Treatment of a Korean Infant with Giant Cell Hepatitis with Autoimmune Hemolytic Anemia Using Rituximab
title_full Successful Treatment of a Korean Infant with Giant Cell Hepatitis with Autoimmune Hemolytic Anemia Using Rituximab
title_fullStr Successful Treatment of a Korean Infant with Giant Cell Hepatitis with Autoimmune Hemolytic Anemia Using Rituximab
title_full_unstemmed Successful Treatment of a Korean Infant with Giant Cell Hepatitis with Autoimmune Hemolytic Anemia Using Rituximab
title_short Successful Treatment of a Korean Infant with Giant Cell Hepatitis with Autoimmune Hemolytic Anemia Using Rituximab
title_sort successful treatment of a korean infant with giant cell hepatitis with autoimmune hemolytic anemia using rituximab
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7073370/
https://www.ncbi.nlm.nih.gov/pubmed/32206631
http://dx.doi.org/10.5223/pghn.2020.23.2.180
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