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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...
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition
2020
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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. |
format | Online Article Text |
id | pubmed-7073370 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition |
record_format | MEDLINE/PubMed |
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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