Case Report: A Case of Epstein-Barr Virus-Associated Acute Liver Failure Requiring Hematopoietic Cell Transplantation After Emergent Liver Transplantation

Hepatic manifestations of Epstein-Barr virus (EBV) infection are relatively common, mild, and self-limiting. Although fulminant hepatic failure has been reported in a few cases, the contributing factors are unclear. This report discusses a pediatric case of EBV-associated acute liver failure that re...

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Bibliographic Details
Published in:Frontiers in immunology 2022-01, Vol.13, p.825806
Main Authors: Nakajima, Koji, Hiejima, Eitaro, Nihira, Hiroshi, Kato, Kentaro, Honda, Yoshitaka, Izawa, Kazushi, Kawabata, Naoko, Kato, Itaru, Ogawa, Eri, Sonoda, Mari, Okamoto, Tatsuya, Okajima, Hideaki, Yasumi, Takahiro, Takita, Junko
Format: Article
Language:English
Subjects:
acute liver failure (ALF)
CAEBV infection
case report
CD8-Positive T-Lymphocytes - virology
Child, Preschool
Epstein-Barr virus (EBV)
Epstein-Barr Virus Infections - immunology
Epstein-Barr Virus Infections - virology
hematopoietic cell transplantation (HCT)
Hematopoietic Stem Cell Transplantation
hemophagocytic lymphohistiocytosis (HLH)
Herpesvirus 4, Human - genetics
Humans
Immunology
Liver - diagnostic imaging
Liver - pathology
Liver Failure, Acute - therapy
Liver Failure, Acute - virology
Liver Transplantation
Lymphohistiocytosis, Hemophagocytic - therapy
Lymphohistiocytosis, Hemophagocytic - virology
Male
Positron Emission Tomography Computed Tomography
RNA, Viral - analysis
Treatment Outcome
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Summary:Hepatic manifestations of Epstein-Barr virus (EBV) infection are relatively common, mild, and self-limiting. Although fulminant hepatic failure has been reported in a few cases, the contributing factors are unclear. This report discusses a pediatric case of EBV-associated acute liver failure that required urgent liver transplantation; however, liver damage continued to progress post-liver replacement. Monoclonal CD8+ T cells that preferentially infiltrated the native and transplanted liver were positive for EBV-encoded small RNA, suggesting a pathophysiology similar to that of EBV-associated hemophagocytic lymphohistiocytosis and chronic active EBV infection. Therefore, subsequent chemotherapy and hematopoietic cell transplantation was conducted, which led to cure. This is the first case of EBV-associated acute liver failure that relapsed post-liver transplant. As such, it sheds light on an under-recognized clinical entity: liver-restricted hyperinflammation caused by EBV-infected monoclonal CD8+ T cells. This phenomenon needs to be recognized and differentiated from hepatitis/hepatic failure caused by EBV-infected B cells, which has a relatively benign clinical course.
ISSN:1664-3224
1664-3224
DOI:10.3389/fimmu.2022.825806