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Novel Diagnostic and Therapeutic Strategies in Juvenile Autoimmune Hepatitis

Juvenile autoimmune hepatitis (JAIH) is a rare, chronic, inflammatory disease of the liver characterized by a complex interaction between genetic, immunological, and environmental factors leading to loss of immunotolerance to hepatic antigens. It affects both children and adolescents, most commonly...

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Autores principales: Sciveres, Marco, Nastasio, Silvia, Maggiore, Giuseppe
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6763601/
https://www.ncbi.nlm.nih.gov/pubmed/31616649
http://dx.doi.org/10.3389/fped.2019.00382
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author Sciveres, Marco
Nastasio, Silvia
Maggiore, Giuseppe
author_facet Sciveres, Marco
Nastasio, Silvia
Maggiore, Giuseppe
author_sort Sciveres, Marco
collection PubMed
description Juvenile autoimmune hepatitis (JAIH) is a rare, chronic, inflammatory disease of the liver characterized by a complex interaction between genetic, immunological, and environmental factors leading to loss of immunotolerance to hepatic antigens. It affects both children and adolescents, most commonly females, and its clinical manifestations are quite variable. JAIH is progressive in nature and if left untreated may lead to cirrhosis and terminal liver failure. Although JAIH was first described almost 50 years ago, there have been few significant advances in the clinical management of these patients, both in terms of available diagnostic tools and therapeutic options. Aminotransferase activity, class G immunoglobulins and autoantibodies are the biomarkers used to diagnose AIH and monitor treatment response alongside clinical and histological findings. Despite their utility and cost-effectiveness, these biomarkers are neither an accurate expression of AIH pathogenic mechanism nor a precise measure of treatment response. Current standard of care is mainly based on the administration of steroids and azathioprine. This combination of drugs has been proven effective in inducing remission of disease in the majority of patients dramatically improving their survival; however, it not only fails to restore tolerance to hepatic autoantigens, but it also does not halt disease progression in some patients, it is often needed life-long and finally, it has deleterious side-effects. The ideal therapy should be enough selective to contrast immune-mediated live damage while preserving or potentiating the ability to develop permanent tolerance vs. pathogenic autoantigens. By reviewing the state of the art literature, this article highlights novel diagnostic and therapeutic strategies for managing pediatric AIH with a special focus on new strategies of immunotherapy. These promising tools could improve the diagnostic algorithm, more accurately predict disease prognosis, and provide targeted, individualized treatment.
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spelling pubmed-67636012019-10-15 Novel Diagnostic and Therapeutic Strategies in Juvenile Autoimmune Hepatitis Sciveres, Marco Nastasio, Silvia Maggiore, Giuseppe Front Pediatr Pediatrics Juvenile autoimmune hepatitis (JAIH) is a rare, chronic, inflammatory disease of the liver characterized by a complex interaction between genetic, immunological, and environmental factors leading to loss of immunotolerance to hepatic antigens. It affects both children and adolescents, most commonly females, and its clinical manifestations are quite variable. JAIH is progressive in nature and if left untreated may lead to cirrhosis and terminal liver failure. Although JAIH was first described almost 50 years ago, there have been few significant advances in the clinical management of these patients, both in terms of available diagnostic tools and therapeutic options. Aminotransferase activity, class G immunoglobulins and autoantibodies are the biomarkers used to diagnose AIH and monitor treatment response alongside clinical and histological findings. Despite their utility and cost-effectiveness, these biomarkers are neither an accurate expression of AIH pathogenic mechanism nor a precise measure of treatment response. Current standard of care is mainly based on the administration of steroids and azathioprine. This combination of drugs has been proven effective in inducing remission of disease in the majority of patients dramatically improving their survival; however, it not only fails to restore tolerance to hepatic autoantigens, but it also does not halt disease progression in some patients, it is often needed life-long and finally, it has deleterious side-effects. The ideal therapy should be enough selective to contrast immune-mediated live damage while preserving or potentiating the ability to develop permanent tolerance vs. pathogenic autoantigens. By reviewing the state of the art literature, this article highlights novel diagnostic and therapeutic strategies for managing pediatric AIH with a special focus on new strategies of immunotherapy. These promising tools could improve the diagnostic algorithm, more accurately predict disease prognosis, and provide targeted, individualized treatment. Frontiers Media S.A. 2019-09-20 /pmc/articles/PMC6763601/ /pubmed/31616649 http://dx.doi.org/10.3389/fped.2019.00382 Text en Copyright © 2019 Sciveres, Nastasio and Maggiore. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Pediatrics
Sciveres, Marco
Nastasio, Silvia
Maggiore, Giuseppe
Novel Diagnostic and Therapeutic Strategies in Juvenile Autoimmune Hepatitis
title Novel Diagnostic and Therapeutic Strategies in Juvenile Autoimmune Hepatitis
title_full Novel Diagnostic and Therapeutic Strategies in Juvenile Autoimmune Hepatitis
title_fullStr Novel Diagnostic and Therapeutic Strategies in Juvenile Autoimmune Hepatitis
title_full_unstemmed Novel Diagnostic and Therapeutic Strategies in Juvenile Autoimmune Hepatitis
title_short Novel Diagnostic and Therapeutic Strategies in Juvenile Autoimmune Hepatitis
title_sort novel diagnostic and therapeutic strategies in juvenile autoimmune hepatitis
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6763601/
https://www.ncbi.nlm.nih.gov/pubmed/31616649
http://dx.doi.org/10.3389/fped.2019.00382
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