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Use of oral vancomycin in children with autoimmune liver disease: A single centre experience

BACKGROUND: Previous reports showed some beneficial effect of oral vancomycin treatment (OVT) in children with primary sclerosing cholangitis; conversely, the experience in patients with other autoimmune liver diseases (AILD), including autoimmune hepatitis (AIH) and autoimmune sclerosing cholangiti...

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Autores principales: Di Giorgio, Angelo, Tulone, Anna, Nicastro, Emanuele, Norsa, Lorenzo, Sonzogni, Aurelio, D'Antiga, Lorenzo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Baishideng Publishing Group Inc 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8727203/
https://www.ncbi.nlm.nih.gov/pubmed/35070012
http://dx.doi.org/10.4254/wjh.v13.i12.2113
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author Di Giorgio, Angelo
Tulone, Anna
Nicastro, Emanuele
Norsa, Lorenzo
Sonzogni, Aurelio
D'Antiga, Lorenzo
author_facet Di Giorgio, Angelo
Tulone, Anna
Nicastro, Emanuele
Norsa, Lorenzo
Sonzogni, Aurelio
D'Antiga, Lorenzo
author_sort Di Giorgio, Angelo
collection PubMed
description BACKGROUND: Previous reports showed some beneficial effect of oral vancomycin treatment (OVT) in children with primary sclerosing cholangitis; conversely, the experience in patients with other autoimmune liver diseases (AILD), including autoimmune hepatitis (AIH) and autoimmune sclerosing cholangitis (ASC), is scant. AIM: To assess the response to immunosuppressive treatment (IS) and to OVT in children diagnosed with AILD. METHODS: Retrospective study of children diagnosed with AIH (normal biliary tree at cholangiography) and ASC (abnormal biliary tree at cholangiography) in the last 10 years. All underwent standard immunosuppressive therapy (IS), but non-responders received also OVT. Biochemical remission [normal aspartate aminotransferase (AST)] and immunological remission (normal IgG and negative autoantibodies) rates and Sclerosing Cholangitis Outcomes in Pediatrics (SCOPE) index were assessed and compared during the follow up. RESULTS: 75 children were included [69% female, median age 10.5 years (5.6-13.4 years), AIH = 54, ASC= 21]. Sixty-three patients (84%, AIH = 52, ASC = 11) were treated with standard IS and 61 achieved biochemical remission, whereas 12 not responding to IS [16%, F = 75%, median age 13.5 years, (12.2-15.7), 10 with ASC] required OVT and 8 achieved biochemical remission. Overall OVT increased the biochemical remission rate of the whole group of AILD patients from 81% (61/75) to 92% (69/75). Median values of AST, alanine aminotransferase (ALT) and gamma-glutamyl transferase (GGT) decreased significantly after OVT start (P < 0.05). Complete normalization of livers enzymes (AST, ALT and GGT) was observed in 6/12 patients (50%). Decrease in SCOPE index score was reported in 5/12 patients (42%). At last follow up (median of 4.4 years, range 0.6-13.8 years) all 75 patients are alive, 6 (8%, 1 with ASC) successfully discontinued medications, 1 (with ASC) required liver transplantation. CONCLUSION: Children with AIH and ASC respond well to IS treatment. OVT may represent a valuable treatment option to achieve biochemical remission in patients not responding to standard IS. These promising preliminary results suggest that a prospective study is indicated to define the efficacy of OVT in AILD.
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spelling pubmed-87272032022-01-20 Use of oral vancomycin in children with autoimmune liver disease: A single centre experience Di Giorgio, Angelo Tulone, Anna Nicastro, Emanuele Norsa, Lorenzo Sonzogni, Aurelio D'Antiga, Lorenzo World J Hepatol Retrospective Study BACKGROUND: Previous reports showed some beneficial effect of oral vancomycin treatment (OVT) in children with primary sclerosing cholangitis; conversely, the experience in patients with other autoimmune liver diseases (AILD), including autoimmune hepatitis (AIH) and autoimmune sclerosing cholangitis (ASC), is scant. AIM: To assess the response to immunosuppressive treatment (IS) and to OVT in children diagnosed with AILD. METHODS: Retrospective study of children diagnosed with AIH (normal biliary tree at cholangiography) and ASC (abnormal biliary tree at cholangiography) in the last 10 years. All underwent standard immunosuppressive therapy (IS), but non-responders received also OVT. Biochemical remission [normal aspartate aminotransferase (AST)] and immunological remission (normal IgG and negative autoantibodies) rates and Sclerosing Cholangitis Outcomes in Pediatrics (SCOPE) index were assessed and compared during the follow up. RESULTS: 75 children were included [69% female, median age 10.5 years (5.6-13.4 years), AIH = 54, ASC= 21]. Sixty-three patients (84%, AIH = 52, ASC = 11) were treated with standard IS and 61 achieved biochemical remission, whereas 12 not responding to IS [16%, F = 75%, median age 13.5 years, (12.2-15.7), 10 with ASC] required OVT and 8 achieved biochemical remission. Overall OVT increased the biochemical remission rate of the whole group of AILD patients from 81% (61/75) to 92% (69/75). Median values of AST, alanine aminotransferase (ALT) and gamma-glutamyl transferase (GGT) decreased significantly after OVT start (P < 0.05). Complete normalization of livers enzymes (AST, ALT and GGT) was observed in 6/12 patients (50%). Decrease in SCOPE index score was reported in 5/12 patients (42%). At last follow up (median of 4.4 years, range 0.6-13.8 years) all 75 patients are alive, 6 (8%, 1 with ASC) successfully discontinued medications, 1 (with ASC) required liver transplantation. CONCLUSION: Children with AIH and ASC respond well to IS treatment. OVT may represent a valuable treatment option to achieve biochemical remission in patients not responding to standard IS. These promising preliminary results suggest that a prospective study is indicated to define the efficacy of OVT in AILD. Baishideng Publishing Group Inc 2021-12-27 2021-12-27 /pmc/articles/PMC8727203/ /pubmed/35070012 http://dx.doi.org/10.4254/wjh.v13.i12.2113 Text en ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved. https://creativecommons.org/licenses/by-nc/4.0/This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
spellingShingle Retrospective Study
Di Giorgio, Angelo
Tulone, Anna
Nicastro, Emanuele
Norsa, Lorenzo
Sonzogni, Aurelio
D'Antiga, Lorenzo
Use of oral vancomycin in children with autoimmune liver disease: A single centre experience
title Use of oral vancomycin in children with autoimmune liver disease: A single centre experience
title_full Use of oral vancomycin in children with autoimmune liver disease: A single centre experience
title_fullStr Use of oral vancomycin in children with autoimmune liver disease: A single centre experience
title_full_unstemmed Use of oral vancomycin in children with autoimmune liver disease: A single centre experience
title_short Use of oral vancomycin in children with autoimmune liver disease: A single centre experience
title_sort use of oral vancomycin in children with autoimmune liver disease: a single centre experience
topic Retrospective Study
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8727203/
https://www.ncbi.nlm.nih.gov/pubmed/35070012
http://dx.doi.org/10.4254/wjh.v13.i12.2113
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