Retrospective Study
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Hepatol. Dec 27, 2021; 13(12): 2113-2127
Published online Dec 27, 2021. doi: 10.4254/wjh.v13.i12.2113
Use of oral vancomycin in children with autoimmune liver disease: A single centre experience
Angelo Di Giorgio, Anna Tulone, Emanuele Nicastro, Lorenzo Norsa, Aurelio Sonzogni, Lorenzo D'Antiga
Angelo Di Giorgio, Anna Tulone, Emanuele Nicastro, Lorenzo Norsa, Lorenzo D'Antiga, Pediatric Hepatology, Gastroenterology and Transplantation, Hospital Papa Giovanni XXIII , Bergamo 24127, Italy
Aurelio Sonzogni, Liver Pathology, Hospital Papa Giovanni XXIII, Bergamo 24127, Italy
Author contributions: Di Giorgio A, Tulone A drafted the paper; Di Giorgio A wrote the paper; Tulone A collected data; Nicastro E and Sonzogni A contributed to the conception analysis; Nicastro E, Norsa L, Sonzogni A, and D’Antiga L contributed to the interpretation of data; Norsa L performed the research; D’Antiga L supervised the study; and all authors approved the submission of this version of the manuscript and takes full responsibility for the manuscript contents.
Institutional review board statement: At our centre (tertiary referral centre for liver transplantation), no approval by local ethical committee is required for retrospective anonymised study which includes only patients from our centre.
Informed consent statement: The informed consent statement was waived.
Conflict-of-interest statement: There are no conflicts of interest to declare.
Data sharing statement: No additional data are available.
Open-Access: 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/
Corresponding author: Angelo Di Giorgio, MD, PhD, Pediatric Hepatology, Gastroenterology and Transplantation, Hospital Papa Giovanni XXIII , Piazza Oms 1, Bergamo 24127, Italy. adigiorgio@asst-pg23.it
Received: May 27, 2021
Peer-review started: May 27, 2021
First decision: July 6, 2021
Revised: July 7, 2021
Accepted: November 25, 2021
Article in press: November 25, 2021
Published online: December 27, 2021
Processing time: 213 Days and 16.9 Hours
Abstract
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.

Keywords: Autoimmune hepatitis; Autoimmune sclerosing cholangitis; Autoimmune liver disease; Vancomycin; Children; Liver transplantation

Core Tip: Experience with oral vancomycin in children with autoimmune liver disease (AILD) is limited. We enrolled 75 children [median age 10.5 years (5.6-13.4)], 54 with autoimmune hepatitis and 21 with autoimmune sclerosing cholangitis; 63/75 achieved remission by standard immunosuppressive therapy (IS), whereas 12/75 (16%) required oral vancomycin treatment (OVT). In 6/12 patients (50%) the response was complete, whereas it was partial in 2/12 (17%), and absent in 4/12 (33%). Overall OVT increased the remission rate of the whole group of AILD patients from 81% to 92%. OVT may represent a valuable treatment option in children with AILD who do not respond to standard IS.