Low-dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring: Evidence for a spectrum of neonatal injury.

Biliary atresia is a neonatal disease characterized by damage, inflammation, and fibrosis of the liver and bile ducts and by abnormal bile metabolism. It likely results from a prenatal environmental exposure that spares the mother and affects the fetus. Our aim was to develop a model of fetal injury...

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Main Authors: Kapish Gupta, Jimmy P Xu, Tamir Diamond, Iris E M de Jong, Andrew Glass, Jessica Llewellyn, Neil D Theise, Orith Waisbourd-Zinman, Jeffrey D Winkler, Edward M Behrens, Clementina Mesaros, Rebecca G Wells
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2024-01-01
Series:PLoS ONE
Online Access:https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0301824&type=printable
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author Kapish Gupta
Jimmy P Xu
Tamir Diamond
Iris E M de Jong
Andrew Glass
Jessica Llewellyn
Neil D Theise
Orith Waisbourd-Zinman
Jeffrey D Winkler
Edward M Behrens
Clementina Mesaros
Rebecca G Wells
author_facet Kapish Gupta
Jimmy P Xu
Tamir Diamond
Iris E M de Jong
Andrew Glass
Jessica Llewellyn
Neil D Theise
Orith Waisbourd-Zinman
Jeffrey D Winkler
Edward M Behrens
Clementina Mesaros
Rebecca G Wells
author_sort Kapish Gupta
collection DOAJ
description Biliary atresia is a neonatal disease characterized by damage, inflammation, and fibrosis of the liver and bile ducts and by abnormal bile metabolism. It likely results from a prenatal environmental exposure that spares the mother and affects the fetus. Our aim was to develop a model of fetal injury by exposing pregnant mice to low-dose biliatresone, a plant toxin implicated in biliary atresia in livestock, and then to determine whether there was a hepatobiliary phenotype in their pups. Pregnant mice were treated orally with 15 mg/kg/d biliatresone for 2 days. Histology of the liver and bile ducts, serum bile acids, and liver immune cells of pups from treated mothers were analyzed at P5 and P21. Pups had no evidence of histological liver or bile duct injury or fibrosis at either timepoint. In addition, growth was normal. However, serum levels of glycocholic acid were elevated at P5, suggesting altered bile metabolism, and the serum bile acid profile became increasingly abnormal through P21, with enhanced glycine conjugation of bile acids. There was also immune cell activation observed in the liver at P21. These results suggest that prenatal exposure to low doses of an environmental toxin can cause subclinical disease including liver inflammation and aberrant bile metabolism even in the absence of histological changes. This finding suggests a wide potential spectrum of disease after fetal biliary injury.
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spelling doaj-art-74507efcbeca4a1daf62e83fff6e33fb2025-08-20T04:00:43ZengPublic Library of Science (PLoS)PLoS ONE1932-62032024-01-01194e030182410.1371/journal.pone.0301824Low-dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring: Evidence for a spectrum of neonatal injury.Kapish GuptaJimmy P XuTamir DiamondIris E M de JongAndrew GlassJessica LlewellynNeil D TheiseOrith Waisbourd-ZinmanJeffrey D WinklerEdward M BehrensClementina MesarosRebecca G WellsBiliary atresia is a neonatal disease characterized by damage, inflammation, and fibrosis of the liver and bile ducts and by abnormal bile metabolism. It likely results from a prenatal environmental exposure that spares the mother and affects the fetus. Our aim was to develop a model of fetal injury by exposing pregnant mice to low-dose biliatresone, a plant toxin implicated in biliary atresia in livestock, and then to determine whether there was a hepatobiliary phenotype in their pups. Pregnant mice were treated orally with 15 mg/kg/d biliatresone for 2 days. Histology of the liver and bile ducts, serum bile acids, and liver immune cells of pups from treated mothers were analyzed at P5 and P21. Pups had no evidence of histological liver or bile duct injury or fibrosis at either timepoint. In addition, growth was normal. However, serum levels of glycocholic acid were elevated at P5, suggesting altered bile metabolism, and the serum bile acid profile became increasingly abnormal through P21, with enhanced glycine conjugation of bile acids. There was also immune cell activation observed in the liver at P21. These results suggest that prenatal exposure to low doses of an environmental toxin can cause subclinical disease including liver inflammation and aberrant bile metabolism even in the absence of histological changes. This finding suggests a wide potential spectrum of disease after fetal biliary injury.https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0301824&type=printable
spellingShingle Kapish Gupta
Jimmy P Xu
Tamir Diamond
Iris E M de Jong
Andrew Glass
Jessica Llewellyn
Neil D Theise
Orith Waisbourd-Zinman
Jeffrey D Winkler
Edward M Behrens
Clementina Mesaros
Rebecca G Wells
Low-dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring: Evidence for a spectrum of neonatal injury.
PLoS ONE
title Low-dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring: Evidence for a spectrum of neonatal injury.
title_full Low-dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring: Evidence for a spectrum of neonatal injury.
title_fullStr Low-dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring: Evidence for a spectrum of neonatal injury.
title_full_unstemmed Low-dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring: Evidence for a spectrum of neonatal injury.
title_short Low-dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring: Evidence for a spectrum of neonatal injury.
title_sort low dose biliatresone treatment of pregnant mice causes subclinical biliary disease in their offspring evidence for a spectrum of neonatal injury
url https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0301824&type=printable
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