Coordinated development of the mouse extrahepatic bile duct: Implications for neonatal susceptibility to biliary injury.


Journal

Journal of hepatology
ISSN: 1600-0641
Titre abrégé: J Hepatol
Pays: Netherlands
ID NLM: 8503886

Informations de publication

Date de publication:
01 2020
Historique:
received: 13 03 2019
revised: 09 08 2019
accepted: 30 08 2019
pubmed: 29 9 2019
medline: 2 6 2021
entrez: 29 9 2019
Statut: ppublish

Résumé

The extrahepatic bile duct is the primary tissue initially affected by biliary atresia. Biliary atresia is a cholangiopathy which exclusively affects neonates. Current animal models suggest that the developing bile duct is uniquely susceptible to damage. In this study, we aimed to define the anatomical and functional differences between the neonatal and adult mouse extrahepatic bile ducts. We studied mouse passaged cholangiocytes, mouse BALB/c neonatal and adult primary cholangiocytes, as well as isolated extrahepatic bile ducts, and a collagen reporter mouse. The methods used included transmission electron microscopy, lectin staining, immunostaining, rhodamine uptake assays, bile acid toxicity assays, and in vitro modeling of the matrix. The cholangiocyte monolayer of the neonatal extrahepatic bile duct was immature, lacking the uniform apical glycocalyx and mature cell-cell junctions typical of adult cholangiocytes. Functional studies showed that the glycocalyx protected against bile acid injury and that neonatal cholangiocyte monolayers were more permeable than adult monolayers. In adult ducts, the submucosal space was filled with collagen I, elastin, hyaluronic acid, and proteoglycans. In contrast, the neonatal submucosa had little collagen I and elastin, although both increased rapidly after birth. In vitro modeling of the matrix suggested that the composition of the neonatal submucosa relative to the adult submucosa led to increased diffusion of bile. A Col-GFP reporter mouse showed that cells in the neonatal but not adult submucosa were actively producing collagen. We identified 4 key differences between the neonatal and adult extrahepatic bile duct. We showed that these features may have functional implications, suggesting the neonatal extrahepatic bile ducts are particularly susceptible to injury and fibrosis. Biliary atresia is a disease that affects newborns and is characterized by extrahepatic bile duct injury and obstruction, resulting in liver injury. We identify 4 key differences between the epithelial and submucosal layers of the neonatal and adult extrahepatic bile duct and show that these may render the neonatal duct particularly susceptible to injury.

Sections du résumé

BACKGROUND & AIMS
The extrahepatic bile duct is the primary tissue initially affected by biliary atresia. Biliary atresia is a cholangiopathy which exclusively affects neonates. Current animal models suggest that the developing bile duct is uniquely susceptible to damage. In this study, we aimed to define the anatomical and functional differences between the neonatal and adult mouse extrahepatic bile ducts.
METHODS
We studied mouse passaged cholangiocytes, mouse BALB/c neonatal and adult primary cholangiocytes, as well as isolated extrahepatic bile ducts, and a collagen reporter mouse. The methods used included transmission electron microscopy, lectin staining, immunostaining, rhodamine uptake assays, bile acid toxicity assays, and in vitro modeling of the matrix.
RESULTS
The cholangiocyte monolayer of the neonatal extrahepatic bile duct was immature, lacking the uniform apical glycocalyx and mature cell-cell junctions typical of adult cholangiocytes. Functional studies showed that the glycocalyx protected against bile acid injury and that neonatal cholangiocyte monolayers were more permeable than adult monolayers. In adult ducts, the submucosal space was filled with collagen I, elastin, hyaluronic acid, and proteoglycans. In contrast, the neonatal submucosa had little collagen I and elastin, although both increased rapidly after birth. In vitro modeling of the matrix suggested that the composition of the neonatal submucosa relative to the adult submucosa led to increased diffusion of bile. A Col-GFP reporter mouse showed that cells in the neonatal but not adult submucosa were actively producing collagen.
CONCLUSION
We identified 4 key differences between the neonatal and adult extrahepatic bile duct. We showed that these features may have functional implications, suggesting the neonatal extrahepatic bile ducts are particularly susceptible to injury and fibrosis.
LAY SUMMARY
Biliary atresia is a disease that affects newborns and is characterized by extrahepatic bile duct injury and obstruction, resulting in liver injury. We identify 4 key differences between the epithelial and submucosal layers of the neonatal and adult extrahepatic bile duct and show that these may render the neonatal duct particularly susceptible to injury.

Identifiants

pubmed: 31562906
pii: S0168-8278(19)30550-1
doi: 10.1016/j.jhep.2019.08.036
pmc: PMC7079197
mid: NIHMS1544628
pii:
doi:

Substances chimiques

Collagen Type I 0
Collagen Type I, alpha 1 Chain 0
Proteoglycans 0
Green Fluorescent Proteins 147336-22-9
Hyaluronic Acid 9004-61-9
Elastin 9007-58-3

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, Non-P.H.S.

Langues

eng

Sous-ensembles de citation

IM

Pagination

135-145

Subventions

Organisme : NIDDK NIH HHS
ID : R01 DK092111
Pays : United States
Organisme : NIDDK NIH HHS
ID : P30 DK050306
Pays : United States
Organisme : NIDDK NIH HHS
ID : R01 DK111866
Pays : United States
Organisme : NIDDK NIH HHS
ID : R01 DK119290
Pays : United States
Organisme : NIDDK NIH HHS
ID : T32 DK101371
Pays : United States
Organisme : NCATS NIH HHS
ID : TL1 TR001880
Pays : United States
Organisme : NIAAA NIH HHS
ID : U01 AA022614
Pays : United States
Organisme : NIH HHS
ID : S10 OD021633
Pays : United States

Informations de copyright

Copyright © 2019 European Association for the Study of the Liver. Published by Elsevier B.V. All rights reserved.

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Auteurs

Gauri Khandekar (G)

Division of Gastroenterology, Department of Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, United States.

Jessica Llewellyn (J)

Division of Gastroenterology, Department of Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, United States.

Alyssa Kriegermeier (A)

Division of Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, The Children's Hospital of Philadelphia, Philadelphia, PA, United States.

Orith Waisbourd-Zinman (O)

Division of Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, The Children's Hospital of Philadelphia, Philadelphia, PA, United States; Schneider Children's Hospital, Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.

Nicolette Johnson (N)

Cell and Molecular Biology Graduate Group, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, United States.

Yu Du (Y)

Division of Gastroenterology, Department of Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, United States.

Roquibat Giwa (R)

Division of Gastroenterology, Department of Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, United States.

Xiao Liu (X)

Department of Surgery University of California, San Diego, La Jolla, CA, United States.

Tatiana Kisseleva (T)

Department of Surgery University of California, San Diego, La Jolla, CA, United States.

Pierre A Russo (PA)

Department of Pathology and Laboratory Medicine, The Children's Hospital of Philadelphia, Philadelphia, PA, United States.

Neil D Theise (ND)

Department of Pathology, New York University School of Medicine, New York, NY, United States.

Rebecca G Wells (RG)

Division of Gastroenterology, Department of Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, United States; Department of Bioengineering, School of Engineering and Applied Sciences, The University of Pennsylvania, Philadelphia, PA, United States; Department of Pathology and Laboratory Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, United States; Center for Engineering MechanoBiology, The University of Pennsylvania, Philadelphia, PA, United States. Electronic address: rgwells@pennmedicine.upenn.edu.

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Classifications MeSH