Cholangiopathy aggravation is caused by VDR ablation and alleviated by VDR-independent vitamin D signaling in ABCB4 knockout mice.


Journal

Biochimica et biophysica acta. Molecular basis of disease
ISSN: 1879-260X
Titre abrégé: Biochim Biophys Acta Mol Basis Dis
Pays: Netherlands
ID NLM: 101731730

Informations de publication

Date de publication:
01 04 2021
Historique:
received: 30 09 2020
revised: 19 12 2020
accepted: 29 12 2020
pubmed: 9 1 2021
medline: 3 7 2021
entrez: 8 1 2021
Statut: ppublish

Résumé

Cholangiopathies are chronic liver diseases in which damaged cholangiocytes trigger a proinflammatory and profibrotic reaction. The nuclear vitamin D receptor (VDR) is highly expressed in cholangiocytes and exerts immune-regulatory functions in these cells. In the present study, we examined the protective function of VDR and other vitamin D signaling pathways in chronic cholangiopathy and cholangiocytes. Vdr was invalidated in Abcb4 knockout mice, a widely used animal model of chronic cholangiopathy. The impact of vitamin D signaling on cholangiopathy features was examined in vivo and in cholangiocytes (primary and cell lines). Cholangiopathy features (i.e, cholestasis, ductular reaction and fibrosis) were aggravated in Vdr;Abcb4 double knockout mice compared to the Abcb4 simple knockout, and associated with an overexpression of proinflammatory factors. The proinflammatory phenotype of cholangiocytes was also exacerbated following VDR silencing in vitro. The expression of proinflammatory factors and the severity of cholangiopathy were reduced in the double knockout mice treated with the vitamin D analog calcipotriol or with vitamin D. In vitro, the inflammatory response to TNFα was significantly reduced by calcipotriol in biliary cells silenced for VDR, and this effect was abolished by co-silencing the plasma membrane receptor of vitamin D, protein disulfide-isomerase A3 (PDIA3). Our results demonstrate an anti-inflammatory role of VDR signaling in cholangiocytes and cholangiopathy. They also provide evidence for PDIA3-mediated anti-inflammatory effects of vitamin D and vitamin D analog in these settings.

Sections du résumé

BACKGROUND & AIMS
Cholangiopathies are chronic liver diseases in which damaged cholangiocytes trigger a proinflammatory and profibrotic reaction. The nuclear vitamin D receptor (VDR) is highly expressed in cholangiocytes and exerts immune-regulatory functions in these cells. In the present study, we examined the protective function of VDR and other vitamin D signaling pathways in chronic cholangiopathy and cholangiocytes.
METHODS
Vdr was invalidated in Abcb4 knockout mice, a widely used animal model of chronic cholangiopathy. The impact of vitamin D signaling on cholangiopathy features was examined in vivo and in cholangiocytes (primary and cell lines).
RESULTS
Cholangiopathy features (i.e, cholestasis, ductular reaction and fibrosis) were aggravated in Vdr;Abcb4 double knockout mice compared to the Abcb4 simple knockout, and associated with an overexpression of proinflammatory factors. The proinflammatory phenotype of cholangiocytes was also exacerbated following VDR silencing in vitro. The expression of proinflammatory factors and the severity of cholangiopathy were reduced in the double knockout mice treated with the vitamin D analog calcipotriol or with vitamin D. In vitro, the inflammatory response to TNFα was significantly reduced by calcipotriol in biliary cells silenced for VDR, and this effect was abolished by co-silencing the plasma membrane receptor of vitamin D, protein disulfide-isomerase A3 (PDIA3).
CONCLUSIONS
Our results demonstrate an anti-inflammatory role of VDR signaling in cholangiocytes and cholangiopathy. They also provide evidence for PDIA3-mediated anti-inflammatory effects of vitamin D and vitamin D analog in these settings.

Identifiants

pubmed: 33418034
pii: S0925-4439(20)30415-4
doi: 10.1016/j.bbadis.2020.166067
pii:
doi:

Substances chimiques

ATP Binding Cassette Transporter, Subfamily B 0
Receptors, Calcitriol 0
Vdr protein, mouse 0
Vitamins 0
Vitamin D 1406-16-2

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

166067

Informations de copyright

Copyright © 2021 Elsevier B.V. All rights reserved.

Auteurs

Ester Gonzalez-Sanchez (E)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France; Inovarion, Paris, France; Oncology Program, CIBEREHD, National Biomedical Research Institute on Liver and Gastrointestinal Diseases, Instituto de Salud Carlos III, Madrid, Spain; TGF-β and Cancer Group, Oncobell Program, Bellvitge Biomedical Research Institute (IDIBELL), Barcelona, Spain. Electronic address: m.gonzalezsanchez@idibell.cat.

Haquima El Mourabit (H)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France. Electronic address: haquima.el-mourabit@inserm.fr.

Marion Jager (M)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France.

Marie Clavel (M)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France; Inovarion, Paris, France.

Sophie Moog (S)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France; Inovarion, Paris, France. Electronic address: sophie.moog@inovarion.com.

Javier Vaquero (J)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France; Oncology Program, CIBEREHD, National Biomedical Research Institute on Liver and Gastrointestinal Diseases, Instituto de Salud Carlos III, Madrid, Spain; TGF-β and Cancer Group, Oncobell Program, Bellvitge Biomedical Research Institute (IDIBELL), Barcelona, Spain; LPP (Laboratoire de Physique des Plasmas, UMR 7648), Sorbonne Université, Centre National de la Recherche Scientifique (CNRS), Ecole Polytechnique, 75005 Paris, France. Electronic address: jvaquero@idibell.cat.

Tatiana Ledent (T)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France. Electronic address: tatiana.ledent@inserm.fr.

Axelle Cadoret (A)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France. Electronic address: axelle.cadoret@inserm.fr.

Jérémie Gautheron (J)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France. Electronic address: jeremie.gautheron@inserm.fr.

Laura Fouassier (L)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France. Electronic address: laura.fouassier@inserm.fr.

Dominique Wendum (D)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France; Assistance Publique-Hôpitaux de Paris (AP-HP) Sorbonne Université, Hôpital St Antoine, Paris, France. Electronic address: dominique.wendum@aphp.fr.

Nicolas Chignard (N)

Inovarion, Paris, France. Electronic address: nicolas.chignard@inovarion.com.

Chantal Housset (C)

Sorbonne Université, INSERM, Centre de Recherche Saint-Antoine (CRSA), Paris, France; AP-HP, Reference Center for Inflammatory Biliary Diseases and Autoimmune Hepatitis (CRMR, MIVB-H), Department of Hepatology, Saint-Antoine Hospital, Paris, France. Electronic address: chantal.housset@inserm.fr.

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Classifications MeSH