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Loss of hepatic Flcn protects against fibrosis and inflammation by activating autophagy pathways

Non-alcoholic fatty liver disease (NAFLD) is the most frequent liver disease worldwide and can progress to non-alcoholic steatohepatitis (NASH), which is characterized by triglyceride accumulation, inflammation, and fibrosis. No pharmacological agents are currently approved to treat these conditions...

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Autores principales: Paquette, Mathieu, Yan, Ming, Ramírez-Reyes, Josué M. J., El-Houjeiri, Leeanna, Biondini, Marco, Dufour, Catherine R., Jeong, Hyeonju, Pacis, Alain, Giguère, Vincent, Estall, Jennifer L., Siegel, Peter M., Audet-Walsh, Étienne, Pause, Arnim
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8553785/
https://www.ncbi.nlm.nih.gov/pubmed/34711912
http://dx.doi.org/10.1038/s41598-021-99958-7
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author Paquette, Mathieu
Yan, Ming
Ramírez-Reyes, Josué M. J.
El-Houjeiri, Leeanna
Biondini, Marco
Dufour, Catherine R.
Jeong, Hyeonju
Pacis, Alain
Giguère, Vincent
Estall, Jennifer L.
Siegel, Peter M.
Audet-Walsh, Étienne
Pause, Arnim
author_facet Paquette, Mathieu
Yan, Ming
Ramírez-Reyes, Josué M. J.
El-Houjeiri, Leeanna
Biondini, Marco
Dufour, Catherine R.
Jeong, Hyeonju
Pacis, Alain
Giguère, Vincent
Estall, Jennifer L.
Siegel, Peter M.
Audet-Walsh, Étienne
Pause, Arnim
author_sort Paquette, Mathieu
collection PubMed
description Non-alcoholic fatty liver disease (NAFLD) is the most frequent liver disease worldwide and can progress to non-alcoholic steatohepatitis (NASH), which is characterized by triglyceride accumulation, inflammation, and fibrosis. No pharmacological agents are currently approved to treat these conditions, but it is clear now that modulation of lipid synthesis and autophagy are key biological mechanisms that could help reduce or prevent these liver diseases. The folliculin (FLCN) protein has been recently identified as a central regulatory node governing whole body energy homeostasis, and we hypothesized that FLCN regulates highly metabolic tissues like the liver. We thus generated a liver specific Flcn knockout mouse model to study its role in liver disease progression. Using the methionine- and choline-deficient diet to mimic liver fibrosis, we demonstrate that loss of Flcn reduced triglyceride accumulation, fibrosis, and inflammation in mice. In this aggressive liver disease setting, loss of Flcn led to activation of transcription factors TFEB and TFE3 to promote autophagy, promoting the degradation of intracellular lipid stores, ultimately resulting in reduced hepatocellular damage and inflammation. Hence, the activity of FLCN could be a promising target for small molecule drugs to treat liver fibrosis by specifically activating autophagy. Collectively, these results show an unexpected role for Flcn in fatty liver disease progression and highlight new potential treatment strategies.
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spelling pubmed-85537852021-11-01 Loss of hepatic Flcn protects against fibrosis and inflammation by activating autophagy pathways Paquette, Mathieu Yan, Ming Ramírez-Reyes, Josué M. J. El-Houjeiri, Leeanna Biondini, Marco Dufour, Catherine R. Jeong, Hyeonju Pacis, Alain Giguère, Vincent Estall, Jennifer L. Siegel, Peter M. Audet-Walsh, Étienne Pause, Arnim Sci Rep Article Non-alcoholic fatty liver disease (NAFLD) is the most frequent liver disease worldwide and can progress to non-alcoholic steatohepatitis (NASH), which is characterized by triglyceride accumulation, inflammation, and fibrosis. No pharmacological agents are currently approved to treat these conditions, but it is clear now that modulation of lipid synthesis and autophagy are key biological mechanisms that could help reduce or prevent these liver diseases. The folliculin (FLCN) protein has been recently identified as a central regulatory node governing whole body energy homeostasis, and we hypothesized that FLCN regulates highly metabolic tissues like the liver. We thus generated a liver specific Flcn knockout mouse model to study its role in liver disease progression. Using the methionine- and choline-deficient diet to mimic liver fibrosis, we demonstrate that loss of Flcn reduced triglyceride accumulation, fibrosis, and inflammation in mice. In this aggressive liver disease setting, loss of Flcn led to activation of transcription factors TFEB and TFE3 to promote autophagy, promoting the degradation of intracellular lipid stores, ultimately resulting in reduced hepatocellular damage and inflammation. Hence, the activity of FLCN could be a promising target for small molecule drugs to treat liver fibrosis by specifically activating autophagy. Collectively, these results show an unexpected role for Flcn in fatty liver disease progression and highlight new potential treatment strategies. Nature Publishing Group UK 2021-10-28 /pmc/articles/PMC8553785/ /pubmed/34711912 http://dx.doi.org/10.1038/s41598-021-99958-7 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Article
Paquette, Mathieu
Yan, Ming
Ramírez-Reyes, Josué M. J.
El-Houjeiri, Leeanna
Biondini, Marco
Dufour, Catherine R.
Jeong, Hyeonju
Pacis, Alain
Giguère, Vincent
Estall, Jennifer L.
Siegel, Peter M.
Audet-Walsh, Étienne
Pause, Arnim
Loss of hepatic Flcn protects against fibrosis and inflammation by activating autophagy pathways
title Loss of hepatic Flcn protects against fibrosis and inflammation by activating autophagy pathways
title_full Loss of hepatic Flcn protects against fibrosis and inflammation by activating autophagy pathways
title_fullStr Loss of hepatic Flcn protects against fibrosis and inflammation by activating autophagy pathways
title_full_unstemmed Loss of hepatic Flcn protects against fibrosis and inflammation by activating autophagy pathways
title_short Loss of hepatic Flcn protects against fibrosis and inflammation by activating autophagy pathways
title_sort loss of hepatic flcn protects against fibrosis and inflammation by activating autophagy pathways
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8553785/
https://www.ncbi.nlm.nih.gov/pubmed/34711912
http://dx.doi.org/10.1038/s41598-021-99958-7
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