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Small Molecule Anion Carriers Correct Abnormal Airway Surface Liquid Properties in Cystic Fibrosis Airway Epithelia

Cystic fibrosis (CF) is a genetic disease characterized by the lack of cystic fibrosis transmembrane conductance regulator (CFTR) protein expressed in epithelial cells. The resulting defective chloride and bicarbonate secretion and imbalance of the transepithelial homeostasis lead to abnormal airway...

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Autores principales: Gianotti, Ambra, Capurro, Valeria, Delpiano, Livia, Mielczarek, Marcin, García-Valverde, María, Carreira-Barral, Israel, Ludovico, Alessandra, Fiore, Michele, Baroni, Debora, Moran, Oscar, Quesada, Roberto, Caci, Emanuela
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7073096/
https://www.ncbi.nlm.nih.gov/pubmed/32098269
http://dx.doi.org/10.3390/ijms21041488
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author Gianotti, Ambra
Capurro, Valeria
Delpiano, Livia
Mielczarek, Marcin
García-Valverde, María
Carreira-Barral, Israel
Ludovico, Alessandra
Fiore, Michele
Baroni, Debora
Moran, Oscar
Quesada, Roberto
Caci, Emanuela
author_facet Gianotti, Ambra
Capurro, Valeria
Delpiano, Livia
Mielczarek, Marcin
García-Valverde, María
Carreira-Barral, Israel
Ludovico, Alessandra
Fiore, Michele
Baroni, Debora
Moran, Oscar
Quesada, Roberto
Caci, Emanuela
author_sort Gianotti, Ambra
collection PubMed
description Cystic fibrosis (CF) is a genetic disease characterized by the lack of cystic fibrosis transmembrane conductance regulator (CFTR) protein expressed in epithelial cells. The resulting defective chloride and bicarbonate secretion and imbalance of the transepithelial homeostasis lead to abnormal airway surface liquid (ASL) composition and properties. The reduced ASL volume impairs ciliary beating with the consequent accumulation of sticky mucus. This situation prevents the normal mucociliary clearance, favouring the survival and proliferation of bacteria and contributing to the genesis of CF lung disease. Here, we have explored the potential of small molecules capable of facilitating the transmembrane transport of chloride and bicarbonate in order to replace the defective transport activity elicited by CFTR in CF airway epithelia. Primary human bronchial epithelial cells obtained from CF and non-CF patients were differentiated into a mucociliated epithelia in order to assess the effects of our compounds on some key properties of ASL. The treatment of these functional models with non-toxic doses of the synthetic anionophores improved the periciliary fluid composition, reducing the fluid re-absorption, correcting the ASL pH and reducing the viscosity of the mucus, thus representing promising drug candidates for CF therapy.
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spelling pubmed-70730962020-03-19 Small Molecule Anion Carriers Correct Abnormal Airway Surface Liquid Properties in Cystic Fibrosis Airway Epithelia Gianotti, Ambra Capurro, Valeria Delpiano, Livia Mielczarek, Marcin García-Valverde, María Carreira-Barral, Israel Ludovico, Alessandra Fiore, Michele Baroni, Debora Moran, Oscar Quesada, Roberto Caci, Emanuela Int J Mol Sci Article Cystic fibrosis (CF) is a genetic disease characterized by the lack of cystic fibrosis transmembrane conductance regulator (CFTR) protein expressed in epithelial cells. The resulting defective chloride and bicarbonate secretion and imbalance of the transepithelial homeostasis lead to abnormal airway surface liquid (ASL) composition and properties. The reduced ASL volume impairs ciliary beating with the consequent accumulation of sticky mucus. This situation prevents the normal mucociliary clearance, favouring the survival and proliferation of bacteria and contributing to the genesis of CF lung disease. Here, we have explored the potential of small molecules capable of facilitating the transmembrane transport of chloride and bicarbonate in order to replace the defective transport activity elicited by CFTR in CF airway epithelia. Primary human bronchial epithelial cells obtained from CF and non-CF patients were differentiated into a mucociliated epithelia in order to assess the effects of our compounds on some key properties of ASL. The treatment of these functional models with non-toxic doses of the synthetic anionophores improved the periciliary fluid composition, reducing the fluid re-absorption, correcting the ASL pH and reducing the viscosity of the mucus, thus representing promising drug candidates for CF therapy. MDPI 2020-02-21 /pmc/articles/PMC7073096/ /pubmed/32098269 http://dx.doi.org/10.3390/ijms21041488 Text en © 2020 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Gianotti, Ambra
Capurro, Valeria
Delpiano, Livia
Mielczarek, Marcin
García-Valverde, María
Carreira-Barral, Israel
Ludovico, Alessandra
Fiore, Michele
Baroni, Debora
Moran, Oscar
Quesada, Roberto
Caci, Emanuela
Small Molecule Anion Carriers Correct Abnormal Airway Surface Liquid Properties in Cystic Fibrosis Airway Epithelia
title Small Molecule Anion Carriers Correct Abnormal Airway Surface Liquid Properties in Cystic Fibrosis Airway Epithelia
title_full Small Molecule Anion Carriers Correct Abnormal Airway Surface Liquid Properties in Cystic Fibrosis Airway Epithelia
title_fullStr Small Molecule Anion Carriers Correct Abnormal Airway Surface Liquid Properties in Cystic Fibrosis Airway Epithelia
title_full_unstemmed Small Molecule Anion Carriers Correct Abnormal Airway Surface Liquid Properties in Cystic Fibrosis Airway Epithelia
title_short Small Molecule Anion Carriers Correct Abnormal Airway Surface Liquid Properties in Cystic Fibrosis Airway Epithelia
title_sort small molecule anion carriers correct abnormal airway surface liquid properties in cystic fibrosis airway epithelia
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7073096/
https://www.ncbi.nlm.nih.gov/pubmed/32098269
http://dx.doi.org/10.3390/ijms21041488
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