| Literature DB >> 32059764 |
Chiara De Santi1, Elena Fernández Fernández2, Rachel Gaul3, Sebastian Vencken2, Arlene Glasgow2, Irene K Oglesby4, Killian Hurley4, Finn Hawkins5, Nilay Mitash6, Fangping Mu7, Rana Raoof8, David C Henshall8, Meritxell B Cutrona9, Jeremy C Simpson9, Brian J Harvey10, Barry Linnane11, Paul McNally12, Sally Ann Cryan3, Ronan MacLoughlin13, Agnieszka Swiatecka-Urban6, Catherine M Greene2.
Abstract
MicroRNAs that are overexpressed in cystic fibrosis (CF) bronchial epithelial cells (BEC) negatively regulate CFTR and nullify the beneficial effects of CFTR modulators. We hypothesized that it is possible to reverse microRNA-mediated inhibition of CFTR using CFTR-specific target site blockers (TSBs) and to develop a drug-device combination inhalation therapy for CF. Lead microRNA expression was quantified in a series of human CF and non-CF samples and in vitro models. A panel of CFTR 3' untranslated region (UTR)-specific locked nucleic acid antisense oligonucleotide TSBs was assessed for their ability to increase CFTR expression. Their effects on CFTR activity alone or in combination with CFTR modulators were measured in CF BEC models. TSB encapsulation in poly-lactic-co-glycolic acid (PLGA) nanoparticles was assessed as a proof of principle of delivery into CF BECs. TSBs targeting the CFTR 3' UTR 298-305:miR-145-5p or 166-173:miR-223-3p sites increased CFTR expression and anion channel activity and enhanced the effects of ivacaftor/lumacaftor or ivacaftor/tezacaftor in CF BECs. Biocompatible PLGA-TSB nanoparticles promoted CFTR expression in primary BECs and retained desirable biophysical characteristics following nebulization. Alone or in combination with CFTR modulators, aerosolized CFTR-targeting TSBs encapsulated in PLGA nanoparticles could represent a promising drug-device combination therapy for the treatment for CFTR dysfunction in the lung.Entities:
Keywords: ALI culture; CFTR; CFTR modulators; High content screening; Primary bronchial epithelial cells; RNA sequencing; iPSC-derived CF and CFTR gene-corrected bronchosperes; microRNA; nebulised PLGA nanoparticles; target site blocker
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Year: 2020 PMID: 32059764 PMCID: PMC7132615 DOI: 10.1016/j.ymthe.2020.02.001
Source DB: PubMed Journal: Mol Ther ISSN: 1525-0016 Impact factor: 11.454