Site-Specific RNA Editing of Stop Mutations in the CFTR mRNA of Human Bronchial Cultured Cells

Int J Mol Sci. 2023 Jun 30;24(13):10940. doi: 10.3390/ijms241310940.

Abstract

It is reported that about 10% of cystic fibrosis (CF) patients worldwide have nonsense (stop) mutations in the CFTR gene, which cause the premature termination of CFTR protein synthesis, leading to a truncated and non-functional protein. To address this issue, we investigated the possibility of rescuing the CFTR nonsense mutation (UGA) by sequence-specific RNA editing in CFTR mutant CFF-16HBEge, W1282X, and G542X human bronchial cells. We used two different base editor tools that take advantage of ADAR enzymes (adenosine deaminase acting on RNA) to edit adenosine to inosine (A-to-I) within the mRNA: the REPAIRv2 (RNA Editing for Programmable A to I Replacement, version 2) and the minixABE (A to I Base Editor). Immunofluorescence experiments show that both approaches were able to recover the CFTR protein in the CFTR mutant cells. In addition, RT-qPCR confirmed the rescue of the CFTR full transcript. These findings suggest that site-specific RNA editing may efficiently correct the UGA premature stop codon in the CFTR transcript in CFF-16HBEge, W1282X, and G542X cells. Thus, this approach, which is safer than acting directly on the mutated DNA, opens up new therapeutic possibilities for CF patients with nonsense mutations.

Keywords: CRISPR/dCas13; REPAIRv2; RNA editing; cystic fibrosis; mxABE; premature termination codons (PTCs).

MeSH terms

  • Cell Line
  • Codon, Terminator
  • Cystic Fibrosis Transmembrane Conductance Regulator* / genetics
  • Cystic Fibrosis Transmembrane Conductance Regulator* / metabolism
  • Cystic Fibrosis* / drug therapy
  • Cystic Fibrosis* / therapy
  • Humans
  • Mutation
  • RNA Editing / genetics
  • RNA, Messenger / genetics
  • RNA, Messenger / metabolism

Substances

  • Cystic Fibrosis Transmembrane Conductance Regulator
  • RNA, Messenger
  • Codon, Terminator
  • CFTR protein, human