Nonviral delivery of chemically modified tRNA rescues nonsense mutations in cystic fibrosis

Suppressor transfer RNAs (sup-tRNAs) can rescue disease-causing nonsense mutations by promoting readthrough of premature termination codons (PTCs). Their clinical translation is limited by suboptimal activity and inefficient in vivo delivery. In this work, we combined site-specific chemical modification of sup-tRNAs with cargo-tailored pulmonary lipid nanoparticle (LNP) engineering to overcome these barriers. Incorporation of N 1 -methyladenosine in sup-tRNAs improved PTC readthrough, enhanced tRNA aminoacylation, prolonged functional persistence, and reduced innate immune activation. High-throughput ionizable lipid screening and formulation optimization identified a sup-tRNA–tailored LNP that efficiently delivered chemically modified sup-tRNAs to the lung. This approach restored cystic fibrosis transmembrane conductance regulator (CFTR) expression and function in bronchial epithelial cells, mouse models, and patient-derived organoids. Thus, LNP-delivered, chemically engineered sup-tRNAs represent a potential therapeutic platform for treating nonsense mutations.

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Publication Details

Journal
Science
Published
2026-08-27
DOI
https://doi.org/10.1126/science.aeb0054
Citations
1
Primary Topic
RNA Interference and Gene Delivery
Type
article
Field-Weighted Citation Impact
3.03

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article

Nonviral delivery of chemically modified tRNA rescues nonsense mutations in cystic fibrosis

Tanja Gonska, Zhichang Peter Zhou, Sijin Luozhong, Jingyi Pan et al.
1 citations
Science
RNA Interference and Gene Delivery
3.03
article

Nonviral delivery of chemically modified tRNA rescues nonsense mutations in cystic fibrosis

Tanja Gonska, Zhichang Peter Zhou, Sijin Luozhong, Jingyi Pan, Santiago Tijaro‐Bulla, Rasangi Tennakoon, Haissi Cui, Jim Hu, Muye Zhou, Colette Maya Macarios, Bowen Li, Yue Xu, Jingan Chen, Songtao Dong, Breanna Y. Seto, Ziyan Rachel Chen, Fanglin Gong
article en
1 citations

Abstract

Suppressor transfer RNAs (sup-tRNAs) can rescue disease-causing nonsense mutations by promoting readthrough of premature termination codons (PTCs). Their clinical translation is limited by suboptimal activity and inefficient in vivo delivery. In this work, we combined site-specific chemical modification of sup-tRNAs with cargo-tailored pulmonary lipid nanoparticle (LNP) engineering to overcome these barriers. Incorporation of N 1 -methyladenosine in sup-tRNAs improved PTC readthrough, enhanced tRNA aminoacylation, prolonged functional persistence, and reduced innate immune activation. High-throughput ionizable lipid screening and formulation optimization identified a sup-tRNA–tailored LNP that efficiently delivered chemically modified sup-tRNAs to the lung. This approach restored cystic fibrosis transmembrane conductance regulator (CFTR) expression and function in bronchial epithelial cells, mouse models, and patient-derived organoids. Thus, LNP-delivered, chemically engineered sup-tRNAs represent a potential therapeutic platform for treating nonsense mutations.

ScienceVol. 393(6814)
University Health Network (CA), University of Toronto (CA), Hospital for Sick Children (CA), Princess Margaret Cancer Centre (CA)
Cystic Fibrosis Foundation, J.P. Bickell Foundation, Cystic Fibrosis Canada, Canada Research Chairs, Connaught Fund, National Institutes of Health, Harrington Discovery Institute, University Hospitals, Canadian Institutes of Health Research, Natural Sciences and Engineering Research Council of Canada, Leslie Dan Faculty of Pharmacy, University of Toronto
Good health and well-being
Openalex Percentile: Top 6%
RNA Interference and Gene Delivery
3.03
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