7',5'-alpha-bicyclo-DNA: new chemistry for oligonucleotide exon splicing modulation therapy.


Journal

Nucleic acids research
ISSN: 1362-4962
Titre abrégé: Nucleic Acids Res
Pays: England
ID NLM: 0411011

Informations de publication

Date de publication:
02 12 2021
Historique:
accepted: 25 10 2021
revised: 12 10 2021
received: 11 12 2020
pubmed: 2 12 2021
medline: 11 1 2022
entrez: 1 12 2021
Statut: ppublish

Résumé

Antisense oligonucleotides are small pieces of modified DNA or RNA, which offer therapeutic potential for many diseases. We report on the synthesis of 7',5'-α-bc-DNA phosphoramidite building blocks, bearing the A, G, T and MeC nucleobases. Solid-phase synthesis was performed to construct five oligodeoxyribonucleotides containing modified thymidine residues, as well as five fully modified oligonucleotides. Incorporations of the modification inside natural duplexes resulted in strong destabilizing effects. However, fully modified strands formed very stable duplexes with parallel RNA complements. In its own series, 7',5'-α-bc-DNA formed duplexes with a surprising high thermal stability. CD spectroscopy and extensive molecular modeling indicated the adoption by the homo-duplex of a ladder-like structure, while hetero-duplexes with DNA or RNA still form helical structure. The biological properties of this new modification were investigated in animal models for Duchenne muscular dystrophy and spinal muscular atrophy, where exon splicing modulation can restore production of functional proteins. It was found that the 7',5'-α-bc-DNA scaffold confers a high biostability and a good exon splicing modulation activity in vitro and in vivo.

Identifiants

pubmed: 34850138
pii: 6442724
doi: 10.1093/nar/gkab1097
pmc: PMC8643641
doi:

Substances chimiques

Oligonucleotides 0
DNA 9007-49-2

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

12089-12105

Informations de copyright

© The Author(s) 2021. Published by Oxford University Press on behalf of Nucleic Acids Research.

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Auteurs

Damien Evéquoz (D)

Alpha Anomeric, 140 Bis, Rue de Rennes, 75006 Paris, France.

Ingrid E C Verhaart (IEC)

Department of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.

Davy van de Vijver (D)

Department of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.

Wolfgang Renner (W)

Alpha Anomeric, 140 Bis, Rue de Rennes, 75006 Paris, France.

Annemieke Aartsma-Rus (A)

Department of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.

Christian J Leumann (CJ)

Department of Chemistry and Biochemistry, University of Bern, Freiestrasse 3, CH-3012 Bern, Switzerland.

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Classifications MeSH