Synthesis, Biological Activity, and Molecular Dynamics Simulations of LNA‐Charge Neutral Linkages for Enhanced Splice‐Switching Antisense Oligonucleotides

A Alice Kennett (Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK) L Lillian Lie (Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK) M Martin Flerin (Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK) B Belma Zengin Kurt (Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK) Y Ysobel R. Baker (Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK) A Alyssa C. Hill (Department of Paediatrics Institute of Developmental and Regenerative Medicine (IDRM) University of Oxford Oxford OX3 7TY UK) A Abinaya Ramesh (Department of Paediatrics Institute of Developmental and Regenerative Medicine (IDRM) University of Oxford Oxford OX3 7TY UK) M Matthew J.A. Wood (Department of Paediatrics Institute of Developmental and Regenerative Medicine (IDRM) University of Oxford Oxford OX3 7TY UK) D Debashis Dhara (Chemistry Research Laboratory, Department of Chemistry, University of Oxford, 12 Mansfield Road, Oxford OX1 3TA, U.K.) A Afaf H. El‐Sagheer (Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK) F Fernanda Duarte (Chemistry Research Laboratory) T Tom Brown (Chemistry Research Laboratory, Department of Chemistry, University of Oxford, 12 Mansfield Road, Oxford OX1 3TA, U.K.)

Abstract

Abstract Antisense oligonucleotides are promising therapeutic agents for a range of diseases, having found special clinical success for splice‐switching genetic conditions such as spinal muscular atrophy and Duchenne muscular dystrophy. However, novel chemistries are still required to discover modifications which improve their druggable properties. For in vitro studies, thermal duplex stability, resistance to enzymatic degradation and gymnotic cellular activity are important, and biodistribution, toxicology and potency must be optimised for clinical progression. We investigate the combination of locked nucleic acids (LNA) and charge neutral backbones in chimeric ASOs containing 2′‐O‐methyl sugars and phosphorothioate backbones by evaluating their physical and biological properties. Backbones investigated are LNA‐amide, LNA‐carbamate, LNA‐alkoxyamide, and LNA‐sulfamate. Molecular dynamics simulations of these LNA‐charge neutral backbones were conducted to explore the structural features which determine the experimentally observed thermal duplex stability and conformation. The LNA‐sulfamate linkage is of particular interest, forming very stable duplexes with its RNA target and having comparable gymnotic activity to the previously investigated LNA‐amide, while being synthetically more accessible. Together, our studies indicate that a multi‐faceted approach to expanding the ASO chemical space, using a combination of computational and experimental methods, can build structure‐activity relationships and discover novel promising backbones for future therapeutic use.

Article Details

Volume / Issue Vol. 64, Issue 46
Published November 10, 2025
ISSN 1433-7851
Publisher Wiley

Journal Info

Angewandte Chemie International Edition

Wiley

ISSN: 1433-7851 Physical Sciences

Authors (12)

A

Alice Kennett

Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK

L

Lillian Lie

Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK

M

Martin Flerin

Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK

B

Belma Zengin Kurt

Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK

Y

Ysobel R. Baker

Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK

A

Alyssa C. Hill

Department of Paediatrics Institute of Developmental and Regenerative Medicine (IDRM) University of Oxford Oxford OX3 7TY UK

A

Abinaya Ramesh

Department of Paediatrics Institute of Developmental and Regenerative Medicine (IDRM) University of Oxford Oxford OX3 7TY UK

M

Matthew J.A. Wood

Department of Paediatrics Institute of Developmental and Regenerative Medicine (IDRM) University of Oxford Oxford OX3 7TY UK

D

Debashis Dhara

Chemistry Research Laboratory, Department of Chemistry, University of Oxford, 12 Mansfield Road, Oxford OX1 3TA, U.K.

A

Afaf H. El‐Sagheer

Department of Chemistry University of Oxford Chemistry Research Laboratory 12 Mansfield Road Oxford OX1 3TA UK

F

Fernanda Duarte

Chemistry Research Laboratory

T

Tom Brown

Chemistry Research Laboratory, Department of Chemistry, University of Oxford, 12 Mansfield Road, Oxford OX1 3TA, U.K.