Exon 44 Skipping Oligonucleotides for DMD Splicing Specificity

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Solution Overview

Problem

Current treatments for Duchenne Muscular Dystrophy (DMD) and Becker Muscular Dystrophy (BMD) using antisense-mediated exon skipping face challenges in efficiently skipping exon 44 of the DMD gene without affecting conserved splice sites or inducing immunogenicity, and in achieving optimal RNA binding kinetics and thermodynamic properties.

Innovation Solution

A method involving molecules that bind to specific nucleotide sequences within exon 44, such as SEQ ID NO. 1, 2, 3, or 4, to modulate splicing, using oligonucleotides that are designed to avoid CpG pairs and G-quartet motifs, ensuring efficient skipping of exon 44 while minimizing immunogenicity and optimizing RNA binding properties.

Engineering Contradictions & Design Principles

VSEngineering Contradiction Analysis

1Productivity

If antisense oligonucleotides target conserved splice sites to induce exon skipping, then exon skipping efficiency is improved, but specificity deteriorates due to co-targeting of additional exons

Engineering Contradiction:
Improveexon skipping efficiencyVSAvoidsplicing specificity
Core Design Contradiction:
ProductivityVSManufacturing precision

Solution Approach 1:

The patent applies local quality by transitioning from targeting conserved sequences (uniform quality across all splice sites) to targeting exon-specific non-conserved sequences (differentiated quality for each target). The AONs are designed to bind to unique sequences within exon 44 that do not exist in other exons, thereby achieving high specificity while maintaining skipping efficiency.

Inventive Principle:
Principle #3Local quality

Solution Approach 2:

The patent segments the target space by identifying four distinct non-overlapping regions within exon 44 (SEQ ID NO:1-4), each with unique sequences. This segmentation allows selection of region-specific AONs that will not cross-react with other exons, resolving the specificity issue while maintaining efficiency through optimized binding to each segmented region.

Inventive Principle:
Principle #1Segmentation

2Productivity

If oligonucleotides are designed for high binding affinity to exon 44, then skipping efficiency is improved, but immunogenicity increases due to CpG pairs and G-quartet motifs

Engineering Contradiction:
Improveexon skipping efficiencyVSAvoidimmunogenicity
Core Design Contradiction:
ProductivityVSObject-affected harmful factors

Solution Approach 1:

The patent extracts and removes harmful immunogenic elements (CpG pairs and G-quartet motifs) from the oligonucleotide sequence while retaining the essential binding functionality. By carefully selecting sequences from four regions of exon 44 and excluding those containing immunogenic motifs, the patent achieves high skipping efficiency without triggering immune responses.

Inventive Principle:
Principle #2Taking out (Extraction)

Solution Approach 2:

The patent changes the chemical and sequence parameters of the oligonucleotides by selecting specific sequences that optimize RNA binding kinetics and thermodynamic properties while eliminating immunogenic features. This parameter optimization allows maintenance of high binding affinity and skipping efficiency without the harmful side effects of CpG pairs and G-quartet structures.

Inventive Principle:
Principle #35Parameter changes

Applied Scientific Principles

This section explains which scientific principles are used to turn an abstract innovation direction into a practical engineering solution.

Function Achieved in This Case

The method achieves highly efficient skipping of exon 44, restoring the reading frame in DMD patients, producing functional dystrophin protein, and reducing aberrant dystrophin production, thereby alleviating muscle cell characteristics associated with DMD and improving muscle function.

Implementation Method 1

a molecule that binds to a nucleotide sequence comprising SEQ ID NO.1: 5'-GUGGCUAACAGAAGCU; SEQ ID NO.2: 5'-GGGAACAUGCUAAAUAC, SEQ ID NO.3: 5'-AGACACAAAUUCCUGAGA, or SEQ ID NO.4: 5'-CUGUUGAGAAA. This molecule preferably binds or is complementary to any of SEQ ID NO: 1, 2, 3, or 4 when SEQ ID NO:1, 2, 3, or 4 is present within exon 44 of the DMD pre-mRNA.

Methodology Applied
Scientific EffectComplementary base pairing:

Data Source

PatentEP3872178A1Method for efficient exon (44) skipping in duchenne muscular dystrophy and associated means
Publication Date: 2021.09.01 BIOMARIN TECHNOLOGIES BV
  • EP3872178A1 patent drawingFigure 1a~1c
  • EP3872178A1 patent drawingFigure 2a~2b
  • EP3872178A1 patent drawingFigure 3a~3b

AI summary

The invention relates to a nucleic acid molecule that binds and/or is complementary to the nucleotide molecule having sequence 5'-GUGGCUAACAGAAGCU (SEQ ID NO 1) and to its use in a method for inducing skipping of exon 44 of the DMD gene in a DMD patient.