ArticleMethods in molecular biology (Clifton, N.J.)2026
Splice Modulation Studies Using Reporter Mice.
Article in Methods in molecular biology (Clifton, N.J.), 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
Exon-skipping therapy for Duchenne muscular dystrophy (DMD) recovers muscle function by inducing shorter but functional dystrophin protein using antisense oligonucleotides (ASOs) that modulate the pre-mRNA splicing of DMD transcripts to restore the disrupted DMD reading frame. Next-generation ASO should have appropriate sequences and modifications that induce exon skipping and ensure efficient delivery to target tissues. Conventional methods involve analyzing biopsied tissues using RT-PCR or immunoblotting to evaluate exon skipping or the expression of induced proteins. However, these methods are laborious and time-consuming, resulting in a low throughput. In this chapter, we present a novel evaluation method using reporter mice. Exon skipping induces the expression of fluorescent proteins enabling noninvasive monitoring. This in vivo method allows for a high-throughput evaluation of the efficacy of ASO-induced exon skipping.
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