Endogenous Multiple Exon Skipping and Back-Splicing at the DMD Mutation Hotspot

Int J Mol Sci. 2016 Oct 13;17(10):1722. doi: 10.3390/ijms17101722.

Abstract

Duchenne muscular dystrophy (DMD) is a severe muscular disorder. It was reported that multiple exon skipping (MES), targeting exon 45-55 of the DMD gene, might improve patients' symptoms because patients who have a genomic deletion of all these exons showed very mild symptoms. Thus, exon 45-55 skipping treatments for DMD have been proposed as a potential clinical cure. Herein, we detected the expression of endogenous exons 44-56 connected mRNA transcript of the DMD using total RNAs derived from human normal skeletal muscle by reverse transcription polymerase chain reaction (RT-PCR), and identified a total of eight types of MES products around the hotspot. Surprisingly, the 5' splice sites of recently reported post-transcriptional introns (remaining introns after co-transcriptional splicing) act as splicing donor sites for MESs. We also tested exon combinations to generate DMD circular RNAs (circRNAs) and determined the preferential splice sites of back-splicing, which are involved not only in circRNA generation, but also in MESs. Our results fit the current circRNA-generation model, suggesting that upstream post-transcriptional introns trigger MES and generate circRNA because its existence is critical for the intra-intronic interaction or for extremely distal splicing.

Keywords: DMD; circular RNA; multiple exon skipping; pre-mRNA splicing.

MeSH terms

  • Dystrophin / genetics*
  • Exons*
  • Gene Expression
  • Humans
  • Muscular Dystrophy, Duchenne / genetics*
  • Mutation
  • RNA Precursors / genetics*
  • RNA Splicing*
  • RNA, Messenger / genetics
  • Transcription, Genetic

Substances

  • DMD protein, human
  • Dystrophin
  • RNA Precursors
  • RNA, Messenger