DG9 boosts PMO nuclear uptake and exon skipping to restore dystrophic muscle and cardiac function.

DG9 促进 PMO 核摄取和外显子跳跃,从而恢复营养不良的肌肉和心脏功能

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作者:Shah Md Nur Ahad, Wilton-Clark Harry, Haque Farhia, Powell Brooklynn, Sutanto Laura Edellein, Maradiya Radha, Zhabyeyev Pavel, Roshmi Rohini Roy, Anwar Saeed, Aslesh Tejal, Lim Kenji Rowel Q, Maruyama Rika, Bigot Anne, Young Courtney S, Bittner Scott, Spencer Melissa J, Moulton Hong M, Oudit Gavin Y, Yokota Toshifumi
Duchenne muscular dystrophy (DMD) is a severe neuromuscular disorder caused by DMD gene mutations, leading to the loss of functional dystrophin. While antisense oligonucleotide (ASO)-mediated exon skipping offers therapeutic potential, its efficacy in cardiac muscle remains limited. Here, we investigate DG9, a cell-penetrating peptide derived from human polyhomeotic 1 homolog (Hph-1) transcription factor, as an enhancer of phosphorodiamidate morpholino oligomer (PMO)-based therapy targeting exon 44. In a humanized DMD mouse model (hDMDdel45;mdx), DG9-PMO significantly increases exon skipping, restores dystrophin expression, and improves muscle function, particularly in the heart. Mechanistically, DG9-PMO enhances intracellular uptake through multiple endocytic pathways and achieves superior nuclear localization. Compared to the benchmark R6G peptide, DG9-PMO exhibits greater efficacy in cardiac tissue with no detectable toxicity. These findings highlight DG9-PMO as a promising next-generation exon-skipping therapy with potential clinical relevance for improving both skeletal and cardiac outcomes in DMD patients.

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