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EXP001988

Paper

Bi-specific splice-switching PMO oligonucleotides conjugated via a single peptide active in a mouse model of Duchenne muscular dystrophy (2015)

Peptide

Bi-specific D4 (C-terminal disulfide linkage for second PMO)

Sequence: RXRRBRRXRYQFLIRXRBRXRB

RNA

All experiment fields

Experiment Id EXP001988
Paper Bi-specific splice-switching PMO oligonucleotides conjugated via a single peptide active in a mouse
Peptide Bi-specific D4 (C-terminal disulfide linkage for second PMO)
Delivery Success Class no
In Vivo Flag no
Uptake Confirmed no
Label Confidence high
In Vitro Functional Effect yes
Endosomal Escape Evidence
Peptide Concentration 0.5–1 µM conjugate (serum-free Opti-MEM, 4 h transfection; then DMEM/5% horse serum 20 h).
Rna Concentration Same as conjugate concentration (PMO content equimolar to conjugate; for bi-specific constructs total PMO per conjugate contains two PMOs).
Mixing Ratio Covalent bi-specific: second PMO attached via reducible disulfide bond (3′-NPys PMO + Cys on Pip6a derivative).
Formulation Format Covalent CPP–PMO conjugate (P-PMO); bi-specific constructs carry two PMOs on one CPP via amide + triazole (click) or disulfide linkages.
Formulation Components Pip6a CPP conjugated to PMO(s): Dmd exon23 PMO and/or Acvr2b exon5 PMO (see construct-specific linkage notes).
Size Nm
Zeta Mv
Model Scope in_vitro
Model Type in vitro
Cell Lines Or Primary Cells H2K/mdx mouse myoblasts differentiated to myotubes (mdx dystrophin model)
Animal Model
Administration Route
Output Type In vitro functional RNA effect: exon skipping (RT-PCR) for Dmd exon 23 and Acvr2b exon 5
Output Value Exon skipping observed but less effective than D3 (RT-PCR).
Output Units
Output Notes Authors report no advantage of cleavable disulfide vs stable click linkage in this CPP–PMO system.
Toxicity Notes
Curation Notes