eprintid: 1545307 rev_number: 36 eprint_status: archive userid: 608 dir: disk0/01/54/53/07 datestamp: 2017-03-27 13:29:32 lastmod: 2021-12-10 01:13:06 status_changed: 2017-03-27 13:29:32 type: article metadata_visibility: show creators_name: Godfrey, C creators_name: Desviat, LR creators_name: Smedsrød, B creators_name: Piétri-Rouxel, F creators_name: Denti, MA creators_name: Disterer, P creators_name: Lorain, S creators_name: Nogales-Gadea, G creators_name: Sardone, V creators_name: Anwar, R creators_name: El Andaloussi, S creators_name: Lehto, T creators_name: Khoo, B creators_name: Brolin, C creators_name: van Roon-Mom, WM creators_name: Goyenvalle, A creators_name: Aartsma-Rus, A creators_name: Arechavala-Gomeza, V title: Delivery is key: lessons learnt from developing splice-switching antisense therapies ispublished: pub divisions: UCL divisions: B02 divisions: C10 divisions: D17 divisions: G93 divisions: D14 keywords: RNA therapy, antisense oligonucleotides, delivery, pre‐clinical models, toxicity note: This is an open access article under the terms of the Creative Commons Attribution 4.0 License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited. abstract: The use of splice-switching antisense therapy is highly promising, with a wealth of pre-clinical data and numerous clinical trials ongoing. Nevertheless, its potential to treat a variety of disorders has yet to be realized. The main obstacle impeding the clinical translation of this approach is the relatively poor delivery of antisense oligonucleotides to target tissues after systemic delivery. We are a group of researchers closely involved in the development of these therapies and would like to communicate our discussions concerning the validity of standard methodologies currently used in their pre-clinical development, the gaps in current knowledge and the pertinent challenges facing the field. We therefore make recommendations in order to focus future research efforts and facilitate a wider application of therapeutic antisense oligonucleotides. date: 2017-03-01 date_type: published official_url: http://dx.doi.org/10.15252/emmm.201607199 oa_status: green full_text_type: pub language: eng primo: open primo_central: open_green verified: verified_manual elements_id: 1214486 doi: 10.15252/emmm.201607199 pii: emmm.201607199 lyricists_name: Disterer, Petra lyricists_name: Khoo, Bernard lyricists_id: PDIST62 lyricists_id: BKHOO30 actors_name: Disterer, Petra actors_id: PDIST62 actors_role: owner full_text_status: public publication: EMBO Molecular Medicine volume: 9 number: 3 pagerange: 281-394 event_location: England issn: 1757-4684 citation: Godfrey, C; Desviat, LR; Smedsrød, B; Piétri-Rouxel, F; Denti, MA; Disterer, P; Lorain, S; ... Arechavala-Gomeza, V; + view all <#> Godfrey, C; Desviat, LR; Smedsrød, B; Piétri-Rouxel, F; Denti, MA; Disterer, P; Lorain, S; Nogales-Gadea, G; Sardone, V; Anwar, R; El Andaloussi, S; Lehto, T; Khoo, B; Brolin, C; van Roon-Mom, WM; Goyenvalle, A; Aartsma-Rus, A; Arechavala-Gomeza, V; - view fewer <#> (2017) Delivery is key: lessons learnt from developing splice-switching antisense therapies. EMBO Molecular Medicine , 9 (3) pp. 281-394. 10.15252/emmm.201607199 <https://doi.org/10.15252/emmm.201607199>. Green open access document_url: https://discovery.ucl.ac.uk/id/eprint/1545307/1/Godfrey_2017%20Delivery%20is%20key%20-%20lessons%20learnt%20from%20developing%20splice-switching%20antisense%20therapies.pdf