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. 2024 Feb 28;627(8003):416–423. doi: 10.1038/s41586-024-07087-8

Extended Data Fig. 1. In vitro selection of the most effective ETRs for Pcsk9 silencing.

Extended Data Fig. 1

a, Schematic drawing showing on top the Pcsk9 promoter region with the annotated CpG Island (CGI) and, on the bottom, a zoom on the CGI showing the target sites of all the tested single guide RNAs (sgRNAs; black arrows), TALEs (grey arrows), and ZFPs (blue arrows). Filled arrows indicate the most active sgRNA/DBDs used for subsequent experiments. Created with BioRender.com. b, Schematic representation of the plasmid used for ETR expression, either after its direct transfection into cells or as a template for In Vitro Transcription (IVT) of the ETRs’ mRNA. CMV: enhancer/promoter of the Cytomegalovirus. T7: promoter for mRNA production. ATG: start codon; DBDs: DNA-binding domains; SV40 NLS: nuclear localization signal of the simian virus 40; GSGGG: glycine-rich liker peptide; ED: effector domain, either KRAB from the ZNF10 protein, cdDNMT3A or DNMT3L; WPRE: woodchuck hepatitis virus post-transcriptional regulatory element; 64A: stretch of 64 adenines; SpeI: restriction site used to linearize the plasmid for IVT; BGH polyA: polyadenylation signal from the bovine growth hormone gene. Created with BioRender.com. c, Dot plot showing the percentage of Pcsk9tdTomato-negative cells over a period of 22 days post-delivery of plasmids encoding for the indicated dCas9-based ETRs and 8 different sgRNAs. sgRNA-4 was the most active among the tested guides (black dots and connecting line) and thus used for subsequent experiments. Data are reported as mean (n = 2). d, Dot plot showing the percentage of Pcsk9tdTomato-negative cells over a period of 17 days post-delivery of plasmids encoding for 16 different TALE DBDs fused to the KRAB domain. This experiment was meant to identify the most effective TALEs among those tested, using KRAB-mediated epi-silencing of Pcsk9 as a surrogate readout for DBD efficiency. TALE-2, -4 and -6 were the most active ones among those tested (black dots and connecting line) and thus used for subsequent experiments. Data are reported as mean ± s.d. (n = 4). e, Dot plot showing the percentage of Pcsk9tdTomato-negative cells over a period of 22 days post-delivery of plasmids encoding for 16 different ZFP DBDs fused to the KRAB domain. This experiment was meant to identify the most effective ZFPs among those tested, using KRAB-mediated epi-silencing of Pcsk9 as a surrogate readout for DBD efficiency. ZFP-3, -6 and -8 were the most active ones among those tested (blue dots and connecting line) and thus used for subsequent experiments. Data are reported as mean ± s.d. (n = 4). f, Left: heat maps showing the percentage of Pcsk9tdTomato-negative cells at day 7 post-delivery of combinations of plasmids encoding for KRAB-, DNMT3L and cDNMT3A-based ETRs containing TALEs. The matrixes were built by transfecting either one of the TALE-KRAB ETRs from d with all possible combinations of the three best-performing TALE DBDs (namely, 3, 6 and 8) fused to either DNMT3L (y axis) or cdDNMT3A (x axis). Given its highest performance, the triple-ETR combination containing TALE-2-KRAB, TALE-6-DNMT3L, and TALE-4-cDNMT3A was chosen for further studies. Colour intensity refers to average silencing efficiency (n = 2). Right: similar experiment as in left but performed with the three best-performing ZFPs from e. Given its highest activity, the triple-ETR combination containing ZFP-8-KRAB, ZFP-6-DNMT3L and ZFP-3-cDNMT3A was chosen for further studies. Colour intensity refers to average silencing efficiency (n = 3). Best-performing triple combinations of TALE- and ZFP-ETRs are indicated with a red square. g, Time-course analysis of Pcsk9tdTomato-negative cells from the 0.5 µg RNA treatment conditions in Fig. 1b of the main text. Data are reported as mean ± s.d.

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