Fig. 3: SETD1A targets active promoter regions of common SCZ risk genes, regulating chromatin remodeling, DNA repair, and synaptic signaling pathways in human brain development.

a Schematic of brain tissue dissected and Allen Human Brain Atlas image at post-conception week (PCW) 21 https:/atlas.brain-map.org/atlas?atlas=3#atlas=3&plate=101892621&structure=11585&x=10116&y=23872&zoom=-7&resolution=128.08&z=372&zoom=-7&resolution=128.08&z=3. Human prenatal cortices were dissected to include ventricular zone through marginal zone. b Proportion of SETD1A peaks at active promoters in human prenatal cortices and isogenic iPSC-derived NPCs and neurons. c Representative IGV tracks of CUT&Tag signals for H3K4me3, SETD1A_CST, SETD1A_Atlas, and control IgG on chr16. Black bars indicate peaks at active promoters. d Overrepresentation of SETD1A peaks in human prenatal cortices and isogenic iPSC-derived NPCs and neurons on 287 SCZ GWAS loci5. Permutation test, ****p < 0.0001. e LDSC analysis showing heritability enrichment for psychiatric disorders (SCZ, BD, MDD), neurodevelopmental disorders (ASD, ADHD), neurodegenerative disorders (AD, PD), stroke, and T2DM across SETD1A binding sites. *p < 0.05; ***p < 0.001; ****p < 0.0001. f GO:BP terms enriched for SCZ GWAS significant genes with SETD1A-bound active promoters in human prenatal cortices (165 genes), WT NPCs (199 genes) and WT neurons (199 genes) among 682 SCZ GWAS significant genes5. *FDR < 0.05; **FDR < 0.01; ***FDR < 0.001; ****FDR < 0.0001. g Comparison of SETD1A peaks at active promoters between genotypes (one WT and one SETD1Ac.4582-2delAG/+ lines with technical duplicates for NPCs; two WT and two SETD1Ac.4582-2delAG/+ lines for neurons). Unpaired t-test, **p = 0.0085; n.s. not significant. h Venn diagram showing overlap of SETD1A peaks at active promoters between genotypes. Numbers indicate peak counts; parentheses indicate annotated gene counts. Red parentheses indicate SCZ GWAS-significant genes. i Representative IGV tracks showing lost SETD1A binding in SETD1Ac.4582-2delAG/+ cells (dashed boxes). j Comparison of SETD1A and H3K4me3 binding densities at lost/gained regions. Wilcoxon signed-rank test, ***p = 0.00012; ****p < 0.0001. k GO:BP terms enriched in genes with lost promoter regions and enrichment of 116 SCZ GWAS significant genes. *FDR < 0.05; **FDR < 0.01; ***FDR < 0.001; ****FDR < 0.0001. l Expression of 116 SCZ GWAS significant genes with lost promoter regions. Wilcoxon signed-rank test, ****p < 0.0001. Data presented as mean ± S.E.M. (b), mean ± S.D. (g), median with 25th–75th percentiles and min-max whiskers (j, l). All tests two-sided. RPKM reads per kilobase per million, TPM transcripts per million.