The authors performed ATAC-seq on primary human keratinocytes from EEC syndrome patients carrying p63 DNA-binding-domain mutations (R204W, R304W) plus one control donor. They identified 2,492 control-specific open chromatin regions (Ctr-OCRs) and 3,716 mutant-specific OCRs, with differential OCRs correlating positively with nearby differential gene expression (cell-cycle genes near Ctr-OCRs; actin filament-based processes near Mt-OCRs) .
Motif analysis revealed p63 as the most enriched motif in Ctr-OCRs, with CTCF unexpectedly second. Critically, at the 376 CTCF-bound Ctr-OCRs, ATAC signal dropped in mutants but CTCF ChIP-seq signal was unchanged (only 363 of 12,116 CTCF sites were differential), whereas p63 binding clearly decreased at its 1,223-bound Ctr-OCRs. Overlap between p63 and CTCF binding sites was minimal (~435 of 45,350 vs 12,116 sites), arguing against direct protein-protein co-occupancy .
Integrating published keratinocyte promoter-capture Hi-C, the authors mapped 388 p63-bound and 191 CTCF-bound Ctr-OCRs to loop anchors, and found 96 genes significantly co-connected by both (p < 0.001); 39 of these were deregulated in mutants, including KRT5 and KRT75. In 70.6% of same-loop cases both factors sat at the promoter, and in 12.6% p63 and CTCF occupied opposite anchors .
The "inactive loop" interpretation β that CTCF maintains static loop structure while p63 supplies the cell-type-specific transcriptional activity β fits the unchanged CTCF ChIP signal and is broadly consistent with later mechanistic work showing CTCF N-terminusβcohesin interactions stabilize CTCF-anchored loops (2,756 WT vs ~98 mutant loops upon CTCF Y226A/F228A mutation) and that promoter-bound CTCF can have position-dependent non-architectural transcriptional roles .
What would falsify the model: CTCF ChIP-seq performed acutely after p63 loss showing broad binding loss, or loop resolution 3C/HiChIP demonstrating unchanged loop structure and activity across all 96 genes, would undermine the proposed two-tier (static CTCF, dynamic p63) architecture. Conversely, restoring wild-type p63 in patient keratinocytes should rescue accessibility at Ctr-OCRs β a rescue experiment not yet reported.
Confidence in the reported ATAC/ChIP observations is moderate-high (well-validated workflows, DESeq2/MACS2/HOMER/CHiCAGO pipelines, public data at GSE123711); confidence in the mechanistic cooperation model is moderate because it rests on correlative integration rather than direct perturbation of loops.
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