show Abstracthide AbstractTo understand the effect of SET protein accumulation on genomic function we used different accumulating models, cells and in vivo, that we analyzed with different techinques up to the single level. SET accumulation is known to be caused by the accumulation of its direct interactor SETBP1, when mutated, the causative gene of Schinzel Giedion syndrome. SET has been shown to be able to interfere with histones acetylation, thus we wanted to investigated the functional effect on the genome function and cell fate commitment. Overall design: Genomic analysis of human cells (IPSCs, NPCs, Neurons), Zebrafish brain embryo, mouse embryonic and post natal cortex, to understand the effect of SET accumulation on genome function