To expand our knowledge on the role of CCM1 in non-endothelial cell types and the importance of exogenous triggers in CCM formation, we combined our recently established isogenic CCM1 knockout induced pluripotent stem cell (CCM1−/− iPSC) model [20], the differentiation of iPSCs into endothelial-like cells (hereafter referred to as ECs), and RNA sequencing analyses. The gene discussed is KRIT1; the disease is cerebral cavernous malformation.