Taking advantage of KRIT1−/− MEF cells, an established cellular model that allowed the identification of new molecules and mechanisms involved in CCM disease [7,8,9,38,72,76,77,78], we found that KRIT1 loss causes a significant decrease in total GSH and increase in GSSG levels, with a consequent deficit in the GSH/GSSG redox ratio and GSH-mediated antioxidant capacity. This evidence concerns the gene KRIT1 and cerebral cavernous malformation.