KDM2A and Neurodevelopmental delay: Here, we describe the overlapping phenotype of 18 individuals with de novo variants in KDM2A. We combined the power of genetically modified Drosophila melanogaster, which our lab has successfully used to in the delineation of other novel NDDs10,11,12 with human cell culture, as well as methylome data based on blood-derived DNA from affected individuals, to establish the gene-disease association of de novo variants in KDM2A and a syndromic NDD.