Fig. 1

Strategy for identifying neurodevelopmental phenotypes in 16p11.2 fly homologs. a We identified 14 homologs of 16p11.2 deletion genes in Drosophila melanogaster, and b evaluated global, neurodevelopmental, and cellular phenotypes. We also performed transcriptome sequencing and assessed changes in expression of biologically significant genes. c Next, we identified modifiers of the one-hit eye phenotype for select homologs using two-hit interaction models. A subset of these interactions were further assessed for cellular phenotypes in the two-hit knockdown eyes. We incorporated all fly interactions into a human brain-specific genetic interaction network