Multi-omic analyses identify molecular targets of Chd7 that mediate CHARGE syndrome model phenotypes.

ABSTRACT: CHARGE syndrome is a developmental disorder that affects 1 in 10,000 births, and patients exhibit both physical and behavioral characteristics. De novo mutations in CHD7 (chromodomain helicase DNA binding protein 7) cause 67% of CHARGE syndrome cases. CHD7 is a DNA-binding chromatin remodeler with thousands of predicted binding sites in the genome, making it challenging to define molecular pathways linking loss of CHD7 to CHARGE phenotypes. To address this problem, here we used a previously characterized zebrafish CHARGE model to generate transcriptomic and proteomic datasets from larval zebrafish head tissue at two developmental time points. By integrating these datasets with differential expression, pathway, and upstream regulator analyses, we identified multiple con

SUBMITTER: Hancock MB 

PROVIDER: S-EPMC12324285 | biostudies-literature | 2025 Jul

REPOSITORIES: biostudies-literature