CHD7 and SOX2 act in a common gene regulatory network during mammalian semicircular canal and cochlear development.

ABSTRACT: Inner ear morphogenesis requires tightly regulated epigenetic and transcriptional control of gene expression. CHD7, an ATP-dependent chromodomain helicase DNA-binding protein, and SOX2, an SRY-related HMG box pioneer transcription factor, are known to contribute to vestibular and auditory system development, but their genetic interactions in the ear have not been explored. Here, we analyzed inner ear development and the transcriptional regulatory landscapes in mice with variable dosages of Chd7 and/or Sox2. We show that combined haploinsufficiency for Chd7 and Sox2 results in reduced otic cell proliferation, severe malformations of semicircular canals, and shortened cochleae with ectopic hair cells. Examination of mice with conditional, inducible Chd7 los

SUBMITTER: Gao J 

PROVIDER: S-EPMC10927591 | biostudies-literature | 2024 Mar

REPOSITORIES: biostudies-literature