Mutation of foxl1 Results in Reduced Cartilage Markers in a Zebrafish Model of Otosclerosis.

ABSTRACT: Bone diseases such as otosclerosis (conductive hearing loss) and osteoporosis (low bone mineral density) can result from the abnormal expression of genes that regulate cartilage and bone development. The forkhead box transcription factor FOXL1 has been identified as the causative gene in a family with autosomal dominant otosclerosis and has been reported as a candidate gene in GWAS meta-analyses for osteoporosis. This potentially indicates a novel role for foxl1 in chondrogenesis, osteogenesis, and bone remodelling. We created a foxl1 mutant zebrafish strain as a model for otosclerosis and osteoporosis and examined jaw bones that are homologous to the mammalian middle ear bones, and mineralization of the axial skeleton. We demonstrate that foxl1 regulates the ex

SUBMITTER: Hawkey-Noble A 

PROVIDER: S-EPMC9319681 | biostudies-literature | 2022 Jun

REPOSITORIES: biostudies-literature