amer1 Regulates Zebrafish Craniofacial Development by Interacting with the Wnt/β-Catenin Pathway.

ABSTRACT: Microtia-atresia is a rare type of congenital craniofacial malformation causing severe damage to the appearance and hearing ability of affected individuals. The genetic factors associated with microtia-atresia have not yet been determined. The AMER1 gene has been identified as potentially pathogenic for microtia-atresia in two twin families. An amer1 mosaic knockdown zebrafish model was constructed using CRISPR/Cas9. The phenotype and the development process of cranial neural crest cells of the knockdown zebrafish were examined. Components of the Wnt/β-catenin pathway were examined by qPCR, Western blotting, and immunofluorescence assay. IWR-1-endo, a reversible inhibitor of the Wnt/β-catenin pathway, was applied to rescue the abnormal phenotype. The present study showed that

SUBMITTER: Sun L 

PROVIDER: S-EPMC10815499 | biostudies-literature | 2024 Jan

REPOSITORIES: biostudies-literature