FGFR3 is a positive regulator of osteoblast expansion and differentiation during zebrafish skull vault development

ABSTRACT: In order to study FGFR3’s role in cranial vault development, we generated the first fgfr3 loss-of-function zebrafish (fgfr3lof1/lof1). The mutant fish exhibited major changes in the craniofacial skeleton, with a lack of sutures, abnormal frontal and parietal bones, and the presence of ectopic bones. In order to define cellular and molecular processes responsible for this phenotype, we performed single-cell RNA sequencing of cranial vault cells from fgfr3lof1/lof1 and fgfr3+/+ zebrafish. Our data allow us to define transcriptomic profile of each osteogenic subpopulation involved in cranial vault formation and we revealed a defect in osteoblast differentiation associated with changes in the extracellular matrix. These findings demonstrate that fgfr3 is a positive regulator of osteogenesis and we conclude that changes in the extracellular matrix within growing bone impair cell-cell communication, mineralization, and new osteoblast recruitment.

ORGANISM(S): Danio rerio

PROVIDER: GSE144944 | GEO | 2020/05/18

REPOSITORIES: GEO