Targeting fibroblast growth factor receptors causes severe craniofacial malformations in zebrafish larvae.

ABSTRACT:

Background and objective

A key pathway controlling skeletal development is fibroblast growth factor (FGF) and FGF receptor (FGFR) signaling. Major regulatory functions of FGF signaling are chondrogenesis, endochondral and intramembranous bone development. In this study we focus on fgfr2, as mutations in this gene are found in patients with craniofacial malformations. The high degree of conservation between FGF signaling of human and zebrafish (Danio rerio) tempted us to investigate effects of the mutated fgfr2 ^sa10729 allele in zebrafish on cartilage and bone formation.

Methods

We stained cartilage and bone in 5 days post fertilization (dpf) zebrafish larvae and compared mutants with wildtypes. We also determined the expression of genes relate

SUBMITTER: Gebuijs L 

PROVIDER: S-EPMC9700454 | biostudies-literature | 2022

REPOSITORIES: biostudies-literature