Functional analysis of the cfdp1 gene in zebrafish provides evidence for its crucial role in craniofacial development and osteogenesis
The CFDP1 proteins have been linked to craniofacial development and osteogenesis in vertebrates, though
specific human syndromes have not yet been identified. Alterations of craniofacial development represent the
main cause of infant disability and mortality in humans. For this reason, it is crucial to understand the cellular
functions and mechanism of action of the CFDP1 protein in model vertebrate organisms. Using a combination of
genomic, molecular and cell biology approaches, we have performed a functional analysis of the cfdp1 gene and
its encoded protein, zCFDP1, in the zebrafish model system. We found that zCFDP1 is present in the zygote, is
rapidly produced after MTZ transition and is highly abundant in the head structures. Depletion of zCFDP1,
induced by an ATG-blocking morpholino, produces considerable defects in craniofacial structures and bone
mineralization. Together, our results show that zCFDP1 is an essential protein required for proper development
and provide the first experimental evidence showing that in vertebrates it actively participates to the morphogenesis
of craniofacial territories.