ZFIN ID: ZDB-PUB-171107-6
Functional analysis of the cfdp1 gene in zebrafish provides evidence for its crucial role in craniofacial development and osteogenesis
Celauro, E., Carra, S., Rodriguez, A., Cotelli, F., Dimitri, P.
Date: 2017
Source: Experimental cell research   361(2): 236-245 (Journal)
Registered Authors: Cotelli, Franco, Rodriguez-Mari, Adriana
Keywords: BCNT protein family, Craniofacial development, morpholino, zebrafish
MeSH Terms:
  • Amino Acid Sequence
  • Animals
  • Calcification, Physiologic/genetics
  • Cloning, Molecular
  • Embryo, Mammalian
  • Gene Expression Regulation, Developmental*
  • Morpholinos/genetics
  • Morpholinos/metabolism
  • Osteogenesis/genetics*
  • Phosphoproteins/antagonists & inhibitors
  • Phosphoproteins/genetics*
  • Phosphoproteins/metabolism
  • Sequence Alignment
  • Sequence Homology, Amino Acid
  • Signal Transduction
  • Skull/growth & development
  • Skull/metabolism*
  • Zebrafish/embryology
  • Zebrafish/genetics*
  • Zebrafish/metabolism
  • Zebrafish Proteins/antagonists & inhibitors
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/metabolism
PubMed: 29107067 Full text @ Exp. Cell Res.
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.