Functional analysis of the cfdp1 gene in zebrafish provides evidence for its crucial role in craniofacial development and osteogenesis

Emanuele Celauro, Silvia Carra, Adriana Rodriguez, Franco Cotelli, Patrizio Dimitri

Research output: Contribution to journalArticlepeer-review

Abstract

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.

Original languageEnglish
Pages (from-to)236-245
Number of pages10
JournalExperimental Cell Research
Volume361
Issue number2
DOIs
Publication statusPublished - Dec 15 2017
Externally publishedYes

Keywords

  • BCNT protein family
  • Craniofacial development
  • Morpholino
  • Zebrafish

ASJC Scopus subject areas

  • Cell Biology

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