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 language | English |
|---|---|
| Pages (from-to) | 236-245 |
| Number of pages | 10 |
| Journal | Experimental Cell Research |
| Volume | 361 |
| Issue number | 2 |
| DOIs | |
| Publication status | Published - Dec 15 2017 |
| Externally published | Yes |
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Keywords
- BCNT protein family
- Craniofacial development
- Morpholino
- Zebrafish
ASJC Scopus subject areas
- Cell Biology
Cite this
Functional analysis of the cfdp1 gene in zebrafish provides evidence for its crucial role in craniofacial development and osteogenesis. / Celauro, Emanuele; Carra, Silvia; Rodriguez, Adriana; Cotelli, Franco; Dimitri, Patrizio.
In: Experimental Cell Research, Vol. 361, No. 2, 15.12.2017, p. 236-245.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Functional analysis of the cfdp1 gene in zebrafish provides evidence for its crucial role in craniofacial development and osteogenesis
AU - Celauro, Emanuele
AU - Carra, Silvia
AU - Rodriguez, Adriana
AU - Cotelli, Franco
AU - Dimitri, Patrizio
PY - 2017/12/15
Y1 - 2017/12/15
N2 - 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.
AB - 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.
KW - BCNT protein family
KW - Craniofacial development
KW - Morpholino
KW - Zebrafish
UR - http://www.scopus.com/inward/record.url?scp=85032978205&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85032978205&partnerID=8YFLogxK
U2 - 10.1016/j.yexcr.2017.10.022
DO - 10.1016/j.yexcr.2017.10.022
M3 - Article
C2 - 29107067
AN - SCOPUS:85032978205
VL - 361
SP - 236
EP - 245
JO - Experimental Cell Research
JF - Experimental Cell Research
SN - 0014-4827
IS - 2
ER -