Article (Scientific journals)
wnt11f2 Zebrafish, an Animal Model for Development and New Insights in Bone Formation.
Caetano da Silva, Caroline; Ostertag, Agnes; Raman, Ratish et al.
2023In Zebrafish, 20 (1), p. 1 - 9
Peer reviewed
 

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Keywords :
Wnt pathways; development; high bone mass; wnt11f2; zebrafish; Zebrafish Proteins; Humans; Animals; Zebrafish Proteins/genetics; Zebrafish Proteins/metabolism; Signal Transduction; Models, Animal; Wnt Signaling Pathway; Cell Differentiation; Zebrafish; Osteogenesis; Animal Science and Zoology; Developmental Biology
Abstract :
[en] Wnt signaling is a key regulator of osteoblast differentiation and mineralization in humans and animals, mediated by the canonical Wnt/β-catenin and noncanonical signaling pathways. Both pathways are crucial in regulating osteoblastogenesis and bone formation. The zebrafish silberblick (slb) carries a mutation in wnt11f2, a gene that contributes to embryonic morphogenesis; however, its role in bone morphology is unknown. wnt11f2 was originally known as wnt11; it was recently reclassified to avoid confusion in comparative genetics and disease modeling. The goal of this review is to summarize the characterization of the wnt11f2 zebrafish mutant and to deliver some new insights concerning its role in skeletal development. In addition to the previously described defects in early development in this mutant as well as craniofacial dysmorphia, we show an increase in tissue mineral density in the heterozygous mutant that points to a possible role of wnt11f2 in high bone mass phenotypes.
Disciplines :
Biochemistry, biophysics & molecular biology
Author, co-author :
Caetano da Silva, Caroline;  INSERM U1132 and Université Paris-Cité, Reference Centre for Rare Bone Diseases, Hospital Lariboisière, Paris, France
Ostertag, Agnes;  INSERM U1132 and Université Paris-Cité, Reference Centre for Rare Bone Diseases, Hospital Lariboisière, Paris, France
Raman, Ratish ;  Université de Liège - ULiège > GIGA
Muller, Marc  ;  Université de Liège - ULiège > GIGA > GIGA I3 - Laboratory for Organogenesis and Regeneration
Cohen-Solal, Martine;  INSERM U1132 and Université Paris-Cité, Reference Centre for Rare Bone Diseases, Hospital Lariboisière, Paris, France
Collet, Corinne;  INSERM U1132 and Université Paris-Cité, Reference Centre for Rare Bone Diseases, Hospital Lariboisière, Paris, France ; UF de Génétique Moléculaire, Hôpital Robert Debré, APHP, Paris, France
Language :
English
Title :
wnt11f2 Zebrafish, an Animal Model for Development and New Insights in Bone Formation.
Publication date :
February 2023
Journal title :
Zebrafish
ISSN :
1545-8547
eISSN :
1557-8542
Publisher :
NLM (Medline), United States
Volume :
20
Issue :
1
Pages :
1 - 9
Peer reviewed :
Peer reviewed
Available on ORBi :
since 28 February 2023

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