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- Title
Fgfr3 Is a Positive Regulator of Osteoblast Expansion and Differentiation During Zebrafish Skull Vault Development.
- Authors
Dambroise, Emilie; Ktorza, Ivan; Brombin, Alessandro; Abdessalem, Ghaith; Edouard, Joanne; Luka, Marine; Fiedler, Imke; Binder, Olivia; Pelle, Olivier; Patton, E Elizabeth; Busse, Björn; Menager, Mickaël; Sohm, Frederic; Legeai‐Mallet, Laurence
- Abstract
Gain or loss‐of‐function mutations in fibroblast growth factor receptor 3 (FGFR3) result in cranial vault defects highlighting the protein's role in membranous ossification. Zebrafish express high levels of fgfr3 during skull development; in order to study FGFR3's role in cranial vault development, we generated the first fgfr3 loss‐of‐function zebrafish (fgfr3lof/lof). The mutant fish exhibited major changes in the craniofacial skeleton, with a lack of sutures, abnormal frontal and parietal bones, and the presence of ectopic bones. Integrated analyses (in vivo imaging and single‐cell RNA sequencing of the osteoblast lineage) of zebrafish fgfr3lof/lof revealed a delay in osteoblast expansion and differentiation, together with changes in the extracellular matrix. These findings demonstrate that fgfr3 is a positive regulator of osteogenesis. We conclude that changes in the extracellular matrix within growing bone might impair cell–cell communication, mineralization, and new osteoblast recruitment. © 2020 American Society for Bone and Mineral Research.
- Subjects
ZEBRA danio; FIBROBLAST growth factor receptors; BRACHYDANIO; SKULL; BONES; EXTRACELLULAR matrix
- Publication
Journal of Bone & Mineral Research, 2020, Vol 35, Issue 9, p1782
- ISSN
0884-0431
- Publication type
Article
- DOI
10.1002/jbmr.4042