Fgfr3 Is a Positive Regulator of Osteoblast Expansion and Differentiation During Zebrafish Skull Vault Development
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Fgfr3 Is a Positive Regulator of Osteoblast Expansion and Differentiation During Zebrafish Skull Vault Development. / 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.
In: J BONE MINER RES, Vol. 35, No. 9, 09.2020, p. 1782-1797.Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review
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TY - JOUR
T1 - Fgfr3 Is a Positive Regulator of Osteoblast Expansion and Differentiation During Zebrafish Skull Vault Development
AU - Dambroise, Emilie
AU - Ktorza, Ivan
AU - Brombin, Alessandro
AU - Abdessalem, Ghaith
AU - Edouard, Joanne
AU - Luka, Marine
AU - Fiedler, Imke
AU - Binder, Olivia
AU - Pelle, Olivier
AU - Patton, E Elizabeth
AU - Busse, Björn
AU - Menager, Mickaël
AU - Sohm, Frederic
AU - Legeai-Mallet, Laurence
N1 - © 2020 American Society for Bone and Mineral Research.
PY - 2020/9
Y1 - 2020/9
N2 - 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.
AB - 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.
U2 - 10.1002/jbmr.4042
DO - 10.1002/jbmr.4042
M3 - SCORING: Journal article
C2 - 32379366
VL - 35
SP - 1782
EP - 1797
JO - J BONE MINER RES
JF - J BONE MINER RES
SN - 0884-0431
IS - 9
ER -