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Altered bone growth dynamics prefigure craniosynostosis in a zebrafish model of Saethre-Chotzen syndrome.
Elife 2018; 7E

Abstract

Cranial sutures separate the skull bones and house stem cells for bone growth and repair. In Saethre-Chotzen syndrome, mutations in TCF12 or TWIST1 ablate a specific suture, the coronal. This suture forms at a neural-crest/mesoderm interface in mammals and a mesoderm/mesoderm interface in zebrafish. Despite this difference, we show that combinatorial loss of TCF12 and TWIST1 homologs in zebrafish also results in specific loss of the coronal suture. Sequential bone staining reveals an initial, directional acceleration of bone production in the mutant skull, with subsequent localized stalling of bone growth prefiguring coronal suture loss. Mouse genetics further reveal requirements for Twist1 and Tcf12 in both the frontal and parietal bones for suture patency, and to maintain putative progenitors in the coronal region. These findings reveal conservation of coronal suture formation despite evolutionary shifts in embryonic origins, and suggest that the coronal suture might be especially susceptible to imbalances in progenitor maintenance and osteoblast differentiation.

Authors+Show Affiliations

Department of Stem Cell Biology and Regenerative Medicine, University of Southern California, Los Angeles, United States. Department of Biochemistry, Keck School of Medicine, University of Southern California, Los Angeles, United States.Department of Biochemistry, Keck School of Medicine, University of Southern California, Los Angeles, United States.Department of Stem Cell Biology and Regenerative Medicine, University of Southern California, Los Angeles, United States.Department of Biochemistry, Keck School of Medicine, University of Southern California, Los Angeles, United States.Department of Biochemistry, Keck School of Medicine, University of Southern California, Los Angeles, United States.Department of Stem Cell Biology and Regenerative Medicine, University of Southern California, Los Angeles, United States.

Pub Type(s)

Journal Article
Research Support, N.I.H., Extramural
Research Support, Non-U.S. Gov't

Language

eng

PubMed ID

30375332

Citation

Teng, Camilla S., et al. "Altered Bone Growth Dynamics Prefigure Craniosynostosis in a Zebrafish Model of Saethre-Chotzen Syndrome." ELife, vol. 7, 2018.
Teng CS, Ting MC, Farmer DT, et al. Altered bone growth dynamics prefigure craniosynostosis in a zebrafish model of Saethre-Chotzen syndrome. Elife. 2018;7.
Teng, C. S., Ting, M. C., Farmer, D. T., Brockop, M., Maxson, R. E., & Crump, J. G. (2018). Altered bone growth dynamics prefigure craniosynostosis in a zebrafish model of Saethre-Chotzen syndrome. ELife, 7, doi:10.7554/eLife.37024.
Teng CS, et al. Altered Bone Growth Dynamics Prefigure Craniosynostosis in a Zebrafish Model of Saethre-Chotzen Syndrome. Elife. 2018 10 25;7 PubMed PMID: 30375332.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Altered bone growth dynamics prefigure craniosynostosis in a zebrafish model of Saethre-Chotzen syndrome. AU - Teng,Camilla S, AU - Ting,Man-Chun, AU - Farmer,D'Juan T, AU - Brockop,Mia, AU - Maxson,Robert E, AU - Crump,J Gage, Y1 - 2018/10/25/ PY - 2018/03/27/received PY - 2018/10/14/accepted PY - 2018/10/31/entrez PY - 2018/10/31/pubmed PY - 2019/2/9/medline KW - Saethre-Chotzen Syndrome KW - Tcf12 KW - Twist1 KW - coronal suture KW - craniosynostosis KW - developmental biology KW - mouse KW - regenerative medicine KW - skull bones KW - stem cells KW - zebrafish JF - eLife JO - Elife VL - 7 N2 - Cranial sutures separate the skull bones and house stem cells for bone growth and repair. In Saethre-Chotzen syndrome, mutations in TCF12 or TWIST1 ablate a specific suture, the coronal. This suture forms at a neural-crest/mesoderm interface in mammals and a mesoderm/mesoderm interface in zebrafish. Despite this difference, we show that combinatorial loss of TCF12 and TWIST1 homologs in zebrafish also results in specific loss of the coronal suture. Sequential bone staining reveals an initial, directional acceleration of bone production in the mutant skull, with subsequent localized stalling of bone growth prefiguring coronal suture loss. Mouse genetics further reveal requirements for Twist1 and Tcf12 in both the frontal and parietal bones for suture patency, and to maintain putative progenitors in the coronal region. These findings reveal conservation of coronal suture formation despite evolutionary shifts in embryonic origins, and suggest that the coronal suture might be especially susceptible to imbalances in progenitor maintenance and osteoblast differentiation. SN - 2050-084X UR - https://www.unboundmedicine.com/medline/citation/30375332/Altered_bone_growth_dynamics_prefigure_craniosynostosis_in_a_zebrafish_model_of_Saethre_Chotzen_syndrome_ L2 - https://doi.org/10.7554/eLife.37024 DB - PRIME DP - Unbound Medicine ER -