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Craniosynostosis in <TOGGLE>Twist</TOGGLE> heterozygous mice: A model for Saethre-Chotzen syndrome

Authors :
Carver, Ethan A.
Oram, Kathleen F.
Gridley, Thomas
Source :
The Anatomical Record Part A: Discoveries in Molecular, Cellular, and Evolutionary Biology; October 2002, Vol. 268 Issue: 2 p90-92, 3p
Publication Year :
2002

Abstract

Saethre-Chotzen syndrome is a common autosomal dominant form of craniosynostosis, the premature fusion of the sutures of the calvarial bones of the skull. Most Saethre-Chotzen syndrome cases are caused by haploinsufficiency for the TWIST gene. Mice heterozygous for a null mutation of the Twist gene replicate certain features of Saethre-Chotzen syndrome, but have not been reported to exhibit craniosynostosis. We demonstrate that Twist heterozygous mice exhibit fusions of the coronal suture and other cranial suture abnormalities, indicating that Twist heterozygous mice constitute a better animal model for Saethre-Chotzen syndrome than was previously appreciated. Anat Rec 268:90–92, 2002. &#169; 2002 Wiley-Liss, Inc.

Details

Language :
English
ISSN :
15524884 and 15524892
Volume :
268
Issue :
2
Database :
Supplemental Index
Journal :
The Anatomical Record Part A: Discoveries in Molecular, Cellular, and Evolutionary Biology
Publication Type :
Periodical
Accession number :
ejs3783172
Full Text :
https://doi.org/10.1002/ar.10124