Article
Genetic background dependent modifiers of craniosynostosis severity.
Journal of structural biology - 1 Dec 2020
Dudakovic Amel, Nam Hwa Kyung, Wijnen Andre J van, Hatch Nan E
Abstract excerpt
Craniosynostosis severity varies in patients with identical genetic mutations. To understand causes of this phenotypic variation, we backcrossed the FGFR2+/C342Y mouse model of Crouzon syndrome onto congenic C57BL/6 and BALB/c backgrounds. Coronal suture fusion was observed in C57BL/6 (88% incidence, p < .001 between genotypes) but not in BALB/c FGFR2+/C342Y mutant mice at 3 weeks after birth, establishing that...
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