Disruption of the Fbxw8 gene results in pre- and postnatal growth retardation in mice.

ABSTRACT: CUL7 binds to SKP1, RBX1, and FBXW8 to form a cullin-RING ligase, or an SKP1-cullin-F box protein complex. The targeted disruption of the Cul7 gene in mice results in significant reduction in embryo size and neonatal lethality. In humans, CUL7 was found to be mutated in the 3-M dwarfism syndrome characterized by severe pre- and postnatal growth retardation, indicating that CUL7 is closely associated with human and mouse growth. We generated mice lacking Fbxw8 by gene trapping. Similar to Cul7(-/-) animals, Fbxw8(-/-) embryos and placentas were smaller than wild-type and heterozygous littermates and placentas. Approximately 30% of the expected number of Fbxw8(-/-) mice survived birth, but these mice remained smaller than their wild-type and heterozygous littermates throughout postnatal deve

SUBMITTER: Tsutsumi T 

PROVIDER: S-EPMC2223430 | biostudies-literature | 2008 Jan

REPOSITORIES: biostudies-literature