Article
Posterior malformations in Dact1 mutant mice arise through misregulated Vangl2 at the primitive streak.
Nature genetics - 1 Sept 2009
Suriben Rowena, Kivimäe Saul, Fisher Daniel A C, Moon Randall T, Cheyette Benjamin N R
Abstract excerpt
Mice homozygous for mutations in Dact1 (also called Dapper or Frodo) phenocopy human malformations involving the spine, genitourinary system and distal digestive tract. We traced this phenotype to disrupted germ-layer morphogenesis at the primitive streak. Notably, heterozygous mutation of Vangl2, a transmembrane component of the planar cell polarity (PCP) pathway, rescued recessive Dact1 phenotypes, whereas loss...
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