Autonomous and non-cell autonomous role of cilia in structural birth defects in mice
Fig 6
Early tamoxifen deletion of Ift140 with CAGGCre-ER recapitulates the Ift140null1/null1 phenotype.
Ift140flox/+, CAGGCre-ER+ (Control) (A–C) and Ift140flox/null1, CAGGCre-ER+ experimental (D–F) embryos were treated with tamoxifen at E5.5 and harvested at E12.5. The experimental embryos had extensive developmental abnormalities and showed developmental delay (A, D). The experimentals recapitulated the laterality defects seen in Ift140null1/null1 embryos as characterized by reversed heart tube looping and the morphological right ventricle appearing on the embryo’s left side (B vs. E). Similar to the Ift140null1/null1 embryos, tamoxifen-driven deletion of Ift140 using CAGGCre-ER at E5.5 also caused atrial septal defects and outflow track septation defects (PTA) (C vs. F). However, head fold closure defects and exencephaly were not observed under these conditions. fb: forebrain; mb: midbrain; fl: forelimb; hl: hindlimb; e: eye; A: aorta; P: pulmonary trunk; LV: left ventricle; RV: right ventricle; LA: left atria; RA: right atria; PTA: persistent truncus arteriosus. Scales bars: A, D = 0.5 mm, B, C, E, F = 0.25 mm.