Phenotype Image Detail

Image

Caption

A) E12.5 embryos of Fuz⁺/Fuz^{Gt(OSTGST001398)Lex} (Fuz^+/-) and Fuz^{Gt(OSTGST001398)Lex}/Fuz^{Gt(OSTGST001398)Lex} (Fuz^-/-) mice. The overall structure of the ventral craniofacial region is not severely affected at this stage in the homozygous embryos, due to the low expression of Fuz. However, the dorsal structures including the choroid plexus (CP) differentiating from the roof of fourth ventricle, the choroid plexus extending into the lateral ventricle (CPL), and the optic recess of the diencephalon (OR) are missing in the homozygous mice. The corpus striatum mediale (STM), and the cochlea (CO) are displaced in the homozygous mice. Higher magnification of the mandible is shown in the bottom panels and reveals normal Meckel's cartilage formation at this stage. B) At E14.5 the craniofacial defects of the homozygous mice are severe. The tongue muscles are fused with the mandible, the pituitary (PI) is missing and the trigeminal nerve (TG) is seen in its place and the OR is displaced, corresponding to anophthalmia in the mutant. The higher magnification sections showed a lack of upper (UI) and lower incisors (LI), no tongue (TE) and expanded Meckel's cartilage (MC) in the dorsal-ventral axis instead of anterior-posterior axis. The palate is now a piece of displaced palatal tissue (PLT). C) E16.5 coronal sections reveal a cleft palate in the homozygous mice. The palate tissue is displaced to the lateral portions of the oral cavity and the secondary palate is not fused, due to the dorsal-ventral and medial-lateral expansion of Meckel's cartilage. The molars (ML) are shown and appear to be grossly normal in the mutant mice. The plane of section is depicted by the dotted line through the mouse drawing.

Copyright

This image is from Zhang Z, PLoS One 2011;6(9):e24608, and is displayed under the terms of the Creative Commons Attribution 4.0 International License. J:177881

Associated Alleles

Symbol	Name
Fuz^Gt(OSTGST001398)Lex	fuzzy planar cell polarity protein; gene trap OSTGST001398, Lexicon Genetics

Associated Genotypes

Allelic Composition	Genetic Background
Fuz^Gt(OSTGST001398)Lex/Fuz^Gt(OSTGST001398)Lex	involves: 129S5/SvEvBrd * C57BL/6