ZFIN ID: ZDB-FIG-190702-12
Lahrouchi et al., 2019 - Homozygous frameshift mutations in FAT1 cause a syndrome characterized by colobomatous-microphthalmia, ptosis, nephropathy and syndactyly. Nature communications   10:1180 Full text @ Nat. Commun.
Knockdown Reagent:
Observed In:
Stage: Prim-5

Fig. 5

Zebrafish embryos with homozygous alleles of truncated fat1a display coloboma. CRISPR/Cas9-mediated introduction of frame-shift mutations in FAT1 C-terminal resulted in optic fissure closure defects (a and e, scale bar is 0.1 mm). A higher magnification of eye depicting fused margins in WT and unfused margins in homozygous mutant (*, b and f, scale bar is 0.05 mm). Sagittal sections of zebrafish embryos (24–30 hpf) followed by toludene blue staining showing organization of the optic cup (c and g, scale bar is 50 µm). Higher magnification of the optic cup shows morphology of optic fissure margins in WT and homozygous mutant (dh, scale bar is 20 µm)

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
WT + CRISPR1-fat1a standard conditions Prim-5 optic cup retinal pigmented epithelium disorganized, abnormal
Prim-5 optic fissure anatomical margin disorganized, abnormal
Prim-5 optic fissure unfused from optic fissure, abnormal
This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ Nat. Commun.