frizzled 5 mutant zebrafish are genetically sensitised to developing microphthalmia and coloboma
Frizzled class receptor 5 (FZD5) is a Wnt receptor expressed in the developing eye, and individuals with variants in FZD5 exhibit microphthalmia/coloboma, supporting a role for this receptor in human eye formation. Eye development can occur normally in the absence of Fzd5 in zebrafish, but mutants are sensitised to developing eye malformations. By exploiting the sensitised nature of the fzd5 mutants, we further identified angio-associated migratory cell protein (aamp) as a novel gene involved in eye morphogenesis. Overall, our study confirms the importance of considering multiple genetic contributions when searching for the molecular aetiology of ocular malformations in humans.
Read this collaborative work and admire the eye catching cover image
https://journals.biologists.com/dmm/article/18/6/dmm052284/368227/frizzled-5-mutant-zebrafish-are-genetically
Cover: Confocal image showing the eye of a maternal-zygotic frizzled 5 mutant zebrafish 5 days post fertilisation stained to reveal tissue architecture with phalloidin-FITC (actomyosin cortical cytoskeleton, grey) and Sytox-Orange (nuclei, blue). Phalloidin staining allows visualisation of the outlines of the neurons in the retina and the plexiform layers.