Show simple item record

AuthorCaceres, Lucia
AuthorPrykhozhij, Sergey V
AuthorCairns, Elizabeth
AuthorGjerde, Harald
AuthorDuff, Nicole Marie
AuthorCollett, Keon
AuthorNgo, Mike
AuthorNasrallah, Gheyath K
AuthorMcMaster, Christopher R
AuthorLitvak, Matthew
AuthorRobitaille, Johane M
AuthorBerman, Jason N
Available date2019-10-31T08:55:18Z
Publication Date2019-09-01
Publication NameDevelopmental Dynamics
Identifierhttp://dx.doi.org/10.1002/dvdy.117
CitationLucia Caceres. et. al. "Frizzled 4 regulates ventral blood vessel remodeling in the zebrafish retina" Developmental Dynamics 2019, DOI: 10.1002/dvdy.117
URIhttp://hdl.handle.net/10576/12193
AbstractFamilial exudative vitreoretinopathy (FEVR) is a rare congenital disorder characterized by a lack of blood vessel growth to the periphery of the retina with secondary fibrovascular proliferation at the vascular-avascular junction. These structurally abnormal vessels cause leakage and hemorrhage, while the fibroproliferative scarring results in retinal dragging, detachment and blindness. Mutations in the FZD4 gene represent one of the most common causes of FEVR. A loss of function mutation resulting from a 10-nucleotide insertion into exon 1 of the zebrafish fzd4 gene was generated using transcription activator-like effector nucleases (TALENs). Structural and functional integrity of the retinal vasculature was examined by fluorescent microscopy and optokinetic responses. Zebrafish retinal vasculature is asymmetrically distributed along the dorsoventral axis, with active vascular remodeling on the ventral surface of the retina throughout development. fzd4 mutants exhibit disorganized ventral retinal vasculature with discernable tubular fusion by week 8 of development. Furthermore, fzd4 mutants have impaired optokinetic responses requiring increased illumination. We have generated a visually impaired zebrafish FEVR model exhibiting abnormal retinal vasculature. These fish provide a tractable system for studying vascular biology in retinovascular disorders, and demonstrate the feasibility of using zebrafish for evaluating future FEVR genes identified in humans. This article is protected by copyright. All rights reserved.
Languageen
PublisherWiley
SubjectFEVR
Retinal vasculature
optokinetic response
pericytes
TitleFrizzled 4 regulates ventral blood vessel remodeling in the zebrafish retina
TypeArticle


Files in this item

FilesSizeFormatView

There are no files associated with this item.

This item appears in the following Collection(s)

Show simple item record