A mutagenesis-derived Lrp5 mouse mutant with abnormal retinal vasculature and low bone mineral density.

ABSTRACT:

Purpose

Familial exudative vitreoretinopathy (FEVR) is caused by mutations in the genes encoding low-density lipoprotein receptor-related protein (LRP5) or its interacting partners, namely frizzled class receptor 4 (FZD4) and norrin cystine knot growth factor (NDP). Mouse models for Lrp5, Fzd4, and Ndp have proven to be important for understanding the retinal pathophysiology underlying FEVR and systemic abnormalities related to defective Wnt signaling. Here, we report a new mouse mutant, tvrm111B, which was identified by electroretinogram (ERG) screening of mice generated in the Jackson Laboratory Translational Vision Research Models (TVRM) mutagenesis program.

Methods

ERGs were used to examine outer retinal physiology. The retinal vasculature was

SUBMITTER: Charette JR 

PROVIDER: S-EPMC5360454 | biostudies-literature | 2017

REPOSITORIES: biostudies-literature