Neogenin-loss in neural crest cells results in persistent hyperplastic primary vitreous formation. J Mol Cell Biol 2020 Jan 22;12(1):17-31
Date
07/25/2019Pubmed ID
31336386Pubmed Central ID
PMC7053014DOI
10.1093/jmcb/mjz076Scopus ID
2-s2.0-85081130306 (requires institutional sign-in at Scopus site) 12 CitationsAbstract
Neogenin is a transmembrane receptor critical for multiple cellular processes, including neurogenesis, astrogliogenesis, endochondral bone formation, and iron homeostasis. Here we present evidence that loss of neogenin contributes to pathogenesis of persistent hyperplastic primary vitreous (PHPV) formation, a genetic disorder accounting for ~ 5% of blindness in the USA. Selective loss of neogenin in neural crest cells (as observed in Wnt1-Cre; Neof/f mice), but not neural stem cells (as observed in GFAP-Cre and Nestin-Cre; Neof/f mice), resulted in a dysregulation of neural crest cell migration or delamination, exhibiting features of PHPV-like pathology (e.g. elevated retrolental mass), unclosed retinal fissure, and microphthalmia. These results demonstrate an unrecognized function of neogenin in preventing PHPV pathogenesis, implicating neogenin regulation of neural crest cell delamination/migration and retinal fissure formation as potential underlying mechanisms of PHPV.
Author List
Lin S, Liu W, Chen CL, Sun D, Hu JX, Li L, Ye J, Mei L, Xiong WCAuthor
Ling Mei MD Associate Professor in the Medicine department at Medical College of WisconsinMESH terms used to index this publication - Major topics in bold
AnimalsCell Movement
Embryonic Development
Female
Male
Membrane Proteins
Mice
Mice, Inbred C57BL
Mice, Knockout
Microphthalmos
Neural Crest
Neural Stem Cells
Neurogenesis
Persistent Hyperplastic Primary Vitreous
Phenotype
Pregnancy
