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J Mol Cell Biol. 2019 Jul 23. pii: mjz076. doi: 10.1093/jmcb/mjz076. [Epub ahead of print]

Neogenin-loss in neural crest cells results in persistent hyperplastic primary vitreous formation.

Lin S1,2, Liu W1,2, Chen CL1,2, Sun D1,3, Hu JX1, Li L1, Ye J2, Mei L1,3, Xiong WC1,3.

Author information

1
Department of Neuroscience & Regenerative Medicine and Department of Neurology, Augusta University, Augusta, GA, 30912, USA.
2
Department of Ophthalmology, Daping Hospital, Army Medical Center of PLA, Chongqing, China.
3
Department of Neurosciences, School of Medicine, Case Western Reserve University, Cleveland, OH 44106.

Abstract

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 formation (PHPV), a genetic disorder accounting for ~5% of blindness in the USA (United States of America). 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.

PMID:
31336386
DOI:
10.1093/jmcb/mjz076

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