Back to Search Start Over

Cited2 is required for the proper formation of the hyaloid vasculature and for lens morphogenesis

Authors :
Dirk A. Kleinjan
Yong Qiu Doughman
David C. Beebe
Sally L. Dunwoodie
Michiko Watanabe
Ales Cvekl
Shi Gu
Shin Ichi Aota
Yu Chung Yang
Randall S. Johnson
Yu Chen
Andrew Jarrell
Veronica van Heyningen
Source :
Chen, Y, Doughman, Y, Gu, S, Jarrell, A, Aota, S, Cvekl, A, Watanabe, M, Dunwoodie, S L, Johnson, R S, van Heyningen, V, Kleinjan, D A, Beebe, D C & Yang, Y-C 2008, ' Cited2 is required for the proper formation of the hyaloid vasculature and for lens morphogenesis ', Development, vol. 135, no. 17, pp. 2939-48 . https://doi.org/10.1242/dev.021097
Publication Year :
2008

Abstract

Cited2 is a transcriptional modulator with pivotal roles in different biological processes. Cited2-deficient mouse embryos manifested two major defects in the developing eye. An abnormal corneal-lenticular stalk was characteristic of Cited2-/- developing eyes, a feature reminiscent of Peters' anomaly, which can be rescued by increased Pax6 gene dosage in Cited2-/- embryonic eyes. In addition, the hyaloid vascular system showed hyaloid hypercellularity consisting of aberrant vasculature, which might be correlated with increased VEGF expression in the lens. Deletion of Hif1a (which encodes HIF-1α) in Cited2-/- lens specifically eliminated the excessive accumulation of cellular mass and aberrant vasculature in the developing vitreous without affecting the corneal-lenticular stalk phenotype. These in vivo data demonstrate for the first time dual functions for Cited2:one upstream of, or together with, Pax6 in lens morphogenesis; and another in the normal formation of the hyaloid vasculature through its negative modulation of HIF-1 signaling. Taken together, our study provides novel mechanistic revelation for lens morphogenesis and hyaloid vasculature formation and hence might offer new insights into the etiology of Peters'anomaly and ocular hypervascularity.

Details

Language :
English
Database :
OpenAIRE
Journal :
Chen, Y, Doughman, Y, Gu, S, Jarrell, A, Aota, S, Cvekl, A, Watanabe, M, Dunwoodie, S L, Johnson, R S, van Heyningen, V, Kleinjan, D A, Beebe, D C & Yang, Y-C 2008, ' Cited2 is required for the proper formation of the hyaloid vasculature and for lens morphogenesis ', Development, vol. 135, no. 17, pp. 2939-48 . https://doi.org/10.1242/dev.021097
Accession number :
edsair.doi.dedup.....935eae7a75ce39643b4afebffcb58bbc
Full Text :
https://doi.org/10.1242/dev.021097