First Author | Kerr CL | Year | 2014 |
Journal | Dev Dyn | Volume | 243 |
Issue | 10 | Pages | 1298-309 |
PubMed ID | 24753151 | Mgi Jnum | J:214344 |
Mgi Id | MGI:5588786 | Doi | 10.1002/dvdy.24141 |
Citation | Kerr CL, et al. (2014) AP-2alpha is required after lens vesicle formation to maintain lens integrity. Dev Dyn 243(10):1298-309 |
abstractText | BACKGROUND: Transcription factors are critical in regulating lens development. The AP-2 family of transcription factors functions in differentiation, cell growth and apoptosis, and in lens and eye development. AP-2alpha, in particular, is important in early lens development, and when conditionally deleted at the placode stage defective separation of the lens vesicle from the surface ectoderm results. AP-2alpha's role during later stages of lens development is unknown. To address this, the MLR10-Cre transgene was used to delete AP-2alpha from the lens epithelium beginning at embryonic day (E) 10.5. RESULTS: The loss of AP-2alpha after lens vesicle separation resulted in morphological defects beginning at E18.5. By P4, a small highly vacuolated lens with a multilayered epithelium was evident in the MLR10-AP-2alpha mutants. Epithelial cells appeared elongated and expressed fiber cell specific betaB1 and gamma-crystallins. Epithelial cell polarity and lens cell adhesion was disrupted and accompanied by the misexpression of ZO-1, N-Cadherin, and beta-catenin. Cell death was observed in the mutant lens epithelium between postnatal day (P) 14 and P30, and correlated with altered arrangements of cells within the epithelium. CONCLUSIONS: Our findings demonstrate that AP-2alpha continues to be required after lens vesicle separation to maintain a normal lens epithelial cell phenotype and overall lens integrity and to ensure correct fiber cell differentiation. Developmental Dynamics 243:1298-1309, 2014. (c) 2014 Wiley Periodicals, Inc. |