... profound cardiac and trunk muscle dystrophy, along with lens abnormalities, caused by abrupt terminal differentiation of cardiomyocyte, myoblast and lens fiber cells. Loss of ddx39ab hindered splicing of mRNAs encoding epigenetic regulatory factors, including members of the KMT2 gene family, leading...

... of proliferative cells exists in mouse lens epithelium: implications for lens maintenance . Invest. Ophthalmol. Vis. Sci.   47 , 2997 - 3003 . 10.1167/iovs.06-0130 We conducted time-lapse scanning of the equatorial region in N-cadherin morphant lenses combined with Tg(h2afva:GFP; EF1α:mCherry-zGem...

... with the complete detachment of the lens vesicle. The rescue of lens development that we observe following Fgf10 overexpression is incomplete, with lenses being quite small and lacking normal anterior-posterior polarity ( Fig. 4 ; Figs S3 and 4 ). Moreover, lens differentiation is delayed, as seen...

... of P1 lenses (compare Fig. 2 M,N) raised a major question regarding the status of lens epithelium in Snf2h cKO lenses. Lens epithelium is marked by expression of FoxE3 and E-cadherin, and higher levels of Pax6 expression are found in lens epithelium than in lens fibers. In E12.5-E14.5 lens, expression...

... as a global regulator of LF cell gene expression. Intriguingly, Prox1 also controls the expression of fibroblast growth factor receptors (FGFRs) and can bind to their promoters, correlating with decreased downstream signaling through MAPK and AKT in Prox1 mutant lenses. Further, culturing rat lens explants...

... of integrin- and FAK-mediated LE cell adhesion during lens development. Mouse lenses were fixed in 4% formaldehyde for 30 min at room temperature, incubated in 10% sucrose overnight at 4°C, embedded in OCT compound (Tissue-Tek), and cryosectioned at 10 μm. Frozen sections were incubated in 0.3% Triton X...

... and explant assays. Our results provide evidence that at blastula stages, BMP signals inhibit the acquisition of eye-field character, but from neural tube/optic vesicle stages, BMP signals from the lens are crucial for the maintenance of eye-field character, inhibition of dorsal telencephalic cell identity...

... in postmitotic transgenic lenses prompted re-entry into the cell cycle ( Chen et al., 2000 ). The cell cycle regulators p27 Kip1 and p57 Kip2 are inhibitors of cyclin-dependent kinases and are expressed throughout the lens in distinct but overlapping patterns in the lens equatorial region. Depletion of p27...

... of hexagonal cells. The mechanism that controls this morphogenesis from randomly packed cuboidal epithelial cells to highly organized hexagonal fiber cells remains unknown. In Epha2 -/- mouse lenses, equatorial epithelial cells fail to form precisely aligned meridional rows; moreover, the lens fulcrum, where...

... lenses were homogenized in RIPA buffer for protein preparation and then subjected to western blot analysis ( Xie et al., 2007 ). Each western blot was repeated at least twice with independent preparations of lens proteins. Radiographic films were scanned and band intensity was analyzed using Adobe...

... on a complex set of cell movements coordinated between the prospective neural retina, retinal pigmented epithelium (RPE) and lens. Optic vesicle evagination persists for longer than expected; cells move in a pinwheel pattern during optic vesicle elongation and retinal precursors involute around the rim...

... nuclei was calculated. Average and s.d. were determined using images of three sectioned lenses from different embryos. The maximum and minimum thickness of lens fiber cell nuclei were measured from images of lens sections and the ratio of the maximum to the minimum thickness was calculated for individual...

... ; Chow et al., 1999 ). Embryos were then analyzed for Shroom3 expression by in situ hybridization ( Fig. 6D-G ). Control embryos did not produce ectopic lenses and Shroom3 expression was observed at low levels in the developing eye, otic vesicle, branchial arches ( Fig. 6D,F ) and in the lens...

..., but development arrests prior to optic cup formation in both the optic neuroepithelium and lens ectoderm. This is accompanied by failure to maintain or initiate the expression patterns of optic-vesicle-patterning and lens-inducing determinants. Of the signaling pathways examined, only BMP signaling is noticeably...

... recombination experiments( Fujiwara et al., 1994 ), the generation of Pax6 mutant chimeric mice( Collinson et al., 2000 ),lineage-traced ectopic lenses ( Chow et al.,1999 ) and Pax6 conditional deletion( Ashery-Padan et al., 2000 ),Pax6 has an autonomous role in lens development. When Pax6 is misexpressed...

... the regulatory interactions of these two intrinsic factors with the canonical Wnt pathway. Analysis of the Pax6-deficient lenses revealed a requirement for Pax6 in cell cycle exit and differentiation into lens fiber cells. In addition, Pax6 disruption led to apoptosis of lens epithelial cells. We show that Pax6...

... and analyzed the major enhancer of FoxE3 ( Lens1 ), a gene essential for lens formation that is activated in the presumptive lens ectoderm (PLE) when commitment to the lens fate occurs. Deletion and mutation analyses of the enhancer based on comparison of Xenopus and mammalian sequences and in vitro...

... of phospho-Erk and ERM were also downregulated in Ndst1 -mutant lenses. Taken together, these results establish an important role of Ndst1 function in FGF signaling during lens development. * Author for correspondence (e-mail: xz4@iupui.edu ) 4 10 2006 © 2006. 2006 Ndst HSPG BMP...

...- to four-cell stage and the effects on opacity were analyzed by bright-field microscopy, confocal microscopy and histology ( Fig. 4 ). Wild-type embryo lenses were clear (0% opaque, n =300)( Fig. 4A , lane 1). Uninjected cloche embryos had a cloudy and opaque lens (84% opaque, n =633), as measured...

... -defective lenses lose Foxe3 expression, placing Foxe3 downstream of Sip1. In the Sip1 -defective lens, β-crystallin-expressing immature lens fiber cells were produced, but γ-crystallin-expressing mature fiber cells were absent, indicating the requirement for Sip1 activity in lens fiber maturation. A 6.2 kb...