Project description:Gene expression profiling of zebrafish early eye development on 3 to 5 days post fertilization (dpf) Gene expression on 3 to 5 dpf eyes were compared. Each sample contains three replicates.
Project description:Mutations in Peroxidasin (PXDN) cause severe inherited eye disorders in humans, such as congenital cataract, corneal opacity, and developmental glaucoma. The role of peroxidasin during eye development is poorly understood. Here we describe the first Pxdn mouse mutant which was induced by ENU (N-ethyl-N-nitrosourea) and led to a recessive phenotype. Sequence analysis of cDNA revealed a T3816A mutation resulting in a premature stop codon (Cys1272X) in the peroxidase domain. This mutation causes severe anterior segment dysgenesis and microphthalmia resembling the manifestations in patients with PXDN mutations. The proliferation and differentiation of the lens is disrupted in association with aberrant expression of transcription factor genes (Pax6 and Foxe3) in mutant eyes. Additionally, Pxdn is involved in the consolidation of the basement membrane and lens epithelium adhesion in the ocular lens. Lens material including γ-crystallin is extruded into the anterior and posterior chamber due to local loss of structural integrity of the lens capsule as a secondary damage to the anterior segment development leading to congenital ocular inflammation. Moreover, Pxdn mutants exhibited an early-onset glaucoma and progressive retinal dysgenesis. Transcriptome profiling revealed that peroxidasin affects the transcription of developmental and eye diseases-related genes at early eye development. These findings suggest that peroxidasin is necessary for cell proliferation and differentiation and for basement membrane consolidation during eye development. Our studies provide pathogenic mechanisms of PXDN mutation-induced congenital eye diseases.
Project description:eyeless (ey) is one of the most critical transcription factors for initiating the entire eye development in Drosophila. However, the molecular mechanisms through which Ey regulates target genes and pathways have not been characterized at the genomic level. Using ChIP-Seq, we generated an endogenous Ey binding profile in Drosophila developing eyes. These data sets will serve as a valuable resource for future studies on Ey and eye development.
Project description:We report the proteome composition of the Drosophila eye – a compound organ that is highly enriched in membrane proteins. The fly eye is a popular model to study the physiology of vision by means of genetic, pharmacological, and dietary interference.While the eye transcriptome and development-related changes of gene expression profiles have been extensively studied, little is known about the eye proteome.we employed GeLC-MS/MS to identify and rank the abundances of 3516 eye proteins. Moreover, we applied our MS Western method to determine the absolute (molar) abundances of a related set of proteins that are important for photoreceptor structure (including maintenance) and function (phototransduction). Altogether, we provide a comprehensive and expandable proteomics resource that will be valuable for a variety of studies of ocular biochemistry, physiology, and development.
Project description:Coordinating cell proliferation and differentiation is essential during organogenesis. In Drosophila, the photoreceptor, pigment and support cells of the eye are specified in an orchestrated wave as the morphogenetic furrow passes across the eye imaginal disc. Cells anterior of the furrow are uncommitted to cell type and remain mitotically active, while most cells in the furrow arrest at G1 and adopt specified ommatidial fates. We used microarray expression analysis to monitor changes in transcription at the furrow and identified genes whose expression correlates with either proliferation or fate specification.