ArticleDevelopmental biology2017
Proper ciliary assembly is critical for restricting Hedgehog signaling during early eye development in mice.
Article in Developmental biology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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Who cites it
20 citing papers in PubMed.
- Research Progress on the Pathogenesis and Diagnostic and Therapeutic Potential of Ciliopathies Regulated by IFT172.Clinical genetics · 2026Review
- Primary Cilia of Astrocytes in the Retina in Development and Disease.Investigative ophthalmology & visual science · 2026Article
- Targeting the Hedgehog Signaling Pathway in the Retina Using Quercetin-Loaded Lipid Nanoparticles for Myopia Control.Translational vision science & technology · 2026Article
- Foxn3 is part of a transcriptional network that regulates primary cilia in the developing retina.Development (Cambridge, England) · 2026Article
- Construction and Identification of a Novel Mice Model of Microphthalmia.Translational vision science & technology · 2024Article
- Primary Cilium in Neural Crest Cells Crucial for Anterior Segment Development and Corneal Avascularity.Investigative ophthalmology & visual science · 2024Article
- Compartmentalized ciliation changes of oligodendrocytes in aged mouse optic nerve.Journal of neuroscience research · 2024Article
- Role of ciliopathy protein TMEM107 in eye development: insights from a mouse model and retinal organoid.Life science alliance · 2023Article
- Genome-wide screening reveals the genetic basis of mammalian embryonic eye development.BMC biology · 2023Article
- Primary cilia-associated protein IFT172 in ciliopathies.Frontiers in cell and developmental biology · 2023Review
- Characterization of Primary Cilia Formation in Human ESC-Derived Retinal Organoids.Stem cells international · 2023Article
- Signaling - transcription interactions in mouse retinal ganglion cells early axon pathfinding -a literature review.Frontiers in ophthalmology · 2023Review
- Changes in DNA methylation hallmark alterations in chromatin accessibility and gene expression for eye lens differentiation.Epigenetics & chromatin · 2022Article
- Primary Cilia in Amacrine Cells in Retinal Development.Investigative ophthalmology & visual science · 2021Article
- Optic vesicle morphogenesis requires primary cilia.Developmental biology · 2020Article
- Murine germ cell-specific disruption of Ift172 causes defects in spermiogenesis and male fertility.Reproduction (Cambridge, England) · 2020Article
- Limitations and Promise of Retinal Tissue From Human Pluripotent Stem Cells for Developing Therapies of Blindness.Frontiers in cellular neuroscience · 2020Review
- Primary cilia deficiency in neural crest cells models anterior segment dysgenesis in mouse.eLife · 2019Article
- The hedgehog pathway and ocular developmental anomalies.Human genetics · 2019Review
- Cell cycle-related kinase regulates mammalian eye development through positive and negative regulation of the Hedgehog pathway.Developmental biology · 2018Article
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Authors and funding
3 authors.
Funding
Abstract
Patterning of the vertebrate eye into optic stalk, retinal pigment epithelium (RPE) and neural retina (NR) territories relies on a number of signaling pathways, but how these signals are interpreted by optic progenitors is not well understood. The primary cilium is a microtubule-based organelle that is essential for Hedgehog (Hh) signaling, but it has also been implicated in the regulation of other signaling pathways. Here, we show that the optic primordium is ciliated during early eye development and that ciliogenesis is essential for proper patterning and morphogenesis of the mouse eye. Ift172 mutants fail to generate primary cilia and exhibit patterning defects that resemble those of Gli3 mutants, suggesting that cilia are required to restrict Hh activity during eye formation. Ift122 mutants, which produce cilia with abnormal morphology, generate optic vesicles that fail to invaginate to produce the optic cup. These mutants also lack formation of the lens, RPE and NR. Such phenotypic features are accompanied by strong, ectopic Hh pathway activity, evidenced by altered gene expression patterns. Removal of GLI2 from Ift122 mutants rescued several aspects of optic cup and lens morphogenesis as well as RPE and NR specification. Collectively, our data suggest that proper assembly of primary cilia is critical for restricting the Hedgehog pathway during eye formation in the mouse.
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