ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2012
OTX2 and CRX rescue overlapping and photoreceptor-specific functions in the Drosophila eye.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers, 1 of them a synthesis that pooled it.
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Who cites it
20 citing papers in PubMed, 1 synthesis or guideline pooled it, 30 citations in OpenAlex.
- New Insights on the Regulatory Gene Network Disturbed in Central Areolar Choroidal Dystrophy-Beyond Classical Gene Candidates.Frontiers in genetics · 2022Pooled it
- Pathogenic variants inGenome research · 2024Article
- Transcriptional precision in photoreceptor development and diseases - Lessons from 25 years of CRX research.Frontiers in cellular neuroscience · 2024Review
- Pathogenic variants in Crx have distinct cis-regulatory effects on enhancers and silencers in photoreceptors.bioRxiv : the preprint server for biology · 2023Article
- Cat LCA-CRX Model, Homozygous for an Antimorphic Mutation Has a Unique Phenotype.Translational vision science & technology · 2023Article
- Gene Augmentation for Autosomal Dominant CRX-Associated Retinopathies.Advances in experimental medicine and biology · 2023Review
- Cell-specific occupancy dynamics between the pioneer-like factor Opa/ZIC and Ocelliless/OTX regulate early head development in embryos.Frontiers in cell and developmental biology · 2023Article
- Disease-causing mutations in genes encoding transcription factors critical for photoreceptor development.Frontiers in molecular neuroscience · 2023Review
- Human eye conditions: insights from the fly eye.Human genetics · 2019Review
- Otx2 enhances transdifferentiation of Müller cells-derived retinal stem cells into photoreceptor-like cells.Journal of cellular and molecular medicine · 2019Article
- Overexpression of MiR-183/96/182 Triggers Retina-Like Fate in Human Bone Marrow-Derived Mesenchymal Stem Cells (hBMSCs) in Culture.Journal of ophthalmology · 2019Article
- CrxRdy Cat: A Large Animal Model for CRX-Associated Leber Congenital Amaurosis.Investigative ophthalmology & visual science · 2016Article
- Ancient default activators of terminal photoreceptor differentiation in the pancrustacean compound eye: the homeodomain transcription factors Otd and Pph13.Current opinion in insect science · 2016Review
- Vision from next generation sequencing: multi-dimensional genome-wide analysis for producing gene regulatory networks underlying retinal development, aging and disease.Progress in retinal and eye research · 2015Review
- Mechanisms of blindness: animal models provide insight into distinct CRX-associated retinopathies.Developmental dynamics : an official publication of the American Association of Anatomists · 2014Review
- Common transcriptional mechanisms for visual photoreceptor cell differentiation among Pancrustaceans.PLoS genetics · 2014Article
- Genetic dissection of photoreceptor subtype specification by the Drosophila melanogaster zinc finger proteins elbow and no ocelli.PLoS genetics · 2014Article
- Mechanistically distinct mouse models for CRX-associated retinopathy.PLoS genetics · 2014Article
- OTX2 loss causes rod differentiation defect in CRX-associated congenital blindness.The Journal of clinical investigation · 2014Article
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Authors and funding
7 authors at 3 institutions in 1 country.
Funding
Abstract
backgroundOtd-related transcription factors are evolutionarily conserved to control anterior patterning and neurogenesis. In humans, two such factors, OTX2 and CRX, are expressed in all photoreceptors from early specification through adulthood and associate with several photoreceptor-specific retinopathies. It is not well understood how these factors function independently vs. redundantly, or how specific mutations lead to different disease outcomes. It is also unclear how OTX1 and OTX2 functionally overlap during other aspects of neurogenesis and ocular development. Drosophila encodes a single Otd factor that has multiple functions during eye development. Using the Drosophila eye as a model, we tested the ability of the human OTX1, OTX2, and CRX genes, as well as several disease-associated CRX alleles, to rescue the different functions of Otd.
resultsOur results indicate the following: OTX2 and CRX display overlapping, yet distinct subfunctions of Otd during photoreceptor differentiation; CRX disease alleles can be functionally distinguished based on their rescue properties; and all three factors are able to rescue rhabdomeric photoreceptor morphogenesis.
conclusionsOur findings have important implications for understanding how Otx proteins have subfunctionalized during evolution, and cement Drosophila as an effective tool to unravel the molecular bases of photoreceptor pathogenesis.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.