ArticleDevelopmental biology2015
Sox4 regulates choroid fissure closure by limiting Hedgehog signaling during ocular morphogenesis.
Article in Developmental biology, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 30 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
30 citing papers in PubMed, 50 citations in OpenAlex.
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- Introgression dynamics of sex-linked chromosomal inversions shape the Malawi cichlid radiation.Science (New York, N.Y.) · 2025Article
- IHH enhancer variant within neighboring NHEJ1 intron causes microphthalmia anophthalmia and coloboma.NPJ genomic medicine · 2023Article
- Zebrafish model of RERE syndrome recapitulates key ophthalmic defects that are rescued by small molecule inhibitor of shh signaling.Developmental dynamics : an official publication of the American Association of Anatomists · 2023Article
- Making a head: Neural crest and ectodermal placodes in cranial sensory development.Seminars in cell & developmental biology · 2023Review
- Comparative Analysis of miRNA-mRNA Regulation in the Testes ofInternational journal of molecular sciences · 2023Article
- Embryonic hyperglycemia perturbs the development of specific retinal cell types, including photoreceptors.Journal of cell science · 2022Article
- Biliverdin regulates NR2E3 and zebrafish retinal photoreceptor development.Scientific reports · 2022Article
- Eyes on CHARGE syndrome: Roles of CHD7 in ocular development.Frontiers in cell and developmental biology · 2022Review
- Characterization of Biological Pathways Regulating Acute Cold Resistance of Zebrafish.International journal of molecular sciences · 2021Article
- Nf2 fine-tunes proliferation and tissue alignment during closure of the optic fissure in the embryonic mouse eye.Human molecular genetics · 2020Article
- Genetics Underlying the Interactions between Neural Crest Cells and Eye Development.Journal of developmental biology · 2020Review
- Dual SMAD inhibition and Wnt inhibition enable efficient and reproducible differentiations of induced pluripotent stem cells into retinal ganglion cells.Scientific reports · 2020Article
- Her9/Hes4 is required for retinal photoreceptor development, maintenance, and survival.Scientific reports · 2020Article
- The Ocular Neural Crest: Specification, Migration, and Then What?Frontiers in cell and developmental biology · 2020Review
- An update on the genetics of ocular coloboma.Human genetics · 2019Review
- Genetics of congenital eye malformations: insights from chick experimental embryology.Human genetics · 2019Review
- Ubiquitin-mediated proteasome degradation regulates optic fissure fusion.Biology open · 2019Article
- Hedgehog signaling regulates cell motility and optic fissure and stalk formation during vertebrate eye morphogenesis.Development (Cambridge, England) · 2018Article
Corrections and comments
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
Abstract
SoxC transcription factors play critical roles in many developmental processes, including neurogenesis, cardiac formation, and skeletal differentiation. In vitro and in vivo loss-of-function studies have suggested that SoxC genes are required for oculogenesis; however the mechanism was poorly understood. Here, we have explored the function of the SoxC factor Sox4 during zebrafish eye development. We show that sox4a and sox4b are expressed in the forebrain and periocular mesenchyme adjacent to the optic stalk during early eye development. Knockdown of sox4 in zebrafish resulted in coloboma, a structural malformation of the eye that is a significant cause of pediatric visual impairment in humans, in which the choroid fissure fails to close. Sox4 morphants displayed altered proximo-distal patterning of the optic vesicle, including expanded pax2 expression in the optic stalk, as well as ectopic cell proliferation in the retina. We show that the abnormal ocular morphogenesis observed in Sox4-deficient zebrafish is caused by elevated Hedgehog (Hh) signaling, and this is due to increased expression of the Hh pathway ligand Indian Hedgehog b (ihhb). Consistent with these results, coloboma in sox4 morphants could be rescued by pharmacological treatment with the Hh inhibitor cyclopamine, or by co-knockdown of ihhb. Conversely, overexpression of sox4 reduced Hh signaling and ihhb expression, resulting in cyclopia. Finally, we demonstrate that sox4 and sox11 have overlapping, but not completely redundant, functions in regulating ocular morphogenesis. Taken together, our data demonstrate that Sox4 is required to limit the extent of Hh signaling during eye development, and suggest that mutations in SoxC factors could contribute to the development of coloboma.
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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.