ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2014
Role of heparan sulfate proteoglycans in optic disc and stalk morphogenesis.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2014. 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, 23 citations in OpenAlex.
- Receptor tyrosine kinase signaling in mammalian eye development.Developmental biology · 2026Review
- A novel missense variant of HS6ST2 gene in Paganini-Miozzo syndrome with a rare neurodevelopmental and Endocrine phenotypes.Frontiers in genetics · 2025Article
- Signaling - transcription interactions in mouse retinal ganglion cells early axon pathfinding -a literature review.Frontiers in ophthalmology · 2023Review
- MicroRNA-96-5p is negatively regulating GPC3 in the metastasis of papillary thyroid cancer.SAGE open medicine · 2023Article
- Chondroitin sulfate enhances the barrier function of basement membrane assembled by heparan sulfate.Development (Cambridge, England) · 2022Article
- Ehlers-Danlos syndromes and their manifestations in the visual system.Frontiers in medicine · 2022Review
- Jack of all trades, master of each: the diversity of fibroblast growth factor signalling in eye development.Open biology · 2022Review
- MicroRNA-96: A therapeutic and diagnostic tumor marker.Iranian journal of basic medical sciences · 2022Review
- EGFR-Dependent Extracellular Matrix Protein Interactions Might Light a Candle in Cell Behavior of Non-Small Cell Lung Cancer.Frontiers in oncology · 2021Review
- Ubiquitin-mediated proteasome degradation regulates optic fissure fusion.Biology open · 2019Article
- A HS6ST2 gene variant associated with X-linked intellectual disability and severe myopia in two male twins.Clinical genetics · 2019Article
- Deciphering functional glycosaminoglycan motifs in development.Current opinion in structural biology · 2018Review
- miR-96 promotes invasion and metastasis by targeting GPC3 in non-small cell lung cancer cells.Oncology letters · 2018Article
- Multiple roles of epithelial heparan sulfate in stomach morphogenesis.Journal of cell science · 2018Article
- Flexible Roles for Proteoglycan Sulfation and Receptor Signaling.Trends in neurosciences · 2018Review
- Distinct Requirements for FGFR1 and FGFR2 in Primitive Endoderm Development and Exit from Pluripotency.Developmental cell · 2017Article
- Retinal Proteoglycans Act as Cellular Receptors for Basement Membrane Assembly to Control Astrocyte Migration and Angiogenesis.Cell reports · 2016Article
- The role of heparan sulphate in development: the ectodermal story.International journal of experimental pathology · 2016Review
- Development of astrocytes in the vertebrate eye.Developmental dynamics : an official publication of the American Association of Anatomists · 2014Article
- The Fibroblast Growth Factor signaling pathway.Wiley interdisciplinary reviews. Developmental biologyReview
Corrections and comments
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Authors and funding
3 authors at 3 institutions in 2 countries.
Funding
Abstract
backgroundHeparan sulfate proteoglycans (HSPG) are important for embryonic development by means of the regulation of gradient formation and signaling of multiple growth factors and morphogens. Previous studies have shown that Bmp/Shh/Fgf signaling are required for the regionalization of the optic vesicle (OV) and for the closure of the optic fissure (OF), the disturbance of which underlie ocular anomalies such as microphthalmia, coloboma, and optic nerve hypoplasia.
resultsTo study HSPG-dependent coordination of these signaling pathways during mammalian visual system development, we have generated a series of OV-specific mutations in the heparan sulfate (HS) N-sulfotransferase genes (Ndst1 and Ndst2) and HS O-sulfotransferase genes (Hs2st, Hs6st1, and Hs6st2) in mice. Of interest, the resulting HS undersulfation still allowed for normal retinal neurogenesis and optic fissure closure, but led to defective optic disc and stalk development. The adult mutant animals further developed optic nerve aplasia/hypoplasia and displayed retinal degeneration. We observed that MAPK/ERK signaling was down-regulated in Ndst mutants, and consistent with this, HS-related optic nerve morphogenesis defects in mutant mice could partially be rescued by constitutive Kras activation.
conclusionsThese results suggest that HSPGs, depending on their HS sulfation pattern, regulate multiple signaling pathways in optic disc and stalk morphogenesis.
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Registered trials
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.