ArticlePLoS genetics2010
Genetic analysis of fin development in zebrafish identifies furin and hemicentin1 as potential novel fraser syndrome disease genes.
Article in PLoS genetics, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 68 papers.
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Who cites it
68 citing papers in PubMed, 124 citations in OpenAlex.
- A collagen orientation switch reshapes fin architecture.iScience · 2026Article
- Regeneration programs buffer genetic defects in animal development.PLoS biology · 2026Article
- Laminin α5 and integrins α3 and α6 coordinately regulate collective cell migration in vivo.Development (Cambridge, England) · 2026Article
- Review
- Systematic Disruption of Zebrafish Fibrillin Genes Identifies a Translational Zebrafish Model for Marfan Syndrome.JACC. Basic to translational science · 2026Article
- Chemical Synthesis of NICOL, A Coligand Secreted Protein Mediating Mammalian Male Reproductive Tract Trans-luminal Signaling.Chemistry (Weinheim an der Bergstrasse, Germany) · 2025Article
- wnt10a is required for zebrafish median fin fold maintenance and adult unpaired fin metamorphosis.Developmental dynamics : an official publication of the American Association of Anatomists · 2024Article
- Single-cell analysis reveals distinct fibroblast plasticity during tenocyte regeneration in zebrafish.Science advances · 2023Article
- Reciprocal discoidin domain receptor signaling strengthens integrin adhesion to connect adjacent tissues.eLife · 2023Article
- Reciprocal discoidin domain receptor signaling strengthens integrin adhesion to connect adjacent tissues.bioRxiv : the preprint server for biology · 2023Article
- Article
- The Fraser Complex Proteins (Frem1, Frem2, and Fras1) Can Form Anchoring Cords in the Absence of AMACO at the Dermal-Epidermal Junction of Mouse Skin.International journal of molecular sciences · 2023Article
- Hemicentin-mediated type IV collagen assembly strengthens juxtaposed basement membrane linkage.The Journal of cell biology · 2023Article
- Vertebrate extracellular matrix protein hemicentin-1 interacts physically and genetically with basement membrane protein nidogen-2.Matrix biology : journal of the International Society for Matrix Biology · 2022Article
- Slit-Robo signalling establishes a Sphingosine-1-phosphate gradient to polarise fin mesenchyme.EMBO reports · 2022Article
- Article
- A basement membrane discovery pipeline uncovers network complexity, regulators, and human disease associations.Science advances · 2022Article
- Single-nucleus cross-tissue molecular reference maps toward understanding disease gene function.Science (New York, N.Y.) · 2022Article
- Developmental independence of median fins from the larval fin fold revises their evolutionary origin.Scientific reports · 2022Article
- Genomic prediction of growth in a commercially, recreationally, and culturally important marine resource, the Australian snapper (Chrysophrys auratus).G3 (Bethesda, Md.) · 2022Article
8 more citing papers are in PubMed but not listed here.
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Authors and funding
14 authors at 6 institutions in 4 countries.
Funding
Abstract
Using forward genetics, we have identified the genes mutated in two classes of zebrafish fin mutants. The mutants of the first class are characterized by defects in embryonic fin morphogenesis, which are due to mutations in a Laminin subunit or an Integrin alpha receptor, respectively. The mutants of the second class display characteristic blistering underneath the basement membrane of the fin epidermis. Three of them are due to mutations in zebrafish orthologues of FRAS1, FREM1, or FREM2, large basement membrane protein encoding genes that are mutated in mouse bleb mutants and in human patients suffering from Fraser Syndrome, a rare congenital condition characterized by syndactyly and cryptophthalmos. Fin blistering in a fourth group of zebrafish mutants is caused by mutations in Hemicentin1 (Hmcn1), another large extracellular matrix protein the function of which in vertebrates was hitherto unknown. Our mutant and dose-dependent interaction data suggest a potential involvement of Hmcn1 in Fraser complex-dependent basement membrane anchorage. Furthermore, we present biochemical and genetic data suggesting a role for the proprotein convertase FurinA in zebrafish fin development and cell surface shedding of Fras1 and Frem2, thereby allowing proper localization of the proteins within the basement membrane of forming fins. Finally, we identify the extracellular matrix protein Fibrillin2 as an indispensable interaction partner of Hmcn1. Thus we have defined a series of zebrafish mutants modelling Fraser Syndrome and have identified several implicated novel genes that might help to further elucidate the mechanisms of basement membrane anchorage and of the disease's aetiology. In addition, the novel genes might prove helpful to unravel the molecular nature of thus far unresolved cases of the human disease.
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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.