ArticleHuman molecular genetics2009
Formin1 disruption confers oligodactylism and alters Bmp signaling.
Article in Human molecular genetics, 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 27 papers, 1 of them a synthesis that pooled it.
What it found
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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.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
27 citing papers in PubMed, 1 synthesis or guideline pooled it, 49 citations in OpenAlex.
- Pooled it
- Integrated Genome-Wide Association Studies and Selection Signature Analysis Reveal the Genetic Architecture of the Wattle Trait in Goats.Animal genetics · 2026Article
- A 12 Week Fetus with Anophthalmia, Limb Anomalies and Infratemporal Teratoma.International journal of women's health · 2024Article
- Probing the ligand binding specificity of FNBP4 WW domains and interaction with FH1 domain of FMN1.Current research in structural biology · 2024Article
- Genetic cold cases: lessons from solving complex congenital limb disorders.Genes & development · 2023Review
- Unique genetic signature and selection footprints in Dutch population of German Longhaired Pointer dogs.Animal genetics · 2022Article
- Formins in Human Disease.Cells · 2021Review
- Uncovering Modifier Genes of X-Linked Alport Syndrome Using a Novel Multiparent Mouse Model.Journal of the American Society of Nephrology : JASN · 2021Article
- Deletion of morpholino binding sites (DeMOBS) to assess specificity of morphant phenotypes.Scientific reports · 2020Article
- Rab27a co-ordinates actin-dependent transport by controlling organelle-associated motors and track assembly proteins.Nature communications · 2020Article
- PTPN3 suppresses lung cancer cell invasiveness by counteracting Src-mediated DAAM1 activation and actin polymerization.Oncogene · 2019Article
- Phenotype loss is associated with widespread divergence of the gene regulatory landscape in evolution.Nature communications · 2018Article
- Non-canonical activity of the podosomal formin FMNL1γ supports immune cell migration.Journal of cell science · 2017Article
- Is toxicant-induced Sertoli cell injury in vitro a useful model to study molecular mechanisms in spermatogenesis?Seminars in cell & developmental biology · 2016Review
- Regulation of blood-testis barrier by actin binding proteins and protein kinases.Reproduction (Cambridge, England) · 2016Review
- Advances in the Molecular Genetics of Non-syndromic Syndactyly.Current genomics · 2015Article
- Cytoskeletal proteins in cortical development and disease: actin associated proteins in periventricular heterotopia.Frontiers in cellular neuroscience · 2015Review
- Biallelic truncating mutations in FMN2, encoding the actin-regulatory protein Formin 2, cause nonsyndromic autosomal-recessive intellectual disability.American journal of human genetics · 2014Article
- Formin 1 and filamin B physically interact to coordinate chondrocyte proliferation and differentiation in the growth plate.Human molecular genetics · 2014Article
- Interaction between microtubules and the Drosophila formin Cappuccino and its effect on actin assembly.The Journal of biological chemistry · 2014Article
Corrections and comments
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Authors and funding
4 authors at 2 institutions in 2 countries.
Funding
Abstract
Proper limb development requires concerted communication between cells within the developing limb bud. Several molecules have been identified which contribute to the formation of a circuitry loop consisting in large part of secreted proteins. The intracellular actin nucleator, Formin 1 (Fmn1), has previously been implicated in limb development, but questions remain after the identification of a Gremlin transcriptional enhancer within the 3' end of the Fmn 1 locus. To resolve this issue, a knockout mouse devoid of Fmn1 protein was created and characterized. The mice exhibit a reduction of digit number to four, a deformed posterior metatarsal, phalangeal soft tissue fusion as well as the absence of a fibula to 100% penetrance in the FVB genetic background. Importantly, this mutant allele does not genetically disrupt the characterized Gremlin enhancer, and indeed Gremlin RNA expression is upregulated at the 35 somite stage of development. Our data reveal increased Bone Morphogenetic Protein (Bmp) activity in mice which carry a disruption in Fmn1, as evidenced by upregulation of Msx1 and a decrease in Fgf4 within the apical ectodermal ridge. Additionally, these studies show enhanced activity downstream of the Bmp receptor in cells where Fmn1 is perturbed, suggesting a role for Fmn1 in repression of Bmp signaling.
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What OpenQuestion holds
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.