ArticleProceedings of the National Academy of Sciences of the United States of America2004
The extracellular matrix gene Frem1 is essential for the normal adhesion of the embryonic epidermis.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2004. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 62 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
62 citing papers in PubMed, 123 citations in OpenAlex.
- Orphan GPR84 facilitates uropod de-adhesion at multiple steps during leukocyte extravasation.EMBO reports · 2026Article
- Genetic variants in Ecuadorian families: unraveling the complexity of non-syndromic cleft lip and palate a case series.BMC oral health · 2026Article
- Frem2 knockout mice exhibit Fraser syndrome phenotypes and neonatal lethality due to bilateral renal agenesis.Scientific reports · 2025Article
- Article
- Fraser Syndrome: A Narrative Review Based on a Case from Vietnam and the Past 20 Years of Research.Diagnostics (Basel, Switzerland) · 2025Article
- Proteomic analysis of the sponge Aggregation Factor implicates an ancient toolkit for allorecognition and adhesion in animals.Proceedings of the National Academy of Sciences of the United States of America · 2024Article
- Bovine FRAS1: mRNA Expression Profile, Genetic Variations, and Significant Correlations with Ovarian Morphological Traits, Mature Follicle, and Corpus Luteum.Animals : an open access journal from MDPI · 2024Article
- Two novel mutations within FREM1 gene in patients with bifid nose.BMC pediatrics · 2023Article
- Frem1 activity is regulated by Sonic hedgehog signaling in the cranial neural crest mesenchyme during midfacial morphogenesis.Developmental dynamics : an official publication of the American Association of Anatomists · 2023Article
- Transcriptome Response of Differentiating Muscle Satellite Cells to Thermal Challenge in Commercial Turkey.Genes · 2022Article
- METTL3 promotes non-small cell lung cancer (NSCLC) cell proliferation and colony formation in a m6A-YTHDF1 dependent way.BMC pulmonary medicine · 2022Article
- Associations of Heavy Metals with Activities of Daily Living Disability: An Epigenome-Wide View of DNA Methylation and Mediation Analysis.Environmental health perspectives · 2022Article
- Natural Immunity against HIV-1: Progression of Understanding after Association Studies.Viruses · 2022Review
- Cell death in the developing vertebrate limb: A locally regulated mechanism contributing to musculoskeletal tissue morphogenesis and differentiation.Developmental dynamics : an official publication of the American Association of Anatomists · 2021Review
- Human disease-associated extracellular matrix orthologs ECM3 and QBRICK regulate primary mesenchymal cell migration in sea urchin embryos.Experimental animals · 2021Article
- The Potential Role of FREM1 and Its Isoform TILRR in HIV-1 Acquisition through Mediating Inflammation.International journal of molecular sciences · 2021Review
- Circular RNA profiling identifies circ102049 as a key regulator of colorectal liver metastasis.Molecular oncology · 2021Article
- Facial cleft? The first case of manitoba-oculo-tricho-anal syndrome with novel mutations in China: a case report.BMC pediatrics · 2021Article
- Screening and Identification of Key Common and Specific Genes and Their Prognostic Roles in Different Molecular Subtypes of Breast Cancer.Frontiers in molecular biosciences · 2021Article
- Elevated expression of FREM1 in breast cancer indicates favorable prognosis and high-level immune infiltration status.Cancer medicine · 2020Article
2 more citing papers are in PubMed but not listed here.
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Authors and funding
6 authors at 4 institutions in 2 countries.
Funding
Abstract
Fraser syndrome is a rare recessive disorder characterized by cryptophthalmos, syndactyly, renal defects, and a range of other developmental abnormalities. Because of their extensive phenotypic overlap, the mouse blebbing mutants have been considered models of this disorder, and the recent isolation of mutations in Fras1 in both the blebbed mouse and human Fraser patients confirms this hypothesis. Here we report the identification of mutations in an extracellular matrix gene Fras1-related extracellular matrix gene 1 (Frem1) in both the classic head blebs mutant and in an N-ethyl-N-nitrosourea-induced allele. We show that inactivation of the gene results in the formation of in utero epidermal blisters beneath the lamina densa of the basement membrane and also in renal agenesis. Frem1 is expressed widely in the developing embryo in regions of epithelial/mesenchymal interaction and epidermal remodeling. Furthermore, Frem1 appears to act as a dermal mediator of basement membrane adhesion, apparently independently of the other known "blebs" proteins Fras1 and Grip1. Unlike both Fras1 and Grip1 mutants, collagen VI and Fras1 deposition in the basement membrane is normal, indicating that the protein plays an independent role in epidermal differentiation and is required for epidermal adhesion during embryonic development.
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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.