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    FBN1 fibrillin 1 [ Homo sapiens ]

    Gene ID: 2200, updated on 20-May-2012

    Summary

    Official Symbol
    FBN1provided by HGNC
    Official Full Name
    fibrillin 1provided by HGNC
    Primary source
    HGNC:3603
    See related
    Ensembl:ENSG00000166147; HPRD:00618; MIM:134797; Vega:OTTHUMG00000172218
    Gene type
    protein coding
    RefSeq status
    REVIEWED
    Organism
    Homo sapiens
    Lineage
    Eukaryota; Metazoa; Chordata; Craniata; Vertebrata; Euteleostomi; Mammalia; Eutheria; Euarchontoglires; Primates; Haplorrhini; Catarrhini; Hominidae; Homo
    Also known as
    FBN; SGS; WMS; MASS; MFS1; OCTD; SSKS; WMS2; ACMICD; GPHYSD2
    Summary
    This gene encodes a member of the fibrillin family. The encoded protein is a large, extracellular matrix glycoprotein that serve as a structural component of 10-12 nm calcium-binding microfibrils. These microfibrils provide force bearing structural support in elastic and nonelastic connective tissue throughout the body. Mutations in this gene are associated with Marfan syndrome, isolated ectopia lentis, autosomal dominant Weill-Marchesani syndrome, MASS syndrome, and Shprintzen-Goldberg craniosynostosis syndrome. [provided by RefSeq, Jul 2008]

    Genomic context

    Location :
    15q21.1
    Sequence :
    Chromosome: 15; NC_000015.9 (48700503..48937985, complement)
    See FBN1 in Epigenomics, MapViewer

    Chromosome 15 - NC_000015.9Genomic Context describing neighboring genes Neighboring gene solute carrier family 12 (sodium/potassium/chloride transporters), member 1 Neighboring gene deoxyuridine triphosphatase Neighboring gene uncharacterized LOC100506059 Neighboring gene oxidation resistance 1 pseudogene

    Genomic regions, transcripts, and products

    Bibliography

    GeneRIFs: Gene References Into Functions What's a GeneRIF?

    Phenotypes

    A genome-wide association study of breast and prostate cancer in the NHLBI's Framingham Heart Study.

    Acromicric dysplasia

    Aortic aneurysm, ascending, and dissection

    Ectopia lentis, familial

    Geleophysic dysplasia 2

    Marfan syndrome

    Summary from GeneReviews: Go to GeneReviews

    Disease Characteristics
    Marfan syndrome is a systemic disorder of connective tissue with a high degree of clinical variability. Cardinal manifestations involve the ocular, skeletal, and cardiovascular systems. FBN1 mutations associate with a broad phenotypic continuum, ranging from isolated features of Marfan syndrome to neonatal presentation of severe and rapidly progressive disease in multiple organ systems. Myopia is the most common ocular feature; displacement of the lens from the center of the pupil, seen in approximately 60% of affected individuals, is a hallmark feature. People with Marfan syndrome are at increased risk for retinal detachment, glaucoma, and early cataract formation. The skeletal system involvement is characterized by bone overgrowth and joint laxity. The extremities are disproportionately long for the size of the trunk (dolichostenomelia). Overgrowth of the ribs can push the sternum in (pectus excavatum) or out (pectus carinatum). Scoliosis is common and can be mild or severe and progressive. The major sources of morbidity and early mortality in the Marfan syndrome relate to the cardiovascular system. Cardiovascular manifestations include dilatation of the aorta at the level of the sinuses of Valsalva, a predisposition for aortic tear and rupture, mitral valve prolapse with or without regurgitation, tricuspid valve prolapse, and enlargement of the proximal pulmonary artery. With proper management, the life expectancy of someone with Marfan syndrome approximates that of the general population.
    Diagnosis Testing
    Marfan syndrome is a clinical diagnosis based on family history and the observation of characteristic findings in multiple organ systems. Ectopia lentis and aortic aneurysm are given special significance in the diagnosis of Marfan syndrome because of their relative specificity or frequency and clinical significance, respectively. Molecular genetic testing of FBN1 is available in clinical laboratories. It remains unclear whether the lack of full sensitivity of this test relates to an atypical location or character of FBN1 mutations in some individuals (e.g., large deletions or promoter mutations) or to locus heterogeneity.
    Genetic Counseling
    Marfan syndrome is inherited in an autosomal dominant manner. Approximately 75% of individuals with Marfan syndrome have an affected parent; approximately 25% of probands with Marfan syndrome have a de novo mutation. The risk to the sibs of the proband depends on the status of the parents. If a parent is affected, the risk is 50%. If an affected child is born to clinically unaffected parents, it is likely that the child has a de novo mutation, and the risk to sibs is far less than 50% but above the population risk because of reported (but rare) cases of somatic and germline mosaicism. The children of an individual with Marfan syndrome are at 50% risk of inheriting the mutant allele and the disorder. Prenatal testing for pregnancies at increased risk is possible if the disease-causing mutation in the family is known.
    References

    MASS syndrome

    Shprintzen-Goldberg syndrome

    Stiff skin syndrome

    Weill-Marchesani syndrome 2, dominant

    Interactions

    Products Interactant Other Gene Complex Source Pubs Description
    NP_000129.2 NP_001989.1 FBLN2    BIND  PubMed Fibrillin-1 interacts with Fibulin-2. 
    P35555 P27797 CALR    HPRD  PubMed  
    P35555 P07585 DCN    HPRD  PubMed  
    P35555 P15502 ELN    HPRD  PubMed  
    P35555 P98095 FBLN2    HPRD  PubMed  
    P35555 P35556 FBN2    HPRD  PubMed  
    P35555 P98160 HSPG2    HPRD  PubMed  
    P35555 Q14766 LTBP1    HPRD  PubMed  
    P35555 P55001 MFAP2    HPRD  PubMed  
    P35555 Q13361 MFAP5    HPRD  PubMed  
    P35555 Q99972 MYOC    HPRD  PubMed  
    P35555 P13611 VCAN    HPRD  PubMed  
    BioGRID:108494 BioGRID:112221 ATXN7    BioGRID  PubMed Two-hybrid 
    BioGRID:108494 BioGRID:108321 ELN    BioGRID  PubMed Reconstituted Complex 
    BioGRID:108494 BioGRID:108493 FBLN2    BioGRID  PubMed Reconstituted Complex 
    BioGRID:108494 BioGRID:110395 MFAP2    BioGRID  PubMed Reconstituted Complex 
    BioGRID:108494 BioGRID:110736 MYOC    BioGRID  PubMed Reconstituted Complex 
    BioGRID:108494 BioGRID:115547 SPRY2    BioGRID  PubMed Two-hybrid 
    BioGRID:108494 BioGRID:107844 VCAN    BioGRID  PubMed Reconstituted Complex 

    General gene information

    Markers

    Homology

    Pathways from BioSystems

    • Integrin cell surface interactions, organism-specific biosystem (from REACTOME)
      Integrin cell surface interactions, organism-specific biosystemThe extracellular matrix (ECM) is a network of macro-molecules that underlies all epithelia and endothelia and that surrounds all connective tissue cells. This matrix provides the mechanical strength...
    • Signal Transduction, organism-specific biosystem (from REACTOME)
      Signal Transduction, organism-specific biosystemSignal transduction is a process in which extracellular signals elicit changes in cell state and activity. Transmembrane receptors sense changes in the cellular environment by binding ligands, such a...

    Gene Ontology Provided by GOA

    Function Evidence Code Pubs
    calcium ion binding IDA
    Inferred from Direct Assay
    more info
    PubMed 
    extracellular matrix structural constituent IDA
    Inferred from Direct Assay
    more info
    PubMed 
    protein binding IPI
    Inferred from Physical Interaction
    more info
    PubMed 
    Component Evidence Code Pubs
    basement membrane IDA
    Inferred from Direct Assay
    more info
    PubMed 
    colocalizes_with extracellular matrix IDA
    Inferred from Direct Assay
    more info
     
    extracellular region TAS
    Traceable Author Statement
    more info
     
    extracellular space IDA
    Inferred from Direct Assay
    more info
     
    microfibril IDA
    Inferred from Direct Assay
    more info
    PubMed 
    proteinaceous extracellular matrix IDA
    Inferred from Direct Assay
    more info
    PubMed 

    General protein information

    Preferred Names
    fibrillin-1
    Names
    fibrillin-1
    fibrillin 15

    NCBI Reference Sequences (RefSeq)

    RefSeqs maintained independently of Annotated Genomes

    These reference sequences exist independently of genome builds. Explain

    These reference sequences are curated independently of the genome annotation cycle, so their versions may not match the RefSeq versions in the current genome build. Identify version mismatches by comparing the version of the RefSeq in this section to the one reported in Genomic regions, transcripts, and products above.

    Genomic

    1. NG_008805.2 RefSeqGene

      Range
      5001..242483
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    mRNA and Protein(s)

    1. NM_000138.4NP_000129.3  fibrillin-1 precursor

      Status: REVIEWED

      Source sequence(s)
      AC084757, AC084758, BC146854
      Consensus CDS
      CCDS32232.1
      UniProtKB/Swiss-Prot
      P35555
      Related
      ENSP00000325527, OTTHUMP00000247869, ENST00000316623, OTTHUMT00000417355
      Conserved Domains (3) summary
      pfam00683
      Location:15471588
      Blast Score: 133
      TB; TB domain
      cl00057
      Location:11941235
      Blast Score: 116
      vWFA; Von Willebrand factor type A (vWA) domain was originally found in the blood coagulation protein von Willebrand factor (vWF). Typically, the vWA domain is made up of approximately 200 amino acid residues folded into a classic a/b para-rossmann type of ...
      cl09941
      Location:13671402
      Blast Score: 123
      EGF_CA; Calcium-binding EGF-like domain, present in a large number of membrane-bound and extracellular (mostly animal) proteins. Many of these proteins require calcium for their biological function and calcium-binding sites have been found to be located at the ...

    RefSeqs of Annotated Genomes: Build 37.3

    The following sections contain reference sequences that belong to a specific genome build. Explain

    Reference GRCh37.p5 Primary Assembly

    Genomic

    1. NC_000015.9 Reference GRCh37.p5 Primary Assembly

      Range
      48700503..48937985, complement
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate HuRef

    Genomic

    1. AC_000147.1 Alternate HuRef

      Range
      25532899..25770042, complement
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Related Sequences

    Nucleotide Protein
    Heading Accession and Version
    genomic AB177801.1 BAD16737.1
    genomic AB177802.1 BAD16738.1
    genomic AB177803.1 BAD16739.1
    genomic AC022467.7 (12947..193702) None
    genomic AC084757.2 (43086..45549) None
    genomic AC084758.3 (137801..192063) None
    genomic CH471082.1 EAW77353.1
      EAW77354.1
    genomic GU143398.1 ACZ58372.1
    genomic S54426.1 AAB25244.1
    mRNA AB208840.1 BAD92077.1
    mRNA AK304737.1 BAG65498.1
    mRNA AK309616.1 None
    mRNA BC094721.1 None
    mRNA BC146854.1 AAI46855.1
    mRNA L13923.1 AAB02036.1
    mRNA W81203.1 None
    mRNA X62008.1 CAB56534.1
    mRNA X63556.1 CAA45118.1
    Protein Accession Links
    GenPept Link UniProtKB Link
    P35555.3 GenPept UniProtKB/Swiss-Prot:P35555
    Q59HB9 GenPept UniProtKB/TrEMBL:Q59HB9
    Q75N88 GenPept UniProtKB/TrEMBL:Q75N88
    Q75N89 GenPept UniProtKB/TrEMBL:Q75N89
    Q9NP01 GenPept UniProtKB/TrEMBL:Q9NP01

      Supplemental Content

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