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    CLCN7 chloride channel, voltage-sensitive 7 [ Homo sapiens (human) ]

    Gene ID: 1186, updated on 5-May-2013
    Official Symbol
    CLCN7provided by HGNC
    Official Full Name
    chloride channel, voltage-sensitive 7provided by HGNC
    Primary source
    HGNC:2025
    Locus tag
    LA16c-390E6.1
    See related
    Ensembl:ENSG00000103249; HPRD:04103; MIM:602727; Vega:OTTHUMG00000044467
    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
    CLC7; CLC-7; OPTA2; OPTB4; PPP1R63
    Summary
    The product of this gene belongs to the CLC chloride channel family of proteins. Chloride channels play important roles in the plasma membrane and in intracellular organelles. This gene encodes chloride channel 7. Defects in this gene are the cause of osteopetrosis autosomal recessive type 4 (OPTB4), also called infantile malignant osteopetrosis type 2 as well as the cause of autosomal dominant osteopetrosis type 2 (OPTA2), also called autosomal dominant Albers-Schonberg disease or marble disease autosoml dominant. Osteopetrosis is a rare genetic disease characterized by abnormally dense bone, due to defective resorption of immature bone. OPTA2 is the most common form of osteopetrosis, occurring in adolescence or adulthood. [provided by RefSeq, Jul 2008]
    Location :
    16p13
    Sequence :
    Chromosome: 16; NC_000016.9 (1494934..1525085, complement)
    See CLCN7 in Epigenomics, MapViewer

    Chromosome 16 - NC_000016.9Genomic Context describing neighboring genes Neighboring gene chromosome 16 open reading frame 91 Neighboring gene coiled-coil domain containing 154 Neighboring gene ribosomal protein S3A pseudogene 2 Neighboring gene pentraxin 4, long

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

    Osteopetrosis autosomal dominant type 2

    Summary from GeneReviews: CLCN7-Related Osteopetrosis Go to GeneReviews

    Disease Characteristics
    The spectrum of CLCN7-related osteopetrosis includes the following disorders: Infantile malignant CLCN7-related recessive osteopetrosis (ARO). Intermediate autosomal osteopetrosis (IAO). Autosomal dominant osteopetrosis type II (ADOII, Albers-Schonberg disease). Onset of ARO is in infancy; findings may include fractures; poor growth; sclerosis of the skull base (with or without choanal stenosis or hydrocephalus) resulting in optic nerve compression, facial palsy, hearing loss; absence of the bone marrow cavity resulting in severe anemia and thrombocytopenia; dental abnormalities, odontomas, risk for mandibular osteomyelitis; and hypocalcemia with tetanic seizures and secondary hyperparathyroidism. Without treatment maximal life span in ARO is ten years. Onset of IAO is in childhood; findings may include fractures after minor trauma; characteristic skeletal radiographic changes found incidentally; mild anemia; occasional visual impairment secondary to optic nerve compression. Life expectancy in IAO is usually normal. Onset of ADOII is usually late childhood or adolescence; findings may include fractures (in any long bone and/or the posterior arch of a vertebra); scoliosis; hip osteoarthritis; osteomyelitis of the mandible or septic osteitis or osteoarthritis elsewhere. Cranial nerve compression is rare.
    Diagnosis Testing
    Diagnosis of CLCN7-related osteopetrosis usually relies on radiographic changes that are pathognomonic in ARO (generalized osteosclerosis, club-shaped long bones, osteosclerosis of the skull base, bone-within-bone appearance) and characteristic in ADOII (osteosclerosis of the spine ("sandwich vertebra" appearance), bone-within-bone appearance (mainly iliac wings), Erlenmeyer-shaped femoral metaphysis, mild osteosclerosis of the skull base, transverse bands of osteosclerosis in long bones). Molecular genetic testing of CLCN7, the only gene associated with CLCN7-related osteopetrosis, is available on a clinical basis.
    Genetic Counseling
    ARO is inherited in an autosomal recessive manner; ADOII is inherited in an autosomal dominant manner; about 40% of IAO is inherited in an autosomal recessive manner and about 60% in an autosomal dominant manner. Autosomal recessive inheritance: at conception, each sib of an affected individual has a 25% chance of being affected, a 50% chance of being an asymptomatic carrier, and a 25% chance of being unaffected and not a carrier. Individuals with ARO in general only reproduce if successfully treated by HSCT. Autosomal dominant inheritance: most individuals diagnosed with autosomal dominant CLCN7-related osteopetrosis have an affected parent. The proportion of cases caused by de novo mutations is unknown. Each child of an individual with autosomal dominant CLCN7-related osteopetrosis has a 50% chance of inheriting the mutation. Prenatal diagnosis for pregnancies at increased risk for ADOII and ARO is possible if the disease-causing mutation(s) have been identified in the family.
    References
    Products Interactant Other Gene Complex Source Pubs Description
    NP_001278.1 NP_060496.2 PACS1    BIND  PubMed PACS-1 interacts with CLC-7. 
    P51798 Q86WC4 OSTM1    HPRD  PubMed  
    P51798 Q6VY07 PACS1    HPRD  PubMed  
    BioGRID:107600 BioGRID:113164 UBC    BioGRID  PubMed Affinity Capture-MS 

    Markers

    Homology

    Clone Names

    • FLJ26686, FLJ39644, FLJ46423

    Gene Ontology Provided by GOA

    Function Evidence Code Pubs
    ATP binding IEA
    Inferred from Electronic Annotation
    more info
     
    antiporter activity IEA
    Inferred from Electronic Annotation
    more info
     
    chloride channel activity TAS
    Traceable Author Statement
    more info
    PubMed 
    voltage-gated chloride channel activity IEA
    Inferred from Electronic Annotation
    more info
     
    Process Evidence Code Pubs
    ion transmembrane transport TAS
    Traceable Author Statement
    more info
     
    response to pH IEA
    Inferred from Electronic Annotation
    more info
     
    transmembrane transport TAS
    Traceable Author Statement
    more info
     
    transport TAS
    Traceable Author Statement
    more info
    PubMed 
    Component Evidence Code Pubs
    cytoplasmic vesicle IEA
    Inferred from Electronic Annotation
    more info
     
    integral to membrane IEA
    Inferred from Electronic Annotation
    more info
     
    lysosomal membrane TAS
    Traceable Author Statement
    more info
     
    Preferred Names
    H(+)/Cl(-) exchange transporter 7
    Names
    H(+)/Cl(-) exchange transporter 7
    chloride channel protein 7
    chloride channel 7 alpha subunit
    protein phosphatase 1, regulatory subunit 63

    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_007567.1 RefSeqGene

      Range
      5001..35152
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    mRNA and Protein(s)

    1. NM_001114331.2NP_001107803.1  H(+)/Cl(-) exchange transporter 7 isoform b

      Status: REVIEWED

      Description
      Transcript Variant: This variant (2) lacks an in-frame segment of the coding region, compared to variant 1. It encodes a shorter isoform (b), that is missing an internal segment compared to isoform a.
      Source sequence(s)
      AA741117, AK056551, AL031600, DB061109
      Consensus CDS
      CCDS45378.1
      UniProtKB/Swiss-Prot
      P51798
      Related
      ENSP00000410907, ENST00000448525
      Conserved Domains (4) summary
      COG0517
      Location:714771
      Blast Score: 102
      COG0517; FOG: CBS domain [General function prediction only]
      cd04591
      Location:612766
      Blast Score: 237
      CBS_pair_EriC_assoc_euk_bac; This cd contains two tandem repeats of the cystathionine beta-synthase (CBS pair) domains in the EriC CIC-type chloride channels in eukaryotes and bacteria. These ion channels are proteins with a seemingly simple task of allowing the passive flow of ...
      cd03685
      Location:71596
      Blast Score: 1409
      ClC_6_like; ClC-6-like chloride channel proteins. This CD includes ClC-6, ClC-7 and ClC-B, C, D in plants. Proteins in this family are ubiquitous in eukarotes and their functions are unclear. They are expressed in intracellular organelles membranes. This family ...
      pfam00654
      Location:165571
      Blast Score: 388
      Voltage_CLC; Voltage gated chloride channel
    2. NM_001287.5NP_001278.1  H(+)/Cl(-) exchange transporter 7 isoform a

      Status: REVIEWED

      Description
      Transcript Variant: This variant (1) represents the longer transcript, and encodes the longer isoform (a).
      Source sequence(s)
      AA741117, AL031600, AL031705, BC012737
      Consensus CDS
      CCDS32361.1
      UniProtKB/Swiss-Prot
      P51798
      Related
      ENSP00000372193, OTTHUMP00000041762, ENST00000382745, OTTHUMT00000103598
      Conserved Domains (4) summary
      COG0517
      Location:738795
      Blast Score: 102
      COG0517; FOG: CBS domain [General function prediction only]
      cd04591
      Location:636790
      Blast Score: 236
      CBS_pair_EriC_assoc_euk_bac; This cd contains two tandem repeats of the cystathionine beta-synthase (CBS pair) domains in the EriC CIC-type chloride channels in eukaryotes and bacteria. These ion channels are proteins with a seemingly simple task of allowing the passive flow of ...
      cd03685
      Location:95620
      Blast Score: 1400
      ClC_6_like; ClC-6-like chloride channel proteins. This CD includes ClC-6, ClC-7 and ClC-B, C, D in plants. Proteins in this family are ubiquitous in eukarotes and their functions are unclear. They are expressed in intracellular organelles membranes. This family ...
      pfam00654
      Location:189595
      Blast Score: 383
      Voltage_CLC; Voltage gated chloride channel

    RefSeqs of Annotated Genomes: Homo sapiens Annotation Release 104

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

    Reference GRCh37.p10 Primary Assembly

    Genomic

    1. NC_000016.9 Reference GRCh37.p10 Primary Assembly

      Range
      1494934..1525085, complement
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate HuRef

    Genomic

    1. AC_000148.1 Alternate HuRef

      Range
      1421403..1451021, complement
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate CHM1_1.0

    Genomic

    1. NC_018927.1 Alternate CHM1_1.0

      Range
      1429480..1459536, complement
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

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