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    CTNS cystinosin, lysosomal cystine transporter [ Homo sapiens (human) ]

    Gene ID: 1497, updated on 22-May-2013
    Official Symbol
    CTNSprovided by HGNC
    Official Full Name
    cystinosin, lysosomal cystine transporterprovided by HGNC
    Primary source
    HGNC:2518
    See related
    HPRD:05885; MIM:606272
    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
    PQLC4; CTNS-LSB
    Summary
    This gene encodes a seven-transmembrane domain protein that functions to transport cystine out of lysosomes. Its activity is driven by the H+ electrochemical gradient of the lysosomal membrane. Mutations in this gene cause cystinosis, a lysosomal storage disorder. Alternative splicing results in multiple transcript variants. [provided by RefSeq, Jul 2009]
    Location :
    17p13
    Sequence :
    Chromosome: 17; NC_000017.10 (3539762..3566397)
    See CTNS in Epigenomics, MapViewer

    Chromosome 17 - NC_000017.10Genomic Context describing neighboring genes Neighboring gene transient receptor potential cation channel, subfamily V, member 1 Neighboring gene pterin-4 alpha-carbinolamine dehydratase/dimerization cofactor of hepatocyte nuclear factor 1 alpha (TCF1) 2 pseudogene Neighboring gene sedoheptulokinase Neighboring gene P2RX5-TAX1BP3 readthrough Neighboring gene ribosomal protein L21 pseudogene 125 Neighboring gene Tax1 (human T-cell leukemia virus type I) binding protein 3 Neighboring gene ER membrane protein complex subunit 6

    Related articles in PubMed

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

    Cystinosis

    Summary from GeneReviews: Cystinosis Go to GeneReviews

    Disease Characteristics
    Nephropathic cystinosis in untreated children is characterized by renal tubular Fanconi syndrome, poor growth, hypophosphatemic rickets, impaired glomerular function, and accumulation of cystine crystals in almost all cells, leading to tissue destruction. The typical untreated child has short stature, light complexion, rickets, and photophobia. Growth failure is generally noticed between ages six and nine months; signs of renal tubular Fanconi syndrome (polyuria, polydipsia, dehydration, and acidosis) appear as early as age six months; corneal crystals can be present before age one year and are always present after age 16 months. Prior to the use of renal transplantation and cystine-depleting therapy, the life span in nephropathic cystinosis was no longer than ten years. With these therapies, affected individuals can survive at least into the mid-forties or fifties with satisfactory quality of life. Intermediate cystinosis is characterized by all the typical manifestations of nephropathic cystinosis, but onset is at a later age. Renal glomerular failure occurs in all untreated affected individuals, usually between ages 15 and 25 years. Non-nephropathic cystinosis is characterized only by photophobia resulting from corneal cystine crystal accumulation.
    Diagnosis Testing
    The diagnosis of cystinosis is established by documenting: (1) renal tubular Fanconi syndrome: increased urinary losses of essential nutrients including electrolytes (sodium, potassium, bicarbonate), minerals (calcium, phosphate, magnesium), glucose, amino acids, carnitine, and water; (2) typical cystine crystals in the cornea on slit lamp examination; and (3) increased cystine content of leukocytes. Identification of two mutations in CTNS, the only gene in which mutation is currently known to be cause cystinosis, is confirmatory.
    Genetic Counseling
    Cystinosis is inherited in an autosomal recessive manner. 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. Carrier testing for at-risk relatives and prenatal diagnosis for pregnancies at increased risk are possible when both disease-causing mutations have been identified in the family. For pregnancies at increased risk for nephropathic cystinosis, prenatal testing is also available biochemically, based on elevated cystine concentration in both chorionic villi and amniocytes.
    References

    Cystinosis, ocular nonnephropathic

    Summary from GeneReviews: Cystinosis Go to GeneReviews

    Disease Characteristics
    Nephropathic cystinosis in untreated children is characterized by renal tubular Fanconi syndrome, poor growth, hypophosphatemic rickets, impaired glomerular function, and accumulation of cystine crystals in almost all cells, leading to tissue destruction. The typical untreated child has short stature, light complexion, rickets, and photophobia. Growth failure is generally noticed between ages six and nine months; signs of renal tubular Fanconi syndrome (polyuria, polydipsia, dehydration, and acidosis) appear as early as age six months; corneal crystals can be present before age one year and are always present after age 16 months. Prior to the use of renal transplantation and cystine-depleting therapy, the life span in nephropathic cystinosis was no longer than ten years. With these therapies, affected individuals can survive at least into the mid-forties or fifties with satisfactory quality of life. Intermediate cystinosis is characterized by all the typical manifestations of nephropathic cystinosis, but onset is at a later age. Renal glomerular failure occurs in all untreated affected individuals, usually between ages 15 and 25 years. Non-nephropathic cystinosis is characterized only by photophobia resulting from corneal cystine crystal accumulation.
    Diagnosis Testing
    The diagnosis of cystinosis is established by documenting: (1) renal tubular Fanconi syndrome: increased urinary losses of essential nutrients including electrolytes (sodium, potassium, bicarbonate), minerals (calcium, phosphate, magnesium), glucose, amino acids, carnitine, and water; (2) typical cystine crystals in the cornea on slit lamp examination; and (3) increased cystine content of leukocytes. Identification of two mutations in CTNS, the only gene in which mutation is currently known to be cause cystinosis, is confirmatory.
    Genetic Counseling
    Cystinosis is inherited in an autosomal recessive manner. 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. Carrier testing for at-risk relatives and prenatal diagnosis for pregnancies at increased risk are possible when both disease-causing mutations have been identified in the family. For pregnancies at increased risk for nephropathic cystinosis, prenatal testing is also available biochemically, based on elevated cystine concentration in both chorionic villi and amniocytes.
    References

    Juvenile nephropathic cystinosis

    Summary from GeneReviews: Cystinosis Go to GeneReviews

    Disease Characteristics
    Nephropathic cystinosis in untreated children is characterized by renal tubular Fanconi syndrome, poor growth, hypophosphatemic rickets, impaired glomerular function, and accumulation of cystine crystals in almost all cells, leading to tissue destruction. The typical untreated child has short stature, light complexion, rickets, and photophobia. Growth failure is generally noticed between ages six and nine months; signs of renal tubular Fanconi syndrome (polyuria, polydipsia, dehydration, and acidosis) appear as early as age six months; corneal crystals can be present before age one year and are always present after age 16 months. Prior to the use of renal transplantation and cystine-depleting therapy, the life span in nephropathic cystinosis was no longer than ten years. With these therapies, affected individuals can survive at least into the mid-forties or fifties with satisfactory quality of life. Intermediate cystinosis is characterized by all the typical manifestations of nephropathic cystinosis, but onset is at a later age. Renal glomerular failure occurs in all untreated affected individuals, usually between ages 15 and 25 years. Non-nephropathic cystinosis is characterized only by photophobia resulting from corneal cystine crystal accumulation.
    Diagnosis Testing
    The diagnosis of cystinosis is established by documenting: (1) renal tubular Fanconi syndrome: increased urinary losses of essential nutrients including electrolytes (sodium, potassium, bicarbonate), minerals (calcium, phosphate, magnesium), glucose, amino acids, carnitine, and water; (2) typical cystine crystals in the cornea on slit lamp examination; and (3) increased cystine content of leukocytes. Identification of two mutations in CTNS, the only gene in which mutation is currently known to be cause cystinosis, is confirmatory.
    Genetic Counseling
    Cystinosis is inherited in an autosomal recessive manner. 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. Carrier testing for at-risk relatives and prenatal diagnosis for pregnancies at increased risk are possible when both disease-causing mutations have been identified in the family. For pregnancies at increased risk for nephropathic cystinosis, prenatal testing is also available biochemically, based on elevated cystine concentration in both chorionic villi and amniocytes.
    References
    • Lysosome, organism-specific biosystem (from KEGG)
      Lysosome, organism-specific biosystemLysosomes are membrane-delimited organelles in animal cells serving as the cell's main digestive compartment to which all sorts of macromolecules are delivered for degradation. They contain more than...
    • Lysosome, conserved biosystem (from KEGG)
      Lysosome, conserved biosystemLysosomes are membrane-delimited organelles in animal cells serving as the cell's main digestive compartment to which all sorts of macromolecules are delivered for degradation. They contain more than...

    Markers

    Homology

    Gene Ontology Provided by GOA

    Function Evidence Code Pubs
    L-cystine transmembrane transporter activity IMP
    Inferred from Mutant Phenotype
    more info
    PubMed 
    L-cystine transmembrane transporter activity NAS
    Non-traceable Author Statement
    more info
    PubMed 
    Process Evidence Code Pubs
    ATP metabolic process IMP
    Inferred from Mutant Phenotype
    more info
    PubMed 
    L-cystine transport IDA
    Inferred from Direct Assay
    more info
    PubMed 
    L-cystine transport IMP
    Inferred from Mutant Phenotype
    more info
    PubMed 
    L-cystine transport NAS
    Non-traceable Author Statement
    more info
    PubMed 
    adult walking behavior IEA
    Inferred from Electronic Annotation
    more info
     
    brain development IMP
    Inferred from Mutant Phenotype
    more info
    PubMed 
    cellular amino acid metabolic process NAS
    Non-traceable Author Statement
    more info
    PubMed 
    cognition IMP
    Inferred from Mutant Phenotype
    more info
    PubMed 
    compound eye corneal lens development IEA
    Inferred from Electronic Annotation
    more info
     
    glutathione metabolic process IMP
    Inferred from Mutant Phenotype
    more info
    PubMed 
    grooming behavior IEA
    Inferred from Electronic Annotation
    more info
     
    long-term memory IEA
    Inferred from Electronic Annotation
    more info
     
    visual learning IEA
    Inferred from Electronic Annotation
    more info
     
    Component Evidence Code Pubs
    NOT early endosome IDA
    Inferred from Direct Assay
    more info
    PubMed 
    integral to membrane IEA
    Inferred from Electronic Annotation
    more info
     
    late endosome IDA
    Inferred from Direct Assay
    more info
    PubMed 
    lysosomal membrane IDA
    Inferred from Direct Assay
    more info
    PubMed 
    lysosomal membrane NAS
    Non-traceable Author Statement
    more info
    PubMed 
    lysosome IDA
    Inferred from Direct Assay
    more info
    PubMed 
    plasma membrane IDA
    Inferred from Direct Assay
    more info
    PubMed 
    Preferred Names
    cystinosin
    Names
    cystinosin

    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_012489.1 RefSeqGene

      Range
      5001..31636
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    mRNA and Protein(s)

    1. NM_001031681.2NP_001026851.2  cystinosin isoform 1 precursor

      Status: REVIEWED

      Description
      Transcript Variant: This variant (1) represents the shorter transcript but encodes the longer isoform (1).
      Source sequence(s)
      AC027796, AC132942, BC032850, BX369529, CA454595, DB026195
      Consensus CDS
      CCDS32530.1
      UniProtKB/Swiss-Prot
      O60931
      Conserved Domains (2) summary
      TIGR00951
      Location:125357
      Blast Score: 733
      2A43; Lysosomal Cystine Transporter
      pfam04193
      Location:126186
      Blast Score: 167
      PQ-loop; PQ loop repeat
    2. NM_004937.2NP_004928.2  cystinosin isoform 2 precursor

      Status: REVIEWED

      Description
      Transcript Variant: This variant (2) differs in the 5' UTR, 3' UTR and 3' coding region, compared to variant 1, resulting in an isoform (2) with a distinct and shorter C-terminus, compared to isoform 1.
      Source sequence(s)
      AC027796, AC132942, BC032850, BX369529, DB026195
      Consensus CDS
      CCDS11031.1
      UniProtKB/Swiss-Prot
      O60931
      Conserved Domains (2) summary
      TIGR00951
      Location:125357
      Blast Score: 716
      2A43; Lysosomal Cystine Transporter
      pfam04193
      Location:126186
      Blast Score: 163
      PQ-loop; PQ loop repeat

    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_000017.10 Reference GRCh37.p10 Primary Assembly

      Range
      3539762..3566397
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate HuRef

    Genomic

    1. AC_000149.1 Alternate HuRef

      Range
      3431470..3458104
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate CHM1_1.0

    Genomic

    1. NC_018928.1 Alternate CHM1_1.0

      Range
      3528761..3555398
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

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