Display Settings:

Format

Send to:

Choose Destination
We are sorry, but NCBI web applications do not support your browser and may not function properly. More information

    RYR1 ryanodine receptor 1 (skeletal) [ Homo sapiens (human) ]

    Gene ID: 6261, updated on 22-May-2013
    Official Symbol
    RYR1provided by HGNC
    Official Full Name
    ryanodine receptor 1 (skeletal)provided by HGNC
    Primary source
    HGNC:10483
    See related
    Ensembl:ENSG00000196218; HPRD:01618; MIM:180901; Vega:OTTHUMG00000182403
    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
    CCO; MHS; RYR; MHS1; RYDR; SKRR; RYR-1
    Summary
    This gene encodes a ryanodine receptor found in skeletal muscle. The encoded protein functions as a calcium release channel in the sarcoplasmic reticulum but also serves to connect the sarcoplasmic reticulum and transverse tubule. Mutations in this gene are associated with malignant hyperthermia susceptibility, central core disease, and minicore myopathy with external ophthalmoplegia. Alternatively spliced transcripts encoding different isoforms have been described. [provided by RefSeq, Jul 2008]
    Location :
    19q13.1
    Sequence :
    Chromosome: 19; NC_000019.9 (38924340..39078204)
    See RYR1 in Epigenomics, MapViewer

    Chromosome 19 - NC_000019.9Genomic Context describing neighboring genes Neighboring gene sprouty-related, EVH1 domain containing 3 Neighboring gene family with sequence similarity 98, member C Neighboring gene RAS guanyl releasing protein 4 Neighboring gene mitogen-activated protein kinase kinase kinase kinase 1 Neighboring gene eukaryotic translation initiation factor 3, subunit K

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

    Central core disease

    Summary from GeneReviews: Central Core Disease Go to GeneReviews

    Disease Characteristics
    Central core disease (CCD) is characterized by muscle weakness ranging from mild to severe. Most affected individuals have mild disease with symmetric proximal muscle weakness and variable involvement of facial and neck muscles. The extraocular muscles are often spared. Motor development is usually delayed, but in general, most affected individuals acquire independent ambulation. Life span is usually normal. Severe disease is early in onset with profound hypotonia often accompanied by poor fetal movement, spinal deformities, hip dislocation, joint contractures, poor suck, and respiratory insufficiency requiring assisted ventilation. The outcome ranges from death in infancy to survival beyond age five years. Typically the weakness in CCD is not progressive.
    Diagnosis Testing
    The diagnosis of CCD is based on clinical findings of muscle weakness, the histopathologic findings of characteristic cores on muscle biopsy, and molecular genetic testing. Most CCD is associated with mutations in RYR1, the gene encoding the ryanodine receptor 1. Molecular genetic testing of RYR1 is available clinically.
    Genetic Counseling
    Central core disease (CCD) is usually inherited in an autosomal dominant (AD) manner but can be inherited in an autosomal recessive (AR) manner. Most individuals diagnosed with AD central core disease have an affected parent or an asymptomatic parent who has a disease-causing mutation. The proportion of AD CCD caused by de novo mutations is unknown. Each child of an individual with AD CCD has a 50% chance of inheriting the mutation. The parents of a child with AR CCD are obligate heterozygotes and therefore carry one mutant allele. Heterozygotes (carriers) are often asymptomatic. At conception, each sib of an individual with AR CCD 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. Prenatal diagnosis for pregnancies at increased risk for AD or AR CCD is possible once the disease-causing mutation(s) has/have been identified in an affected family member.
    References

    Malignant hyperthermia

    Summary from GeneReviews: Malignant Hyperthermia Susceptibility Go to GeneReviews

    Disease Characteristics
    Malignant hyperthermia susceptibility (MHS) is a pharmacogenetic disorder of skeletal muscle calcium regulation associated with uncontrolled skeletal muscle hypermetabolism. Manifestations of malignant hyperthermia (MH) are precipitated by certain volatile anesthetics (i.e., halothane, isoflurane, sevoflurane, desflurane, enflurane), either alone or in conjunction with a depolarizing muscle relaxant (specifically, succinylcholine). The triggering substances release calcium stores from the sarcoplasmic reticulum and may promote entry of calcium from the myoplasm, causing contracture of skeletal muscles, glycogenolysis, and increased cellular metabolism, resulting in production of heat and excess lactate. Affected individuals experience: acidosis, hypercapnia, tachycardia, hyperthermia, muscle rigidity, compartment syndrome, rhabdomyolysis with subsequent increase in serum creatine kinase (CK) concentration, hyperkalemia with a risk for cardiac arrhythmia or even arrest, and myoglobinuria with a risk for renal failure. In nearly all cases, the first manifestations of MH (tachycardia and tachypnea) occur in the operating room; however, MH may also occur in the early postoperative period. There is mounting evidence that some affected individuals will also develop MH with exercise and/or on exposure to hot environments. Without proper and prompt treatment with dantrolene sodium, mortality is extremely high.
    Diagnosis Testing
    A clinical grading scale helps determine if a malignant hyperthermia (MH) episode has occurred. Contracture testing, the standard diagnostic test for MH since the mid-1970s, relies on the in vitro measurement of contracture response of biopsied muscle to graded concentrations of caffeine, the anesthetic halothane, and other calcium-releasing agents. To date, two genes predisposing to MHS have been identified; four additional loci have been mapped, but the genes have not been identified. MHS1 is associated with mutations in RYR1, encoding ryanodine receptor type 1; MHS5 is associated with mutations in CACNA1S, encoding a skeletal muscle calcium channel. Up to 70% of MHS is caused by mutations in RYR1 and about 1% results from mutations in CACNA1S. Molecular genetic testing for RYR1 and CACNA1S is available on a clinical basis.
    Genetic Counseling
    Malignant hyperthermia susceptibility (MHS) is inherited in an autosomal dominant manner. Most individuals diagnosed with MHS have a parent with MHS; however, the parent may not have experienced an episode of MH. The proportion of individuals with MHS caused by de novo mutations is unknown. Each child of an individual with MHS has a 50% chance of inheriting the disease-causing mutation. Although prenatal diagnosis for pregnancies at increased risk for MHS is possible, prenatal testing for pharmacogenetic conditions (like MHS) that have effective treatment and prevention is generally not offered or available.
    References

    Minicore myopathy with external ophthalmoplegia

    Summary from GeneReviews: Multiminicore Disease Go to GeneReviews

    Disease Characteristics
    Multiminicore disease (MmD) is broadly classified into four groups: Classic form (75% of individuals). Moderate form, with hand involvement (<10%). Antenatal form, with arthrogryposis multiplex congenita (<10%). Ophthalmoplegic form (<10%). Onset of the classic form is usually congenital or early in childhood with neonatal hypotonia, delayed motor development, axial muscle weakness, scoliosis, and significant respiratory involvement (often with secondary cardiac impairment). Spinal rigidity of varying severity is present.
    Diagnosis Testing
    The diagnosis of MmD is based on the presence of multiple "minicores" visible on muscle biopsy using oxidative stains, clinical findings of static or slowly progressive weakness, and absence of findings diagnostic of other neuromuscular disorders. Mutations in SEPN1 and RYR1 are known to cause 50% of MmD cases reported; further genetic heterogeneity is suggested, but no other candidate region or gene has been identified to date.
    Genetic Counseling
    MmD is most often inherited in an autosomal recessive manner. The occurrence of MMD in two generations in a few families has been reported, suggestive of autosomal dominant inheritance. Assuming autosomal recessive inheritance, each sib of an affected individual has at conception 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 testing for pregnancies at increased risk are possible if the disease-causing mutations in the family have been identified.
    References
    Protein Gene Interaction Pubs
    Tat, p14 tat HIV-1 Tat activates RyRs via a calcium- and calpain-mediated mechanism that upregulates DAT trafficking to the PM PubMed
    tat Neurons exposed to HIV-1 Tat induces calcium loss from the endoplasmic reticulum via ryanodine receptor (RyR) and increases the phosphorylated levels of PERK, eIF2a, and XBP1 PubMed

    Go to the HIV-1, Human Protein Interaction Database

    Products Interactant Other Gene Complex Source Pubs Description
    P21817 Q13698 CACNA1S    HPRD  PubMed  
    P21817 Calmodulin 1 CALM1    HPRD  PubMed  
    P21817 Q13555 CAMK2G    HPRD  PubMed  
    P21817 P62942 FKBP1A    HPRD  PubMed  
    P21817 Q86YM7 HOMER1    HPRD  PubMed  
    P21817 Q9NSB8 HOMER2    HPRD  PubMed  
    P21817 Q9NSC5 HOMER3    HPRD  PubMed  
    P21817 P17612 PRKACA    HPRD  PubMed  
    P21817 P14619 PRKG1    HPRD  PubMed  
    P21817 P21817 RYR1    HPRD  PubMed  
    P21817 Q92736 RYR2    HPRD  PubMed  
    P21817 P23297 S100A1    HPRD  PubMed  
    P21817 Q13061 TRDN    HPRD  PubMed  
    BioGRID:112173 BioGRID:108570 FKBP1A    BioGRID  PubMed Affinity Capture-Western; Reconstituted Complex 
    BioGRID:112173 BioGRID:114845 HOMER1    BioGRID  PubMed Affinity Capture-Western; Reconstituted Complex 
    BioGRID:112173 BioGRID:114844 HOMER2    BioGRID  PubMed Affinity Capture-Western; Reconstituted Complex 
    BioGRID:112173 BioGRID:114843 HOMER3    BioGRID  PubMed Affinity Capture-Western; Reconstituted Complex 
    BioGRID:112173 BioGRID:112174 RYR2    BioGRID  PubMed Affinity Capture-Western 
    BioGRID:112173 BioGRID:112179 S100A1    BioGRID  PubMed Reconstituted Complex 
    BioGRID:112173 BioGRID:115627 TRDN    BioGRID  PubMed Far Western; Protein-peptide; Reconstituted Complex 
    BioGRID:112173 BioGRID:113164 UBC    BioGRID  PubMed Affinity Capture-MS 
    • Calcium Regulation in the Cardiac Cell, organism-specific biosystem (from WikiPathways)
      Calcium Regulation in the Cardiac Cell, organism-specific biosystemCalcium is a common signaling mechanism, as once it enters the cytoplasm it exerts allosteric regulatory affects on many enzymes and proteins. Calcium can act in signal transduction after influx resu...
    • Calcium signaling pathway, organism-specific biosystem (from KEGG)
      Calcium signaling pathway, organism-specific biosystemCa2+ that enters the cell from the outside is a principal source of signal Ca2+. Entry of Ca2+ is driven by the presence of a large electrochemical gradient across the plasma membrane. Cells use this...
    • Calcium signaling pathway, conserved biosystem (from KEGG)
      Calcium signaling pathway, conserved biosystemCa2+ that enters the cell from the outside is a principal source of signal Ca2+. Entry of Ca2+ is driven by the presence of a large electrochemical gradient across the plasma membrane. Cells use this...
    • Circadian entrainment, organism-specific biosystem (from KEGG)
      Circadian entrainment, organism-specific biosystemCircadian entrainment is a fundamental property by which the period of the internal biological clock is entrained by recurring exogenous signals, such that the organism's endocrine and behavioral rhy...
    • Circadian entrainment, conserved biosystem (from KEGG)
      Circadian entrainment, conserved biosystemCircadian entrainment is a fundamental property by which the period of the internal biological clock is entrained by recurring exogenous signals, such that the organism's endocrine and behavioral rhy...
    • Ion channel transport, organism-specific biosystem (from REACTOME)
      Ion channel transport, organism-specific biosystemIon channels mediate the flow of ions across the plasma membrane of cells. They are integral membrane proteins, typically a multimer of proteins, which, when arranged in the membrane, create a pore f...
    • Long-term depression, organism-specific biosystem (from KEGG)
      Long-term depression, organism-specific biosystemCerebellar long-term depression (LTD), thought to be a molecular and cellular basis for cerebellar learning, is a process involving a decrease in the synaptic strength between parallel fiber (PF) and...
    • Long-term depression, conserved biosystem (from KEGG)
      Long-term depression, conserved biosystemCerebellar long-term depression (LTD), thought to be a molecular and cellular basis for cerebellar learning, is a process involving a decrease in the synaptic strength between parallel fiber (PF) and...
    • Myometrial Relaxation and Contraction Pathways, organism-specific biosystem (from WikiPathways)
      Myometrial Relaxation and Contraction Pathways, organism-specific biosystemThis pathway illustrates signaling networks implicated in uterine muscle contraction at labor and quiescence throughout gestation (pregnancy). The muscle of the uterus, responsible for contractile ac...
    • Stimuli-sensing channels, organism-specific biosystem (from REACTOME)
      Stimuli-sensing channels, organism-specific biosystemIon channels that mediate sensations such as pain, warmth, cold, taste pressure and vision. Channels that mediate these sensations include acid-sensing ion channels (ASICs) (Wang & Xu 2011, Qadri et ...
    • Transmembrane transport of small molecules, organism-specific biosystem (from REACTOME)
      Transmembrane transport of small molecules, organism-specific biosystem
      Transmembrane transport of small molecules

    Markers

    Homology

    Gene Ontology Provided by GOA

    Function Evidence Code Pubs
    calcium channel activity ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    calcium ion binding IEA
    Inferred from Electronic Annotation
    more info
     
    calcium-release channel activity TAS
    Traceable Author Statement
    more info
    PubMed 
    calmodulin binding ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    protease binding IEA
    Inferred from Electronic Annotation
    more info
     
    ryanodine-sensitive calcium-release channel activity ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    ryanodine-sensitive calcium-release channel activity TAS
    Traceable Author Statement
    more info
    PubMed 
    voltage-gated calcium channel activity ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    Process Evidence Code Pubs
    calcium ion transport ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    cellular response to caffeine ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    cytosolic calcium ion homeostasis ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    ion transmembrane transport TAS
    Traceable Author Statement
    more info
     
    muscle contraction ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    ossification involved in bone maturation ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    outflow tract morphogenesis ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    release of sequestered calcium ion into cytosol ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    release of sequestered calcium ion into cytosol by sarcoplasmic reticulum ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    response to caffeine ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    response to hypoxia IDA
    Inferred from Direct Assay
    more info
    PubMed 
    skeletal muscle fiber development ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    skin development ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    transmembrane transport TAS
    Traceable Author Statement
    more info
     
    Component Evidence Code Pubs
    I band IDA
    Inferred from Direct Assay
    more info
    PubMed 
    T-tubule IEA
    Inferred from Electronic Annotation
    more info
     
    cell cortex IDA
    Inferred from Direct Assay
    more info
    PubMed 
    cytoplasm IDA
    Inferred from Direct Assay
    more info
    PubMed 
    integral to plasma membrane TAS
    Traceable Author Statement
    more info
    PubMed 
    junctional membrane complex IEA
    Inferred from Electronic Annotation
    more info
     
    junctional sarcoplasmic reticulum membrane TAS
    Traceable Author Statement
    more info
    PubMed 
    plasma membrane IDA
    Inferred from Direct Assay
    more info
    PubMed 
    sarcoplasmic reticulum ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    sarcoplasmic reticulum membrane ISS
    Inferred from Sequence or Structural Similarity
    more info
     
    sarcoplasmic reticulum membrane TAS
    Traceable Author Statement
    more info
     
    smooth endoplasmic reticulum TAS
    Traceable Author Statement
    more info
    PubMed 
    terminal cisterna ISS
    Inferred from Sequence or Structural Similarity
    more info
    PubMed 
    Preferred Names
    ryanodine receptor 1
    Names
    ryanodine receptor 1
    ryanodine receptor type1
    type 1 ryanodine receptor
    central core disease of muscle
    type 1-like ryanodine receptor
    skeletal muscle ryanodine receptor
    skeletal muscle calcium release channel
    sarcoplasmic reticulum calcium release channel

    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_008866.1 RefSeqGene

      Range
      5001..158865
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    mRNA and Protein(s)

    1. NM_000540.2NP_000531.2  ryanodine receptor 1 isoform 1

      Status: REVIEWED

      Description
      Transcript Variant: This variant (1) encodes the longer isoform (1).
      Source sequence(s)
      AC005933, AC011469, AC067969
      Consensus CDS
      CCDS33011.1
      UniProtKB/Swiss-Prot
      P21817
      Related
      ENSP00000352608, OTTHUMP00000269480, ENST00000359596, OTTHUMT00000462137
      Conserved Domains (11) summary
      smart00449
      Location:10841206
      Blast Score: 329
      SPRY; Domain in SPla and the RYanodine Receptor.
      pfam06459
      Location:44464671
      Blast Score: 382
      RR_TM4-6; Ryanodine Receptor TM 4-6
      pfam08454
      Location:38703992
      Blast Score: 304
      RIH_assoc; RyR and IP3R Homology associated
      smart00472
      Location:210263
      Blast Score: 118
      MIR; Domain in ryanodine and inositol trisphosphate receptors and protein O-mannosyltransferases
      pfam00520
      Location:47914901
      Blast Score: 103
      Ion_trans; Ion transport protein
      pfam00622
      Location:14291569
      Blast Score: 273
      SPRY; SPRY domain
      pfam01365
      Location:440643
      Blast Score: 627
      RYDR_ITPR; RIH domain
      pfam02026
      Location:849943
      Blast Score: 403
      RyR; RyR domain
      pfam02815
      Location:211389
      Blast Score: 552
      MIR; MIR domain
      pfam08709
      Location:8206
      Blast Score: 626
      Ins145_P3_rec; Inositol 1,4,5-trisphosphate/ryanodine receptor
      pfam13833
      Location:40834133
      Blast Score: 103
      EF_hand_6; EF-hand domain pair
    2. NM_001042723.1NP_001036188.1  ryanodine receptor 1 isoform 2

      Status: REVIEWED

      Description
      Transcript Variant: This variant (2) lacks an alternate in-frame exon, compared to variant 1, resulting in a shorter protein (isoform 2).
      Source sequence(s)
      AC005933, AC011469, BI000785, BP231595, J05200
      Consensus CDS
      CCDS42563.1
      UniProtKB/Swiss-Prot
      P21817
      Related
      ENSP00000347667, OTTHUMP00000269192, ENST00000355481, OTTHUMT00000461017
      Conserved Domains (11) summary
      smart00449
      Location:10841206
      Blast Score: 329
      SPRY; Domain in SPla and the RYanodine Receptor.
      pfam06459
      Location:44414666
      Blast Score: 382
      RR_TM4-6; Ryanodine Receptor TM 4-6
      pfam08454
      Location:38653987
      Blast Score: 304
      RIH_assoc; RyR and IP3R Homology associated
      smart00472
      Location:210263
      Blast Score: 118
      MIR; Domain in ryanodine and inositol trisphosphate receptors and protein O-mannosyltransferases
      pfam00520
      Location:47864896
      Blast Score: 103
      Ion_trans; Ion transport protein
      pfam00622
      Location:14291569
      Blast Score: 273
      SPRY; SPRY domain
      pfam01365
      Location:440643
      Blast Score: 627
      RYDR_ITPR; RIH domain
      pfam02026
      Location:849943
      Blast Score: 403
      RyR; RyR domain
      pfam02815
      Location:211389
      Blast Score: 552
      MIR; MIR domain
      pfam08709
      Location:8206
      Blast Score: 626
      Ins145_P3_rec; Inositol 1,4,5-trisphosphate/ryanodine receptor
      pfam13833
      Location:40784128
      Blast Score: 103
      EF_hand_6; EF-hand domain pair

    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_000019.9 Reference GRCh37.p10 Primary Assembly

      Range
      38924340..39078204
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate HuRef

    Genomic

    1. AC_000151.1 Alternate HuRef

      Range
      35371495..35526332
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate CHM1_1.0

    Genomic

    1. NC_018930.1 Alternate CHM1_1.0

      Range
      39073097..39227009
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

      Supplemental Content

      Recent activity

      Your browsing activity is empty.

      Activity recording is turned off.

      Turn recording back on

      See more...