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    HMGCL 3-hydroxymethyl-3-methylglutaryl-CoA lyase [ Homo sapiens (human) ]

    Gene ID: 3155, updated on 14-May-2013
    Official Symbol
    HMGCLprovided by HGNC
    Official Full Name
    3-hydroxymethyl-3-methylglutaryl-CoA lyaseprovided by HGNC
    Primary source
    HGNC:5005
    See related
    Ensembl:ENSG00000117305; HPRD:02003; MIM:613898; Vega:OTTHUMG00000002963
    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
    HL
    Summary
    The protein encoded by this gene belongs to the HMG-CoA lyase family. It is a mitochondrial enzyme that catalyzes the final step of leucine degradation and plays a key role in ketone body formation. Mutations in this gene are associated with HMG-CoA lyase deficiency. Alternatively spliced transcript variants encoding different isoforms have been found for this gene. [provided by RefSeq, Oct 2009]
    Location :
    1p36.1-p35
    Sequence :
    Chromosome: 1; NC_000001.10 (24128367..24151949, complement)
    See HMGCL in Epigenomics, MapViewer

    Chromosome 1 - NC_000001.10Genomic Context describing neighboring genes Neighboring gene lysophospholipase II Neighboring gene UDP-galactose-4-epimerase Neighboring gene fucosidase, alpha-L- 1, tissue Neighboring gene cannabinoid receptor 2 (macrophage) Neighboring gene BTB (POZ) domain containing 6 pseudogene

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

    Deficiency of hydroxymethylglutaryl-CoA lyase

    Summary from GeneReviews: Organic Acidemias Overview Go to GeneReviews

    Disease Characteristics
    The term "organic acidemia" or "organic aciduria" (OA) applies to a group of disorders characterized by the excretion of non-amino organic acids in urine. Most organic acidemias result from dysfunction of a specific step in amino acid catabolism, usually the result of deficient enzyme activity. The majority of the classic organic acid disorders are caused by abnormal amino acid catabolism of branched-chain amino acids or lysine. They include maple syrup urine disease (MSUD), propionic acidemia, methylmalonic acidemia (MMA), methylmalonic aciduria and homocystinuria, isovaleric acidemia, biotin-unresponsive 3-methylcrotonyl-CoA carboxylase deficiency, 3-hydroxy-3-methylglutaryl-CoA (HMG-CoA) lyase deficiency, ketothiolase deficiency, and glutaricacidemia type I (GA I). A neonate affected with an OA is usually well at birth and for the first few days of life. The usual clinical presentation is that of toxic encephalopathy and includes vomiting, poor feeding, neurologic symptoms such as seizures and abnormal tone, and lethargy progressing to coma. Outcome is enhanced by diagnosis and treatment in the first ten days of life. In the older child or adolescent, variant forms of the OAs can present as loss of intellectual function, ataxia or other focal neurologic signs, Reye syndrome, recurrent ketoacidosis, or psychiatric symptoms.
    Diagnosis Testing
    Clinical laboratory findings that suggest an organic acidemia include acidosis, ketosis, hyperammonemia, abnormal liver function tests, hypoglycemia, and neutropenia. First-line diagnosis in the organic acidemias is urine organic acid analysis using gas chromatography with mass spectrometry (GC/MS), utilizing a capillary column. The urinary organic acid profile is nearly always abnormal in the face of acute illness with decompensation; however, in some disorders diagnostic analytes may be present only in small or barely detectable amounts when the affected individual is not acutely ill. Depending on the specific disorder, plasma amino acid analysis using a quantitative method such as column chromatography, high-performance liquid chromatography (HPLC), or GC/MS can also be helpful. A plasma or serum acylcarnitine profile can also provide a rapid clue to the diagnosis. Urine acylcarnitine profiling is more complex and interpretation can be difficult. Confirmatory testing involves assay of the activity of the deficient enzyme in lymphocytes or cultured fibroblasts and/or molecular genetic testing.
    Genetic Counseling
    The organic acidemias considered in this overview are inherited in an autosomal recessive manner. At conception, each sib of a proband 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 family members is possible if the disease-causing mutations in the family are known. Prenatal diagnosis for pregnancies at increased risk varies by disorder and may include measurement of analytes in amniotic fluid, measurement of enzyme activity, or molecular genetic testing in cells obtained by CVS or amniocentesis.
    References
    Products Interactant Other Gene Complex Source Pubs Description
    P35914 Q9GZW8 MS4A7    HPRD  PubMed  
    BioGRID:109398 BioGRID:123586 ADAMTS10    BioGRID  PubMed Two-hybrid 
    BioGRID:109398 BioGRID:116812 ARL6IP1    BioGRID  PubMed Two-hybrid 
    BioGRID:109398 BioGRID:109534 DNAJA1    BioGRID  PubMed Two-hybrid 
    BioGRID:109398 BioGRID:109214 GTF2B    BioGRID  PubMed Two-hybrid 
    BioGRID:109398 BioGRID:109514 HES1    BioGRID  PubMed Two-hybrid 
    BioGRID:109398 BioGRID:121808 MS4A7    BioGRID  PubMed Two-hybrid 
    BioGRID:109398 BioGRID:111788 PEX5    BioGRID  PubMed Protein-peptide 
    BioGRID:109398 BioGRID:120790 RNF126    BioGRID  PubMed Two-hybrid 

    Markers

    Potential readthrough

    Included gene: GALE

    Homology

    Gene Ontology Provided by GOA

    Function Evidence Code Pubs
    carboxylic acid binding IEA
    Inferred from Electronic Annotation
    more info
     
    fatty-acyl-CoA binding IEA
    Inferred from Electronic Annotation
    more info
     
    hydroxymethylglutaryl-CoA lyase activity IDA
    Inferred from Direct Assay
    more info
     
    hydroxymethylglutaryl-CoA lyase activity TAS
    Traceable Author Statement
    more info
     
    magnesium ion binding IDA
    Inferred from Direct Assay
    more info
    PubMed 
    manganese ion binding IDA
    Inferred from Direct Assay
    more info
    PubMed 
    metal ion binding IDA
    Inferred from Direct Assay
    more info
    PubMed 
    protein homodimerization activity IDA
    Inferred from Direct Assay
    more info
    PubMed 
    receptor binding IPI
    Inferred from Physical Interaction
    more info
     
    Process Evidence Code Pubs
    acyl-CoA metabolic process IEA
    Inferred from Electronic Annotation
    more info
     
    cellular ketone body metabolic process TAS
    Traceable Author Statement
    more info
     
    cellular lipid metabolic process TAS
    Traceable Author Statement
    more info
     
    embryo development IEA
    Inferred from Electronic Annotation
    more info
     
    ketone body biosynthetic process IDA
    Inferred from Direct Assay
    more info
     
    ketone body biosynthetic process TAS
    Traceable Author Statement
    more info
     
    leucine catabolic process NAS
    Non-traceable Author Statement
    more info
    PubMed 
    liver development IEA
    Inferred from Electronic Annotation
    more info
     
    mitochondrion organization IEA
    Inferred from Electronic Annotation
    more info
     
    protein tetramerization IDA
    Inferred from Direct Assay
    more info
    PubMed 
    response to fatty acid IEA
    Inferred from Electronic Annotation
    more info
     
    response to nutrient IEA
    Inferred from Electronic Annotation
    more info
     
    response to starvation IEA
    Inferred from Electronic Annotation
    more info
     
    small molecule metabolic process TAS
    Traceable Author Statement
    more info
     
    Component Evidence Code Pubs
    mitochondrial inner membrane IEA
    Inferred from Electronic Annotation
    more info
     
    mitochondrial matrix NAS
    Non-traceable Author Statement
    more info
    PubMed 
    mitochondrial matrix TAS
    Traceable Author Statement
    more info
     
    mitochondrion IDA
    Inferred from Direct Assay
    more info
    PubMed 
    peroxisome IDA
    Inferred from Direct Assay
    more info
    PubMed 
    Preferred Names
    hydroxymethylglutaryl-CoA lyase, mitochondrial
    Names
    hydroxymethylglutaryl-CoA lyase, mitochondrial
    HMG-CoA lyase
    hydroxymethylglutaricaciduria
    3-hydroxy-3-methylglutaryl-CoA lyase
    3-hydroxy-3-methylglutarate-CoA lyase
    3-hydroxymethyl-3-methylglutaryl-Coenzyme A lyase
    mitochondrial 3-hydroxy-3-methylglutaryl-CoA lyase
    NP_000182.2
    NP_001159531.1

    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_013061.1 RefSeqGene

      Range
      5001..28583
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    mRNA and Protein(s)

    1. NM_000191.2NP_000182.2  hydroxymethylglutaryl-CoA lyase, mitochondrial isoform 1 precursor

      Status: REVIEWED

      Description
      Transcript Variant: This variant (1) encodes the longer isoform (1).
      Source sequence(s)
      AI202943, AL031295, AL590728
      Consensus CDS
      CCDS243.1
      UniProtKB/Swiss-Prot
      P35914
      UniProtKB/TrEMBL
      Q6IBC0
      Related
      ENSP00000363614, OTTHUMP00000003005, ENST00000374490, OTTHUMT00000008253
      Conserved Domains (2) summary
      COG0119
      Location:31313
      Blast Score: 505
      LeuA; Isopropylmalate/homocitrate/citramalate synthases [Amino acid transport and metabolism]
      cd07938
      Location:35308
      Blast Score: 1209
      DRE_TIM_HMGL; 3-hydroxy-3-methylglutaryl-CoA lyase, catalytic TIM barrel domain
    2. NM_001166059.1NP_001159531.1  hydroxymethylglutaryl-CoA lyase, mitochondrial isoform 2 precursor

      Status: REVIEWED

      Description
      Transcript Variant: This variant is missing two consecutive in-frame coding exons compared to variant 1, resulting in a shorter isoform (2) lacking an internal protein segment compared to isoform 1.
      Source sequence(s)
      BC010570, BM981610, CA306929, DA425822, L07033
      Consensus CDS
      CCDS53279.1
      UniProtKB/Swiss-Prot
      P35914
      Related
      ENSP00000389281, OTTHUMP00000223529, ENST00000436439, OTTHUMT00000372570
      Conserved Domains (2) summary
      PLN02746
      Location:115254
      Blast Score: 599
      PLN02746; hydroxymethylglutaryl-CoA lyase
      cl07974
      Location:35117
      Blast Score: 411
      DRE_TIM_metallolyase; DRE-TIM metallolyase superfamily

    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_000001.10 Reference GRCh37.p10 Primary Assembly

      Range
      24128367..24151949, complement
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate HuRef

    Genomic

    1. AC_000133.1 Alternate HuRef

      Range
      22379944..22403530, complement
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

    Alternate CHM1_1.0

    Genomic

    1. NC_018912.1 Alternate CHM1_1.0

      Range
      24342444..24366107, complement
      Download
      GenBank, FASTA, Sequence Viewer (Graphics)

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