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GeneE
26 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
β“˜GeneE is for informational purposes only. It is not a substitute for professional medical advice, diagnosis, or treatment.
ATP7B
ATPase copper transporting beta
Chromosome 13 Β· 13q14.3
NCBI Gene: 540Ensembl: ENSG00000123191.18HGNC: HGNC:870UniProt: A0A669KB88
388PubMed Papers
21Diseases
0Drugs
861Pathogenic Variants
FUNCTIONAL ROLE
Transporter
RESEARCH IMPACT
Highly StudiedVariant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
cellular detoxification of copper ionresponse to copper ioncytoplasmic vesicleperinuclear region of cytoplasmWilson diseasegenetic disorderDystoniaspastic ataxia
✦AI Summary

ATP7B encodes a copper-transporting ATPase essential for hepatic copper homeostasis 1. The protein functions as a P-type ATPase that exports copper from hepatocytes into bile, facilitating copper detoxification and preventing toxic accumulation 2. ATP7B localizes to the trans-Golgi network and late endosomes, where it mediates ATP-dependent copper transport across membranes 2. Mutations in ATP7B cause Wilson disease, an autosomal recessive disorder of copper metabolism characterized by progressive copper accumulation in the liver, brain, and other tissues 1. Over 700 disease-associated variants have been identified, with diverse functional consequences 3. While some variants completely abolish transport activity, others retain partial function or normal phosphor-intermediate formation but impaired copper translocation 2. Notably, ATP7B variants exhibit variable effects on protein stability and subcellular localization, contributing to the broad clinical phenotype of Wilson disease 2. Genotype-phenotype correlation studies demonstrate that patients carrying loss-of-function variants show worse long-term survival during chelation therapy for chr13 liver disease 4. Early genetic diagnosis through ATP7B sequencing is clinically significant for identifying affected individuals and enabling timely intervention to prevent progressive hepatoneurologic deterioration 5.

Sources cited
1
ATP7B mutation causes Wilson disease with impaired biliary copper excretion and copper accumulation
PMID: 17390257
2
ATP7B variants have diverse functional properties including loss of transport activity, mislocalization, and reduced protein stability
PMID: 22240481
3
Early genetic diagnosis of ATP7B variants facilitates early intervention and individualized treatment
PMID: 27022412
4
Patients with loss-of-function ATP7B variants have worse transplant-free survival on chelation therapy
PMID: 36096368
5
Over 700 disease-associated ATP7B variants exist with varying degrees of residual copper transport activity
PMID: 40661833
Disease Associationsβ“˜21
Wilson diseaseOpen Targets
0.87Strong
genetic disorderOpen Targets
0.55Moderate
DystoniaOpen Targets
0.50Moderate
developmental and epileptic encephalopathy 93Open Targets
0.34Weak
genetic developmental and epileptic encephalopathyOpen Targets
0.34Weak
hearing loss, autosomal recessiveOpen Targets
0.34Weak
intellectual disability, Wolff typeOpen Targets
0.34Weak
spastic ataxiaOpen Targets
0.34Weak
Epileptic encephalopathyOpen Targets
0.29Weak
Abruptio PlacentaeOpen Targets
0.28Weak
frozen shoulderOpen Targets
0.28Weak
Abnormality of metabolism/homeostasisOpen Targets
0.27Weak
Hand tremorOpen Targets
0.27Weak
Kayser-Fleischer ringOpen Targets
0.27Weak
asthmaOpen Targets
0.20Weak
neuropathyOpen Targets
0.20Weak
optic neuritisOpen Targets
0.20Weak
neoplasmOpen Targets
0.13Weak
anhaptoglobinemiaOpen Targets
0.12Weak
TendinopathyOpen Targets
0.10Suggestive
Wilson diseaseUniProt
Pathogenic Variants861
NM_000053.4(ATP7B):c.1934T>G (p.Met645Arg)Pathogenic
Wilson disease|not provided|Inborn genetic diseases|ATP7B-related disorder
β˜…β˜…β˜†β˜†2026β†’ Residue 645
NM_000053.4(ATP7B):c.3191A>C (p.Glu1064Ala)Pathogenic
Wilson disease|not provided|Inborn genetic diseases
β˜…β˜…β˜†β˜†2026β†’ Residue 1064
NM_000053.4(ATP7B):c.1870-2A>GLikely pathogenic
Wilson disease
β˜…β˜…β˜†β˜†2026
NM_000053.4(ATP7B):c.3809A>G (p.Asn1270Ser)Pathogenic
Wilson disease|not provided|Inborn genetic diseases|ATP7B-related disorder
β˜…β˜…β˜†β˜†2026β†’ Residue 1270
NM_000053.4(ATP7B):c.3443T>C (p.Ile1148Thr)Pathogenic
Wilson disease|not provided|Inborn genetic diseases
β˜…β˜…β˜†β˜†2026β†’ Residue 1148
NM_000053.4(ATP7B):c.2605G>A (p.Gly869Arg)Pathogenic
Wilson disease|not provided|Inborn genetic diseases|ATP7B-related disorder
β˜…β˜…β˜†β˜†2026β†’ Residue 869
NM_000053.4(ATP7B):c.2804C>T (p.Thr935Met)Pathogenic
Wilson disease|not provided|ATP7B-related disorder
β˜…β˜…β˜†β˜†2026β†’ Residue 935
NM_000053.4(ATP7B):c.3402del (p.Ala1135fs)Pathogenic
Wilson disease|not provided|Inborn genetic diseases
β˜…β˜…β˜†β˜†2026β†’ Residue 1135
NM_000053.4(ATP7B):c.2755C>T (p.Arg919Trp)Pathogenic
Wilson disease|not provided|ATP7B-related disorder
β˜…β˜…β˜†β˜†2026β†’ Residue 919
NM_000053.4(ATP7B):c.2975C>T (p.Pro992Leu)Pathogenic
Wilson disease|not provided|Inborn genetic diseases
β˜…β˜…β˜†β˜†2026β†’ Residue 992
NM_000053.4(ATP7B):c.3207C>A (p.His1069Gln)Pathogenic
Wilson disease|not provided|Inborn genetic diseases
β˜…β˜…β˜†β˜†2026β†’ Residue 1069
NM_000053.4(ATP7B):c.2827G>A (p.Gly943Ser)Pathogenic
Wilson disease|not provided
β˜…β˜…β˜†β˜†2026β†’ Residue 943
NM_000053.4(ATP7B):c.3060+5G>TPathogenic
Wilson disease
β˜…β˜…β˜†β˜†2026
NM_000053.4(ATP7B):c.122A>G (p.Asn41Ser)Pathogenic
Wilson disease|not provided|Inborn genetic diseases|ATP7B-related disorder
β˜…β˜…β˜†β˜†2026β†’ Residue 41
NM_000053.4(ATP7B):c.3089G>T (p.Gly1030Val)Likely pathogenic
Wilson disease
β˜…β˜…β˜†β˜†2026β†’ Residue 1030
NM_000053.4(ATP7B):c.2333G>T (p.Arg778Leu)Pathogenic
Wilson disease|not provided|Inborn genetic diseases|ATP7B-related disorder
β˜…β˜…β˜†β˜†2026β†’ Residue 778
NM_000053.4(ATP7B):c.2906G>A (p.Arg969Gln)Pathogenic
Wilson disease|not provided|ATP7B-related disorder|Inborn genetic diseases
β˜…β˜…β˜†β˜†2026β†’ Residue 969
NM_000053.4(ATP7B):c.2930C>T (p.Thr977Met)Pathogenic
Wilson disease|not provided|Inborn genetic diseases|ATP7B-related disorder|Hearing loss, autosomal recessive 109
β˜…β˜…β˜†β˜†2026β†’ Residue 977
NM_000053.4(ATP7B):c.3243+1G>APathogenic
Wilson disease|not provided
β˜…β˜…β˜†β˜†2026
NM_000053.4(ATP7B):c.3960G>C (p.Arg1320Ser)Pathogenic
Wilson disease
β˜…β˜…β˜†β˜†2026β†’ Residue 1320
View on ClinVar β†—
Related Genes
CCSProtein interaction99%CPProtein interaction98%COMMD1Protein interaction98%GLRXProtein interaction86%ATP13A3Protein interaction83%MT1EProtein interaction82%
Tissue Expression6 tissues
Liver
100%
Bone Marrow
75%
Heart
23%
Ovary
20%
Lung
11%
Brain
7%
Gene Interaction Network
Click a node to explore
ATP7BCCSCPCOMMD1GLRXATP13A3MT1E
PROTEIN STRUCTURE
Preparing viewer…
PDB6A71 Β· 1.60 Γ… Β· X-ray
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
1.06LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.91 [0.78–1.06]
RankingsWhere ATP7B stands among ~20K protein-coding genes
  • #766of 20,598
    Most Researched388 Β· top 5%
  • #48of 5,498
    Most Pathogenic Variants861 Β· top 1%
  • #10,696of 17,882
    Most Constrained (LOEUF)1.06
Genes detectedATP7B
Sources retrieved26 papers
Response timeβ€”
πŸ“„ Sources
26β–Ό
1
Spectrum and Classification of ATP7B Variants in a Large Cohort of Chinese Patients with Wilson's Disease Guides Genetic Diagnosis.
PMID: 27022412
Theranostics Β· 2016
1.00
2
A structural model of the copper ATPase ATP7B to facilitate analysis of Wilson disease-causing mutations and studies of the transport mechanism.
PMID: 22692182
Metallomics Β· 2012
0.90
3
Diverse functional properties of Wilson disease ATP7B variants.
PMID: 22240481
Gastroenterology Β· 2012
0.80
4
Pathogenicity of Intronic and Synonymous Variants of ATP7B in Wilson Disease.
PMID: 36343861
J Mol Diagn Β· 2023
0.70
5
Wilson disease.
PMID: 20955957
Best Pract Res Clin Gastroenterol Β· 2010
0.64