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10 sources retrieved · Most recent: April 2026 · Index updated 14 days ago
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ATP6V1E1
ATPase H+ transporting V1 subunit E1
Chromosome 22 · 22q11.21
NCBI Gene: 529Ensembl: ENSG00000131100.14HGNC: HGNC:857UniProt: P36543
95PubMed Papers
21Diseases
0Drugs
1Pathogenic Variants
FUNCTIONAL ROLE
Transporter
CLINICAL
OMIM Disease Gene
DATA QUALITY
✓ Experimental GO Evidence✓ Swiss-Prot Reviewed
ATPase bindingguanyl nucleotide exchange factor activator activitylysosomal proton-transporting V-type ATPase complexcellular response to amino acid stimulusneurodegenerative diseaseautosomal recessive cutis laxa type 2, classic typecutis laxaautosomal recessive cutis laxa type 2
✦AI Summary

ATP6V1E1 encodes the E1 subunit of the V1 domain of vacuolar H+-ATPase (V-ATPase), a multisubunit enzyme complex responsible for ATP hydrolysis and proton translocation 1. The V-ATPase complex acidifies intracellular compartments including lysosomes, endosomes, and secretory granules, and in some cell types localizes to the plasma membrane for extracellular acidification 1. ATP6V1E1 mutations cause autosomal-recessive cutis laxa with dysmorphic features and cardiopulmonary involvement; these mutations impair V-ATPase assembly and stability, disrupting protein glycosylation and vesicular trafficking while causing abnormal elastic and collagen fiber organization 2. ATP6V1E1 is classified as a congenital disorder of glycosylation (CDG) gene 34. Beyond cutis laxa, ATP6V1E1 dysfunction has been implicated in Alzheimer's disease pathogenesis, where downregulation correlates with amyloid-beta and tau pathology and impairs lysosomal acidification 567. Additionally, Mycobacterium tuberculosis exploits ATP6V1E1 phosphorylation to suppress lysosomal acidification and promote intracellular survival, suggesting ATP6V1E1 as a therapeutic target for tuberculosis 8.

Sources cited
1
ATP6V1E1 is a V1 subunit of V-ATPase that hydrolyzes ATP and translocates protons; V-ATPase acidifies intracellular compartments and extracellular environment
PMID: 32001091
2
ATP6V1E1 mutations cause autosomal-recessive cutis laxa with dysmorphic features and cardiopulmonary involvement; mutations impair V-ATPase assembly and stability, disrupting protein glycosylation and vesicular trafficking
PMID: 28065471
3
ATP6V1E1 is identified as a congenital disorder of glycosylation (CDG) gene
PMID: 28484880
4
ATP6V1E1 is associated with carbohydrate-linked congenital disorders of glycosylation presenting cardiac complications
PMID: 37239976
5
ATP6V1E1 is downregulated in Alzheimer's disease, correlates with amyloid-beta and tau pathology, and impairs lysosomal acidification
PMID: 41242442
6
ATP6V1E1 expression levels have diagnostic potential as a biomarker for Alzheimer's disease in blood and brain samples
PMID: 40187519
7
ATP6V1E1 is dysregulated in Alzheimer's disease and correlates with amyloid-beta and tau pathology; shows high diagnostic potential for AD
PMID: 37334737
8
ATP6V1E1 phosphorylation at Tyr56/57 suppresses lysosomal acidification; Mycobacterium tuberculosis exploits this pathway to promote intracellular survival
PMID: 41651829
Disease Associationsⓘ21
neurodegenerative diseaseOpen Targets
0.48Moderate
autosomal recessive cutis laxa type 2, classic typeOpen Targets
0.47Moderate
cutis laxaOpen Targets
0.46Moderate
autosomal recessive cutis laxa type 2Open Targets
0.38Weak
eye diseaseOpen Targets
0.32Weak
placenta praeviaOpen Targets
0.29Weak
sign or symptomOpen Targets
0.06Suggestive
chronic obstructive pulmonary diseaseOpen Targets
0.05Suggestive
hepatocellular carcinomaOpen Targets
0.04Suggestive
Phenotypic abnormalityOpen Targets
0.04Suggestive
pneumonitisOpen Targets
0.04Suggestive
infectionOpen Targets
0.04Suggestive
Alzheimer diseaseOpen Targets
0.03Suggestive
osteosarcomaOpen Targets
0.02Suggestive
cancerOpen Targets
0.02Suggestive
gliomaOpen Targets
0.02Suggestive
cat-eye syndromeOpen Targets
0.02Suggestive
breast cancerOpen Targets
0.02Suggestive
head and neck squamous cell carcinomaOpen Targets
0.02Suggestive
Autoimmune lymphoproliferative syndrome with recurrent viral infectionsOpen Targets
0.02Suggestive
Cutis laxa, autosomal recessive, 2CUniProt
Pathogenic Variants1
NM_001696.4(ATP6V1E1):c.634C>T (p.Arg212Trp)Pathogenic
Autosomal recessive cutis laxa type 2C|not provided|Cutis laxa
★★☆☆2025→ Residue 212
View on ClinVar ↗
Related Genes
ATP5F1AProtein interaction100%ATP5POProtein interaction100%ATP5F1CProtein interaction100%ATP6V1HProtein interaction100%ATP6V0A1Protein interaction100%ATP6V1G1Protein interaction100%
Tissue Expression6 tissues
Brain
100%
Heart
75%
Lung
42%
Ovary
25%
Bone Marrow
24%
Liver
21%
Gene Interaction Network
Click a node to explore
ATP6V1E1ATP5F1AATP5POATP5F1CATP6V1HATP6V0A1ATP6V1G1
PROTEIN STRUCTURE
Preparing viewer…
PDB6WLZ · 2.90 Å · EM
View on RCSB ↗
Constraintⓘ
LOEUFⓘ
0.85LoF Tolerant
pLIⓘ
0.00Tolerant
Observed/Expected LoF0.57 [0.39–0.85]
RankingsWhere ATP6V1E1 stands among ~20K protein-coding genes
  • #5,017of 20,598
    Most Researched95 · top quartile
  • #4,903of 5,498
    Most Pathogenic Variants1
  • #7,406of 17,882
    Most Constrained (LOEUF)0.85
Genes detectedATP6V1E1
Sources retrieved10 papers
Response time—
📄 Sources
10▼
1
Metabolic Cardiomyopathies and Cardiac Defects in Inherited Disorders of Carbohydrate Metabolism: A Systematic Review.
PMID: 37239976
Int J Mol Sci · 2023
1.00
2
What is new in CDG?
PMID: 28484880
J Inherit Metab Dis · 2017
0.90
3
ATP6V1E1 and NDUFB5 identified as potential biomarkers for Alzheimer's disease through integrative analysis.
PMID: 41242442
Int J Biol Macromol · 2025
0.80
4
Alterations in NFAT5 and ATP6V1E1 expression as potential diagnostic biomarkers in blood and brain for Alzheimer's disease: A study of gene overlap.
PMID: 40187519
Brain Res · 2025
0.70
5
Mycobacterium tuberculosis modulates phosphorylation of host ATP6V1E1 to promote intracellular survival.
PMID: 41651829
Nat Commun · 2026
0.60