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25 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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ATP6AP2
ATPase H+ transporting accessory protein 2
Chromosome X Β· Xp11.4
NCBI Gene: 10159Ensembl: ENSG00000182220.16HGNC: HGNC:18305UniProt: O75787
182PubMed Papers
23Diseases
0Drugs
6Pathogenic Variants
FUNCTIONAL ROLE
Highly ConstrainedReceptor
RESEARCH IMPACT
Trending
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
protein bindingmembranelysosomecentral nervous system maturationsyndromic X-linked intellectual disability Hedera typeX-linked intellectual disability, Hedera typecongenital disorder of glycosylation type IIX-linked intellectual disability - epilepsy
✦AI Summary

ATP6AP2 is a multifunctional X-linked gene encoding a protein with dual roles in cellular homeostasis. Functionally, ATP6AP2 serves as a (pro)renin receptor and as an accessory subunit of the V-type ATPase (V-ATPase) complex 1. Through its V-ATPase function, ATP6AP2 mediates lysosomal acidification and endo-lysosomal system function, critical for protein degradation and autophagy 2. As a renin receptor, ATP6AP2 participates in local renin-angiotensin system signaling within tissues including the brain, pancreas, and cardiovascular system 134. Clinically, ATP6AP2 mutations cause X-linked neurological disorders. Splicing variants cause X-linked intellectual disability (Hedera type) and X-linked parkinsonism with spasticity, characterized by developmental delay, epilepsy, and abnormal glycosylation 56. Missense mutations lead to congenital disorder of glycosylation 2R with hepatic involvement 5. ATP6AP2 dysfunction impairs lysosomal biogenesis and cellular viability; restoring lysosomal function can ameliorate defects in affected cells 23. Sex-linked inheritance and X-chromosome X complicate phenotypic presentation, with heterozygous females showing milder manifestations 57. The gene's role in maintaining cellular homeostasis suggests therapeutic potential through targeting lysosomal pathways.

Sources cited
1
ATP6AP2 functions as (pro)renin receptor and V-ATPase subunit involved in brain renin-angiotensin system
PMID: 30323117
2
ATP6AP2 involved in lysosomal acidification; decreased expression in Nasu-Hakola disease with lysosomal dysfunction
PMID: 37115208
3
ATP6AP2 robustly expressed in pancreatic Ξ² cells; knockdown decreases cell viability and increases apoptosis
PMID: 37286698
4
ATP6AP2 splicing variants cause X-linked intellectual disability Hedera type and parkinsonism with spasticity; associated with abnormal glycosylation; first affected female described with milder phenotype
PMID: 41131679
5
ATP6AP2 mutations associated with X-linked epilepsy and developmental and epileptic encephalopathies
PMID: 38612920
6
ATP6AP2 (PRR) expression modulated in macrophages during atherosclerotic inflammation
PMID: 40654251
7
ATP6AP2 involved in autophagy-mediated sex differences in disease pathology
PMID: 32264724
8
ATP6AP2 activated during zebrafish heart regeneration
PMID: 40253397
Disease Associationsβ“˜23
syndromic X-linked intellectual disability Hedera typeOpen Targets
0.70Moderate
X-linked intellectual disability, Hedera typeOpen Targets
0.66Moderate
congenital disorder of glycosylation type IIOpen Targets
0.61Moderate
X-linked intellectual disability - epilepsyOpen Targets
0.59Moderate
neurodegenerative diseaseOpen Targets
0.54Moderate
X-linked parkinsonism-spasticity syndromeOpen Targets
0.52Moderate
ATP6AP2-related disorderOpen Targets
0.41Moderate
genetic disorderOpen Targets
0.19Weak
neoplasmOpen Targets
0.11Weak
dilated cardiomyopathyOpen Targets
0.10Suggestive
breast cancerOpen Targets
0.09Suggestive
diabetic nephropathyOpen Targets
0.08Suggestive
triple-negative breast cancerOpen Targets
0.08Suggestive
colorectal carcinomaOpen Targets
0.08Suggestive
melanomaOpen Targets
0.08Suggestive
neuroendocrine neoplasmOpen Targets
0.08Suggestive
fetal growth restrictionOpen Targets
0.08Suggestive
bladder transitional cell carcinomaOpen Targets
0.08Suggestive
insulinomaOpen Targets
0.08Suggestive
infectionOpen Targets
0.07Suggestive
Congenital disorder of glycosylation 2RUniProt
Intellectual developmental disorder, X-linked, syndromic, Hedera typeUniProt
Parkinsonism with spasticity, X-linkedUniProt
Pathogenic Variants6
NM_005765.3(ATP6AP2):c.321C>T (p.Asp107=)Pathogenic
Syndromic X-linked intellectual disability Hedera type
β˜…β˜…β˜†β˜†2023β†’ Residue 107
NM_005765.3(ATP6AP2):c.284C>A (p.Ser95Ter)Pathogenic
Syndromic X-linked intellectual disability Hedera type
β˜…β˜†β˜†β˜†2024β†’ Residue 95
NM_005765.3(ATP6AP2):c.628G>T (p.Asp210Tyr)Likely pathogenic
Syndromic X-linked intellectual disability Hedera type
β˜…β˜†β˜†β˜†2020β†’ Residue 210
NM_005765.3(ATP6AP2):c.301-11_301-10delPathogenic
Syndromic X-linked intellectual disability Hedera type
β˜†β˜†β˜†β˜†2021
NM_005765.3(ATP6AP2):c.293T>C (p.Leu98Ser)Pathogenic
Congenital disorder of glycosylation, type IIr
β˜†β˜†β˜†β˜†2020β†’ Residue 98
NM_005765.3(ATP6AP2):c.345C>T (p.Ser115=)Pathogenic
X-linked parkinsonism-spasticity syndrome
β˜†β˜†β˜†β˜†2013β†’ Residue 115
View on ClinVar β†—
Related Genes
AGTR1Protein interaction100%RENProtein interaction100%ATP6V0D2Protein interaction100%ATP6V1C2Protein interaction100%ATP5MC1Protein interaction100%ATP5MC2Protein interaction100%
Tissue Expression6 tissues
Bone Marrow
100%
Brain
91%
Lung
42%
Heart
40%
Ovary
36%
Liver
34%
Gene Interaction Network
Click a node to explore
ATP6AP2AGTR1RENATP6V0D2ATP6V1C2ATP5MC1ATP5MC2
PROTEIN STRUCTURE
Preparing viewer…
PDB3LC8 Β· 2.00 Γ… Β· X-ray
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
0.20Highly Constrained
pLIβ“˜
1.00Intolerant
Observed/Expected LoF0.04 [0.02–0.20]
RankingsWhere ATP6AP2 stands among ~20K protein-coding genes
  • #2,378of 20,598
    Most Researched182 Β· top quartile
  • #3,317of 5,498
    Most Pathogenic Variants6
  • #464of 17,882
    Most Constrained (LOEUF)0.20 Β· top 5%
Genes detectedATP6AP2
Sources retrieved25 papers
Response timeβ€”
πŸ“„ Sources
25β–Ό
1
X-Linked Epilepsies: A Narrative Review.
PMID: 38612920
Int J Mol Sci Β· 2024
1.00
2
An organ-wide spatiotemporal transcriptomic and cellular atlas of the regenerating zebrafish heart.
PMID: 40253397
Nat Commun Β· 2025
0.90
3
Defects in lysosomal function and lipid metabolism in human microglia harboring a TREM2 loss of function mutation.
PMID: 37115208
Acta Neuropathol Β· 2023
0.80
4
Roles and functions of Atp6ap2 in the brain.
PMID: 30323117
Neural Regen Res Β· 2018
0.70
5
The (pro)renin receptor in health and disease.
PMID: 31164719
Nat Rev Nephrol Β· 2019
0.64