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GeneE
10 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.
ATCAY
ATCAY kinesin light chain interacting caytaxin
Chromosome 19 Β· 19p13.3
NCBI Gene: 85300Ensembl: ENSG00000167654.19HGNC: HGNC:779UniProt: A0A0S2Z5T8
21PubMed Papers
21Diseases
0Drugs
4Pathogenic Variants
FUNCTIONAL ROLE
ApoptosisTransporter
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
negative regulation of L-glutamine biosynthetic processcytoplasmkinesin bindingmitochondrionCayman type cerebellar ataxiaCerebellar ataxia, Cayman typegenetic disorderHydrocephalus
✦AI Summary

ATCAY encodes caytaxin, a protein essential for cerebellar development and neuronal function. The protein localizes to mitochondria and interacts with kinesin light chains, suggesting roles in mitochondrial distribution within axons and dendrites 1. ATCAY may regulate glutaminase to modulate glutamate neurotransmission, though its precise molecular mechanism remains incompletely characterized 2. ATCAY expression correlates directly with phenotypic severity in animal models; complete absence causes severe ataxia while reduced expression produces milder motor deficits 1. The protein is highly expressed in developing cerebellar cortex, particularly in molecular and granular layers during postnatal maturation, with transcript levels peaking around postnatal day 7-14 2. Loss of ATCAY function impairs Purkinje neuron firing and increases deep cerebellar nuclear neuron excitability, contributing to both ataxia and dystonic symptoms 3. Homozygous ATCAY mutations cause Cayman cerebellar ataxia, an autosomal recessive disorder featuring psychomotor retardation, cerebellar dysfunction, nystagmus, intention tremor, and ataxic gait 45. Recent evidence suggests elevated anti-ATCAY autoantibodies correlate with Alzheimer's disease risk and cognitive impairment severity 6. Additionally, ATCAY homologs mediate antiviral defense against dengue virus in both human cells and mosquito vectors, indicating evolutionary conservation of function beyond neural development 7.

Sources cited
1
Caytaxin protein expression correlates with disease severity in ataxic mouse models; expression levels are absent in severe ataxia lines and markedly decreased in mild ataxia lines
PMID: 23226316
2
ATCAY transcript is exclusively expressed in brain tissues with developmental regulation; transcript levels peak during early postnatal cerebellar development and localize to all cerebellar layers
PMID: 16246457
3
Loss of ATCAY function causes abnormal Purkinje neuron firing patterns and increased deep cerebellar nuclear neuron excitability, contributing to ataxia and dystonic gait
PMID: 24727095
4
Homozygous frameshift variants in ATCAY cause Cayman cerebellar ataxia presenting with developmental delay, motor dysfunction, hypotonia, nystagmus, and ataxic gait
PMID: 37752557
5
Novel homozygous ATCAY variants identified in consanguineous families present with complex movement disorders including ataxia and dystonia
PMID: 29449188
6
Elevated serum anti-ATCAY autoantibody levels significantly correlate with Alzheimer's disease and mild cognitive impairment and correlate inversely with cognitive test scores
PMID: 35079008
7
ATCAY homologs mediate resistance to dengue virus infection in both human cells and mosquito vectors, demonstrating evolutionarily conserved antiviral function
PMID: 30717390
Disease Associationsβ“˜21
Cayman type cerebellar ataxiaOpen Targets
0.72Strong
Cerebellar ataxia, Cayman typeOpen Targets
0.72Strong
genetic disorderOpen Targets
0.19Weak
HydrocephalusOpen Targets
0.14Weak
spinocerebellar ataxia type 23Open Targets
0.09Suggestive
spastic paraplegia 72b, autosomal recessiveOpen Targets
0.09Suggestive
Spinocerebellar ataxia type 40Open Targets
0.09Suggestive
Ataxia - oculomotor apraxia type 1Open Targets
0.08Suggestive
Dysequilibrium syndromeOpen Targets
0.08Suggestive
Rare hereditary ataxiaOpen Targets
0.08Suggestive
spinocerebellar ataxia type 35Open Targets
0.08Suggestive
spinocerebellar ataxia, autosomal recessive 25Open Targets
0.08Suggestive
Autosomal recessive spastic paraplegia type 72Open Targets
0.08Suggestive
hereditary spastic paraplegia 72Open Targets
0.08Suggestive
spinocerebellar ataxia type 4Open Targets
0.07Suggestive
Adult-onset autosomal recessive cerebellar ataxiaOpen Targets
0.07Suggestive
autosomal dominant sensory ataxia 1Open Targets
0.07Suggestive
Autosomal dominant striatal neurodegenerationOpen Targets
0.07Suggestive
Spinocerebellar ataxia type 41Open Targets
0.07Suggestive
infantile-onset autosomal recessive nonprogressive cerebellar ataxiaOpen Targets
0.07Suggestive
Cerebellar ataxia, cayman typeUniProt
Pathogenic Variants4
NM_033064.5(ATCAY):c.556G>T (p.Glu186Ter)Pathogenic
Cayman type cerebellar ataxia
β˜…β˜†β˜†β˜†2024β†’ Residue 186
NM_033064.5(ATCAY):c.903C>G (p.Ser301Arg)Pathogenic
not provided
β˜…β˜†β˜†β˜†2017β†’ Residue 301
NM_033064.5(ATCAY):c.965+3G>TPathogenic
not provided
β˜…β˜†β˜†β˜†2017
NM_033064.5(ATCAY):c.602_608del (p.Asp201fs)Pathogenic
Cayman type cerebellar ataxia
β˜†β˜†β˜†β˜†2021β†’ Residue 201
View on ClinVar β†—
Related Genes
TTPAProtein interaction91%KLC1Protein interaction82%FKBP1CProtein interaction71%GPRIN3Shared pathway33%PRUNE2Shared pathway33%GPRIN1Shared pathway33%
Tissue Expression6 tissues
Brain
100%
Liver
1%
Ovary
1%
Lung
0%
Bone Marrow
0%
Heart
0%
Gene Interaction Network
Click a node to explore
ATCAYTTPAKLC1FKBP1CGPRIN3PRUNE2GPRIN1
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q86WG3
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.72LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.51 [0.36–0.72]
RankingsWhere ATCAY stands among ~20K protein-coding genes
  • #13,832of 20,598
    Most Researched21
  • #3,853of 5,498
    Most Pathogenic Variants4
  • #5,620of 17,882
    Most Constrained (LOEUF)0.72
Genes detectedATCAY
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Homologs of Human Dengue-Resistance Genes,
PMID: 30717390
Insects Β· 2019
1.00
2
Novel homozygous frameshift variant in the ATCAY gene in an Iranian patient with Cayman cerebellar ataxia; expanding the neuroimaging and clinical features: a case report.
PMID: 37752557
BMC Med Genomics Β· 2023
0.90
3
Expression of Caytaxin protein in Cayman Ataxia mouse models correlates with phenotype severity.
PMID: 23226316
PLoS One Β· 2012
0.80
4
Caytaxin deficiency causes generalized dystonia in rats.
PMID: 16246457
Brain Res Mol Brain Res Β· 2005
0.70
5
A combination of multiple autoantibodies is associated with the risk of Alzheimer's disease and cognitive impairment.
PMID: 35079008
Sci Rep Β· 2022
0.60