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10 sources retrieved · Most recent: April 2026 · Index updated 15 days ago
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CLCN6
Cl-/H+ antiporter 6
Chromosome 1 · 1p36.22
NCBI Gene: 1185Ensembl: ENSG00000011021.24HGNC: HGNC:2024UniProt: P51797
42PubMed Papers
21Diseases
0Drugs
1Pathogenic Variants
FUNCTIONAL ROLE
Transporter
CLINICAL
OMIM Disease Gene
DATA QUALITY
✓ Experimental GO Evidence✓ Swiss-Prot Reviewed
chloride transportlysosomal membranevoltage-gated chloride channel activitylate endosomeneurodegeneration, childhood-onset, with hypotonia, respiratory insufficiency, and brain imaging abnormalitiescancerdevelopmental disorder of mental healthhypertension
✦AI Summary

CLCN6 encodes a voltage-gated chloride-proton (Cl⁻/H⁺) antiporter localized to late endosomal and lysosomal membranes, predominantly expressed in the nervous system 1. The protein mediates bidirectional exchange of chloride ions against protons, contributing to late endosomal acidification and maintaining chloride homeostasis 2. ATP binding to cytosolic CLCN6 domains enhances ion transport activity 1. CLCN6 dysfunction manifests in multiple disease contexts. Loss-of-function mutations cause neuronal ceroid lipofuscinosis (NCL), characterized by autophagy impairment, lysosomal dysfunction, and neurodegeneration 2. Conversely, gain-of-function variants disrupt autophagic flux and activate transcription factors TFEB/TFE3, leading to pathological cellular accumulation 2. Disease-associated mutations cluster around residues Y553, F317, and T520 1. Clinically, CLCN6 variants associate with cardiovascular and hematologic phenotypes. Rare coding variants reduce blood pressure and hypertension risk 3, while common variants in the MTHFR-CLCN6-NPPA-NPPB cluster associate with elevated NT-proBNP levels and preeclampsia susceptibility 45. In cancer, CLCN6 upregulation promotes prostate cancer cell proliferation and predicts biochemical recurrence 6. These findings highlight CLCN6's pleiotropic roles in lysosomal homeostasis, blood pressure regulation, and cancer biology.

Sources cited
1
ClC-6 is a late endosomal voltage-gated chloride-proton exchanger predominantly expressed in nervous system; ATP binding enhances ion transport; disease-causing mutations cluster at Y553/F317/T520
PMID: 37831762
2
CLCN6 mutations cause neuronal ceroid lipofuscinosis; disease-causing variants block autophagic flux and activate TFEB/TFE3; Cl⁻/H⁺ transport activity is crucial for lysosomal function
PMID: 38877824
3
Rare CLCN6 coding variants associate with lower diastolic and systolic blood pressure and reduced hypertension risk
PMID: 26658788
4
CLCN6 variants in MTHFR-CLCN6-NPPA-NPPB cluster independently associated with elevated NT-proBNP levels and cardiac dysfunction
PMID: 21273288
5
MTHFR-CLCN6 locus identified in genome-wide association with preeclampsia and gestational hypertension
PMID: 37248299
6
CLCN6 upregulation in prostate cancer promotes cell proliferation and predicts biochemical recurrence
PMID: 36246902
7
Loss of Clcn6 modulates expression of nearby blood pressure regulatory genes MTHFR, NPPA, and NPPB in conserved AGTRAP-PLOD1 locus
PMID: 35927940
Disease Associationsⓘ21
neurodegeneration, childhood-onset, with hypotonia, respiratory insufficiency, and brain imaging abnormalitiesOpen Targets
0.66Moderate
cancerOpen Targets
0.54Moderate
developmental disorder of mental healthOpen Targets
0.37Weak
hypertensionOpen Targets
0.35Weak
Global developmental delayOpen Targets
0.34Weak
movement disorderOpen Targets
0.34Weak
Neurogenic bladderOpen Targets
0.34Weak
Abnormal speech patternOpen Targets
0.34Weak
Abnormality of temperature regulationOpen Targets
0.34Weak
Abnormality of the respiratory systemOpen Targets
0.34Weak
Abnormality of the skinOpen Targets
0.34Weak
Abnormality of visionOpen Targets
0.34Weak
EEG abnormalityOpen Targets
0.34Weak
Feeding difficultiesOpen Targets
0.34Weak
HypotoniaOpen Targets
0.34Weak
Motor delayOpen Targets
0.34Weak
speech disorderOpen Targets
0.34Weak
preeclampsiaOpen Targets
0.32Weak
cardiovascular diseaseOpen Targets
0.29Weak
essential hypertensionOpen Targets
0.27Weak
Ceroid lipofuscinosis, neuronal, 15UniProt
Pathogenic Variants1
NM_001286.5(CLCN6):c.599A>C (p.Glu200Ala)Pathogenic
Neurodegeneration, childhood-onset, with hypotonia, respiratory insufficiency, and brain imaging abnormalities
☆☆☆☆2024→ Residue 200
View on ClinVar ↗
Related Genes
FAM131BProtein interaction86%MTHFRProtein interaction82%OSTM1Protein interaction82%AGTRAPProtein interaction78%KIAA1549Protein interaction77%PPT1Protein interaction76%
Tissue Expression6 tissues
Ovary
100%
Bone Marrow
86%
Brain
80%
Heart
80%
Lung
69%
Liver
68%
Gene Interaction Network
Click a node to explore
CLCN6FAM131BMTHFROSTM1AGTRAPKIAA1549PPT1
PROTEIN STRUCTURE
Preparing viewer…
PDB8JPO · 3.40 Å · EM
View on RCSB ↗
Constraintⓘ
LOEUFⓘ
0.79LoF Tolerant
pLIⓘ
0.00Tolerant
Observed/Expected LoF0.64 [0.53–0.79]
RankingsWhere CLCN6 stands among ~20K protein-coding genes
  • #9,851of 20,598
    Most Researched42
  • #5,150of 5,498
    Most Pathogenic Variants1
  • #6,488of 17,882
    Most Constrained (LOEUF)0.79
Genes detectedCLCN6
Sources retrieved10 papers
Response time—
📄 Sources
10▼
1
Polygenic prediction of preeclampsia and gestational hypertension.
PMID: 37248299
Nat Med · 2023
1.00
2
Genome-wide association analysis and fine mapping of NT-proBNP level provide novel insight into the role of the MTHFR-CLCN6-NPPA-NPPB gene cluster.
PMID: 21273288
Hum Mol Genet · 2011
0.90
3
Mutations in CLCN6 as a Novel Genetic Cause of Neuronal Ceroid Lipofuscinosis in Patients and a Murine Model.
PMID: 38877824
Ann Neurol · 2024
0.80
4
The human and mouse methylenetetrahydrofolate reductase (MTHFR) genes: genomic organization, mRNA structure and linkage to the CLCN6 gene.
PMID: 11080594
Gene · 2000
0.70
5
Rare Exome Sequence Variants in CLCN6 Reduce Blood Pressure Levels and Hypertension Risk.
PMID: 26658788
Circ Cardiovasc Genet · 2016
0.60