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10 sources retrieved · Most recent: April 2026 · Index updated 14 days ago
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JPH3
junctophilin 3
Chromosome 16 · 16q24.2
NCBI Gene: 57338Ensembl: ENSG00000154118.13HGNC: HGNC:14203UniProt: B4DIC1
48PubMed Papers
21Diseases
0Drugs
1Pathogenic Variants
CLINICAL
OMIM Disease Gene
DATA QUALITY
✓ Experimental GO Evidence✓ Swiss-Prot Reviewed
protein bindingregulation of synaptic plasticityendoplasmic reticulum membranejunctional membrane complexHuntington disease-like 2migraine disorderhypertrophic cardiomyopathyHepatomegaly
✦AI Summary

JPH3 (junctophilin 3) is a brain-specific structural protein that forms junctional membrane complexes linking the plasma membrane to the endoplasmic reticulum, facilitating calcium signaling in neurons 1. JPH3 contains six MORN motifs and a transmembrane domain anchoring it to the ER membrane, enabling organized subcellular junctions critical for neuronal function 1. The primary pathogenic mechanism involves CTG trinucleotide repeat expansion in JPH3, causing Huntington disease-like 2 (HDL2), a rare genetic disorder predominantly affecting individuals of African ancestry 2. HDL2 presents with motor, cognitive, and psychiatric features closely resembling Huntington disease, with similar striatal atrophy patterns, though pathogenesis mechanisms remain incompletely understood 2. Beyond neurodegeneration, JPH3 functions as a tumor suppressor in digestive cancers. In hepatocellular and colorectal carcinomas, JPH3 expression is silenced through promoter CpG methylation, associating with poor patient outcomes 34. Restoration of JPH3 expression inhibits cancer cell proliferation, invasion, and migration while promoting mitochondrial-mediated apoptosis through elevated cytosolic calcium and endoplasmic reticulum stress responses 4. Additionally, JPH3 was identified as a potential biomarker for mild cognitive impairment conversion to Alzheimer's disease 5. These findings establish JPH3 as multifunctional—essential for neuronal calcium homeostasis and synaptic function while suppressing tumorigenesis through apoptotic pathways.

Sources cited
1
JPH3 structural composition (MORN motifs, transmembrane domain) and role in forming junctional membrane complexes between plasma membrane and ER
PMID: 35001666
2
JPH3 CTG repeat expansion causes Huntington disease-like 2 with African ancestry pattern and clinical similarity to HD including striatal atrophy
PMID: 38114648
3
JPH3 promoter methylation inactivation in hepatocellular carcinoma and its tumor suppressor function in inhibiting proliferation, invasion and EMT
PMID: 35169860
4
JPH3 promoter CpG methylation in digestive cancers, tumor suppressor function via calcium signaling and mitochondrial-mediated apoptosis
PMID: 28656064
5
JPH3 identified as a key feature distinguishing MCI stable from converter to Alzheimer's disease in omics integration analysis
PMID: 38761503
6
JPH3 trinucleotide repeat expansion in Huntington disease-like 2 causes selective striatal degeneration
PMID: 15651335
Disease Associationsⓘ21
Huntington disease-like 2Open Targets
0.54Moderate
migraine disorderOpen Targets
0.29Weak
hypertrophic cardiomyopathyOpen Targets
0.27Weak
HepatomegalyOpen Targets
0.25Weak
kidney transplantOpen Targets
0.21Weak
blindness (disorder)Open Targets
0.20Weak
Visual impairmentOpen Targets
0.20Weak
bladder diseaseOpen Targets
0.20Weak
genetic disorderOpen Targets
0.19Weak
obesityOpen Targets
0.18Weak
protozoa infectious diseaseOpen Targets
0.18Weak
Ischemic strokeOpen Targets
0.18Weak
HeadacheOpen Targets
0.15Weak
small vessel strokeOpen Targets
0.14Weak
strokeOpen Targets
0.13Weak
secondary malignant neoplasmOpen Targets
0.09Suggestive
neoplasmOpen Targets
0.08Suggestive
PainOpen Targets
0.08Suggestive
hepatocellular carcinomaOpen Targets
0.07Suggestive
atypical teratoid rhabdoid tumorOpen Targets
0.05Suggestive
Huntington disease-like 2UniProt
Pathogenic Variants1
NM_001271604.4(JPH3):c.431CWG[41_?]Pathogenic
Huntington disease-like 2
☆☆☆☆2001
View on ClinVar ↗
Related Genes
CACNA1DProtein interaction98%RYR1Protein interaction95%VPS13AProtein interaction83%CACNA1SProtein interaction73%TRDNProtein interaction73%SYPL2Protein interaction73%
Tissue Expression6 tissues
Brain
100%
Bone Marrow
5%
Ovary
1%
Liver
1%
Lung
0%
Heart
0%
Gene Interaction Network
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JPH3CACNA1DRYR1VPS13ACACNA1STRDNSYPL2
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted · UniProt Q8WXH2
View on AlphaFold ↗
Constraintⓘ
LOEUFⓘ
0.36Moderately Constrained
pLIⓘ
1.00Intolerant
Observed/Expected LoF0.22 [0.14–0.36]
RankingsWhere JPH3 stands among ~20K protein-coding genes
  • #9,090of 20,598
    Most Researched48
  • #5,109of 5,498
    Most Pathogenic Variants1
  • #1,614of 17,882
    Most Constrained (LOEUF)0.36 · top 10%
Genes detectedJPH3
Sources retrieved10 papers
Response time—
📄 Sources
10▼
1
Methylation‑associated inactivation of JPH3 and its effect on prognosis and cell biological function in HCC.
PMID: 35169860
Mol Med Rep · 2022
1.00
2
PMID: 20301701
0.90
3
[Hereditary chorea--update].
PMID: 15651335
Rinsho Shinkeigaku · 2004
0.80
4
Searching for mutation in the JPH3, ATN1 and TBP genes in Polish patients suspected of Huntington's disease and without mutation in the IT15 gene.
PMID: 18651325
Neurol Neurochir Pol · 2008
0.70
5
The role of junctophilin proteins in cellular function.
PMID: 35001666
Physiol Rev · 2022
0.60