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10 sources retrieved · Most recent: April 2026 · Index updated 15 days ago
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PNPLA4
patatin like domain 4, phospholipase and triacylglycerol lipase
Chromosome X · Xp22.31
NCBI Gene: 8228Ensembl: ENSG00000006757.13HGNC: HGNC:24887UniProt: P41247
25PubMed Papers
20Diseases
0Drugs
0Pathogenic Variants
DATA QUALITY
✓ Experimental GO Evidence✓ Swiss-Prot Reviewed
diolein transacylation activitytriacylglycerol lipase activityA2-type glycerophospholipase activitymitochondrionAbnormality of neuronal migrationcongenital heart diseasehead and neck squamous cell carcinomasystemic juvenile idiopathic arthritis
✦AI Summary

PNPLA4 is an X-linked phospholipase and triacylglycerol lipase with multiple enzymatic activities critical for lipid homeostasis. Primary functions include abundant triacylglycerol lipase activity, retinyl ester hydrolysis, fatty acid transfer from triglycerides to retinol, and phospholipase A2 activity 1. PNPLA4 regulates glycerophospholipid turnover in human cells; knockdown reduced phosphatidylcholine, phosphatidylethanolamine, and phosphatidylserine turnover by 30-50% and inhibited G0/G1 to S cell cycle progression 1. At the molecular level, PNPLA4 functions in primary cilia formation and mitochondrial-mediated ATP production through energy metabolism regulation 2. Disease relevance spans multiple systems. PNPLA4 mutations cause mitochondrial respiratory chain complex deficiencies 3. Copy number variations and duplications at Xp22.31 containing PNPLA4 associate with heterotaxy and congenital heart disease, with PNPLA4 expression critical in lateral plate mesoderm and neural crest lineages during left/right patterning and cardiomyocyte differentiation 2. X-linked intellectual disability involves PNPLA4 deletions, supported by high brain transcript expression 4. PNPLA4 also associates with cardiac arrhythmias and gastrointestinal disorders in Xp22.31 deletion carriers 5, and represents a component of mitochondrial lipid metabolism gene signatures prognostic for colorectal cancer 6. Altered PNPLA4 expression occurs during multiple myeloma progression 7.

Sources cited
1
PNPLA4 participates in glycerophospholipid turnover, with knockdown reducing phosphatidylcholine/ethanolamine/serine turnover and inhibiting cell cycle progression
PMID: 27317427
2
PNPLA4 duplication at Xp22.31 associates with heterotaxy and congenital heart disease through regulation of primary cilia formation and mitochondrial ATP production
PMID: 38973237
3
PNPLA4 identified as a novel causative gene for mitochondrial respiratory chain complex deficiencies
PMID: 26741492
4
PNPLA4 proposed as candidate gene for X-linked intellectual disability at Xp22.31, with high brain transcript expression
PMID: 31963867
5
PNPLA4 variants at Xp22.31 associate with atrial fibrillation, gastrointestinal disorders, and asthma in deletion carriers
PMID: 36379544
6
PNPLA4 is a mitochondrial lipid metabolism gene with prognostic significance in colorectal cancer
PMID: 41293172
7
PNPLA4 expression is significantly altered during multiple myeloma progression from newly diagnosed to relapsed/refractory disease
PMID: 33531688
Disease Associationsⓘ20
Abnormality of neuronal migrationOpen Targets
0.11Weak
congenital heart diseaseOpen Targets
0.03Suggestive
head and neck squamous cell carcinomaOpen Targets
0.01Suggestive
systemic juvenile idiopathic arthritisOpen Targets
0.01Suggestive
neoplasmOpen Targets
0.01Suggestive
hyperinsulinemic hypoglycemia, familial, 4Open Targets
0.01Suggestive
atrial fibrillationOpen Targets
0.01Suggestive
Mobius syndromeOpen Targets
0.01Suggestive
colorectal carcinomaOpen Targets
0.01Suggestive
cancerOpen Targets
0.01Suggestive
liposarcomaOpen Targets
0.01Suggestive
obesityOpen Targets
0.01Suggestive
hyperlipidemiaOpen Targets
0.01Suggestive
asthmaOpen Targets
0.01Suggestive
autismOpen Targets
0.00Suggestive
Insulin resistanceOpen Targets
0.00Suggestive
type 2 diabetes mellitusOpen Targets
0.00Suggestive
hepatocellular carcinomaOpen Targets
0.00Suggestive
X-linked intellectual disabilityOpen Targets
0.00Suggestive
constitutional neutropeniaOpen Targets
0.00Suggestive
Pathogenic Variants
No pathogenic variants reported on ClinVar for this gene.
View on ClinVar ↗
Related Genes
RDH10Protein interaction98%DHRS3Protein interaction97%RDH11Protein interaction95%SDR16C5Protein interaction94%DHRS9Protein interaction94%RDH12Protein interaction94%
Tissue Expression6 tissues
Heart
100%
Liver
50%
Brain
41%
Ovary
39%
Bone Marrow
18%
Lung
15%
Gene Interaction Network
Click a node to explore
PNPLA4RDH10DHRS3RDH11SDR16C5DHRS9RDH12
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted · UniProt P41247
View on AlphaFold ↗
Constraintⓘ
LOEUFⓘ
1.26LoF Tolerant
pLIⓘ
0.00Tolerant
Observed/Expected LoF0.82 [0.55–1.26]
RankingsWhere PNPLA4 stands among ~20K protein-coding genes
  • #13,041of 20,598
    Most Researched25
  • #13,312of 17,882
    Most Constrained (LOEUF)1.26
Genes detectedPNPLA4
Sources retrieved10 papers
Response time—
📄 Sources
10▼
1
A Comprehensive Genomic Analysis Reveals the Genetic Landscape of Mitochondrial Respiratory Chain Complex Deficiencies.
PMID: 26741492
PLoS Genet · 2016
1.00
2
Characterising heart rhythm abnormalities associated with Xp22.31 deletion.
PMID: 36379544
J Med Genet · 2023
0.90
3
Association of copy number variation in X chromosome-linked PNPLA4 with heterotaxy and congenital heart disease.
PMID: 38973237
Chin Med J (Engl) · 2024
0.80
4
Comparative Genomic Mapping Implicates LRRK2 for Intellectual Disability and Autism at 12q12, and HDHD1, as Well as PNPLA4, for X-Linked Intellectual Disability at Xp22.31.
PMID: 31963867
J Clin Med · 2020
0.70
5
A mitochondrial lipid metabolism-related gene signature predicts prognosis and immune landscape in colorectal cancer.
PMID: 41293172
Front Immunol · 2025
0.60