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GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 15 days ago
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PNPLA1
patatin like domain 1, omega-hydroxyceramide transacylase
Chromosome 6 Β· 6p21.31
NCBI Gene: 285848Ensembl: ENSG00000180316.13HGNC: HGNC:21246UniProt: A0A1B0GW56
34PubMed Papers
21Diseases
0Drugs
48Pathogenic Variants
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
ceramide biosynthetic processacyltransferase activity, transferring groups other than amino-acyl groupscytoplasmomega-hydroxyceramide biosynthetic processcongenital non-bullous ichthyosiform erythrodermacongenital reticular ichthyosiform erythrodermalamellar ichthyosisichthyosis
✦AI Summary

PNPLA1 (patatin-like phospholipase domain-containing protein 1) is a specialized transacylase enzyme that catalyzes the final step in omega-O-acylceramide (acylceramide) biosynthesis, which is essential for epidermal barrier function 12. The enzyme transfers linoleic acid from triglycerides to omega-hydroxyceramides, producing acylceramides that are critical components of the stratum corneum's lipid lamellae and cornified lipid envelope 13. PNPLA1 shows selective incorporation of linoleic acid over other fatty acids in this transacylation reaction 3. The enzyme requires proper localization to lipid droplets through interaction with the co-activator ABHD5 for optimal function 4. Loss-of-function mutations in PNPLA1 cause autosomal recessive congenital ichthyosis (ARCI) in both humans and dogs, characterized by defective acylceramide synthesis, impaired skin barrier function, and severe scaling 156. Studies have identified numerous pathogenic mutations that either completely abolish enzyme activity or disrupt its subcellular localization to lipid droplets 5. The conservation of PNPLA1 function across mammalian species underscores its fundamental importance in epidermal lipid metabolism and skin barrier maintenance 1.

Sources cited
1
PNPLA1 catalyzes the transfer of linoleic acid from triglycerides to omega-hydroxyceramides and is essential for acylceramide synthesis and skin barrier function
PMID: 27751867
2
PNPLA1 acts as a transacylase in skin-specific sphingolipid metabolism and is linked to autosomal recessive congenital ichthyosis
PMID: 30290227
3
PNPLA1 shows selective incorporation of linoleic acid over oleic acid in acylceramide synthesis
PMID: 37087101
4
PNPLA1 requires ABHD5 for proper localization to lipid droplets and enzymatic function
PMID: 40818613
5
Multiple PNPLA1 mutations cause complete loss of acylceramide-producing activity and disrupt enzyme localization
PMID: 35970721
6
PNPLA1 mutations cause ichthyosis in dogs, demonstrating conservation across mammalian species
PMID: 40150930
Disease Associationsβ“˜21
congenital non-bullous ichthyosiform erythrodermaOpen Targets
0.73Strong
congenital reticular ichthyosiform erythrodermaOpen Targets
0.62Moderate
lamellar ichthyosisOpen Targets
0.54Moderate
ichthyosisOpen Targets
0.50Moderate
inherited ichthyosisOpen Targets
0.46Moderate
autosomal recessive congenital ichthyosisOpen Targets
0.38Weak
ulcerative colitisOpen Targets
0.30Weak
placenta praeviaOpen Targets
0.28Weak
rheumatoid arthritisOpen Targets
0.28Weak
ovarian neoplasmOpen Targets
0.28Weak
osteomyelitisOpen Targets
0.27Weak
Abnormality of the skinOpen Targets
0.27Weak
neuroendocrine neoplasmOpen Targets
0.23Weak
genetic disorderOpen Targets
0.19Weak
ulerythema ophryogenesisOpen Targets
0.05Suggestive
ichthyosis, congenital, autosomal recessive 14Open Targets
0.05Suggestive
ichthyosis with erythrokeratodermaOpen Targets
0.05Suggestive
erythrokeratodermia variabilisOpen Targets
0.05Suggestive
ichthyosis hystrix of Curth-MacklinOpen Targets
0.05Suggestive
peeling skin syndrome 6Open Targets
0.05Suggestive
Ichthyosis, congenital, autosomal recessive 10UniProt
Pathogenic Variants48
NM_001374623.1(PNPLA1):c.892C>T (p.Arg298Ter)Pathogenic
not provided|Autosomal recessive congenital ichthyosis 10
β˜…β˜…β˜†β˜†2026β†’ Residue 298
NM_001374623.1(PNPLA1):c.1300del (p.Ala434fs)Pathogenic
Congenital ichthyosiform erythroderma|not provided|Autosomal recessive congenital ichthyosis 10
β˜…β˜…β˜†β˜†2026β†’ Residue 434
NM_001374623.1(PNPLA1):c.100G>C (p.Ala34Pro)Pathogenic
Autosomal recessive congenital ichthyosis 10|Lamellar ichthyosis|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 34
NM_001374623.1(PNPLA1):c.464C>T (p.Pro155Leu)Likely pathogenic
Congenital ichthyosiform erythroderma|Autosomal recessive congenital ichthyosis 10|Lamellar ichthyosis
β˜…β˜…β˜†β˜†2025β†’ Residue 155
NM_001374623.1(PNPLA1):c.514G>A (p.Asp172Asn)Pathogenic
Congenital ichthyosiform erythroderma|not provided|Lamellar ichthyosis
β˜…β˜…β˜†β˜†2025β†’ Residue 172
NM_001374623.1(PNPLA1):c.149C>A (p.Ala50Glu)Pathogenic
Autosomal recessive congenital ichthyosis 10
β˜…β˜…β˜†β˜†2025β†’ Residue 50
NM_001374623.1(PNPLA1):c.488C>T (p.Pro163Leu)Pathogenic
Autosomal recessive congenital ichthyosis 10|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 163
NM_001374623.1(PNPLA1):c.387C>A (p.Asp129Glu)Pathogenic
Ichthyosis|Autosomal recessive congenital ichthyosis 10|Autosomal recessive congenital ichthyosis|not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 129
NM_001374623.1(PNPLA1):c.646T>C (p.Cys216Arg)Pathogenic
not provided|Autosomal recessive congenital ichthyosis 10|Congenital ichthyosiform erythroderma|Lamellar ichthyosis
β˜…β˜…β˜†β˜†2024β†’ Residue 216
NM_001374623.1(PNPLA1):c.417_418delinsTC (p.Ser140Pro)Pathogenic
Lamellar ichthyosis|Autosomal recessive congenital ichthyosis 10
β˜…β˜…β˜†β˜†2024β†’ Residue 140
NM_001374623.1(PNPLA1):c.418T>C (p.Ser140Pro)Pathogenic
Congenital ichthyosiform erythroderma|Autosomal recessive congenital ichthyosis 10|Lamellar ichthyosis|not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 140
NM_001374623.1(PNPLA1):c.158C>T (p.Ser53Leu)Likely pathogenic
Lamellar ichthyosis|Autosomal recessive congenital ichthyosis 10
β˜…β˜…β˜†β˜†2024β†’ Residue 53
NM_001374623.1(PNPLA1):c.158C>G (p.Ser53Trp)Pathogenic
Congenital ichthyosiform erythroderma|Autosomal recessive congenital ichthyosis 10|not provided
β˜…β˜…β˜†β˜†2023β†’ Residue 53
NM_001374623.1(PNPLA1):c.38del (p.Pro13fs)Likely pathogenic
not provided|Lamellar ichthyosis
β˜…β˜…β˜†β˜†2022β†’ Residue 13
NM_001374623.1(PNPLA1):c.1318C>T (p.Arg440Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2026β†’ Residue 440
NM_001374623.1(PNPLA1):c.504+2T>CLikely pathogenic
not provided
β˜…β˜†β˜†β˜†2025
NM_001374623.1(PNPLA1):c.157T>C (p.Ser53Pro)Pathogenic
Congenital ichthyosiform erythroderma|Lamellar ichthyosis
β˜…β˜†β˜†β˜†2025β†’ Residue 53
NM_001374623.1(PNPLA1):c.56C>T (p.Ser19Leu)Pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 19
NM_001374623.1(PNPLA1):c.485C>G (p.Pro162Arg)Likely pathogenic
Autosomal recessive congenital ichthyosis 10
β˜…β˜†β˜†β˜†2025β†’ Residue 162
NM_001374623.1(PNPLA1):c.536A>G (p.Gln179Arg)Pathogenic
Autosomal recessive congenital ichthyosis 10
β˜…β˜†β˜†β˜†2025β†’ Residue 179
View on ClinVar β†—
Related Genes
NIPAL4Protein interaction96%SDR9C7Protein interaction89%CERS3Protein interaction89%LIPNProtein interaction87%ABHD5Protein interaction86%PNPLA5Shared pathway80%
Tissue Expression6 tissues
Bone Marrow
100%
Lung
7%
Brain
5%
Ovary
2%
Liver
1%
Heart
0%
Gene Interaction Network
Click a node to explore
PNPLA1NIPAL4SDR9C7CERS3LIPNABHD5PNPLA5
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q8N8W4
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.94LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.71 [0.54–0.94]
RankingsWhere PNPLA1 stands among ~20K protein-coding genes
  • #11,211of 20,598
    Most Researched34
  • #1,378of 5,498
    Most Pathogenic Variants48
  • #8,819of 17,882
    Most Constrained (LOEUF)0.94
Genes detectedPNPLA1
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Meta-Analysis of Mutations in
PMID: 33435499
Genes (Basel) Β· 2021
1.00
2
PNPLA1-Mediated Acylceramide Biosynthesis and Autosomal Recessive Congenital Ichthyosis.
PMID: 35893253
Metabolites Β· 2022
0.90
3
The role of PNPLA1 in Ο‰-O-acylceramide synthesis and skin barrier function.
PMID: 30290227
Biochim Biophys Acta Mol Cell Biol Lipids Β· 2019
0.80
4
Defective targeting of PNPLA1 to lipid droplets causes ichthyosis in ABHD5-syndromic epidermal differentiation disorder.
PMID: 40818613
J Lipid Res Β· 2025
0.70
5
Intragenic PNPLA1 duplication in Labrador retrievers with nonepidermolytic ichthyosis.
PMID: 40150930
Vet Dermatol Β· 2025
0.60