PNPLA1

Chr 6

patatin like domain 1, omega-hydroxyceramide transacylase

Also known as: ARCI10, dJ50J22.1

The protein encoded by this gene belongs to the patatin-like phospholipase (PNPLA) family, which is characterized by the presence of a highly conserved patatin domain. PNPLA family members have diverse lipolytic and acyltransferase activities, and are key elements in lipid metabolism. While other members of this family have been well characterized, the function of this gene remained an enigma. However, recent studies show that this gene is expressed in the skin epidermal keratinocytes, and has a role in glycerophospholipid metabolism in the cutaneous barrier. Consistent with these observations, mutations in this gene are associated with ichthyosis in human (autosomal recessive congenital ichthyoses, ARCI) and dog. Alternatively spliced transcript variants encoding different isoforms have been found for this gene. [provided by RefSeq, Jun 2012]

GeneReviewsResearchGenerating clinical summary…

Primary Disease Associations & Inheritance

UniProtIchthyosis, congenital, autosomal recessive 10
0
Active trials
14
Pubs (1 yr)
P/LP submissions
P/LP missense
0.75
LOEUF
Multiple*
Mechanism· predicted
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GeneReview available — PNPLA1
Authoritative clinical overview · Recommended first read
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Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?
0.75LOEUF
pLI 0.000
Z-score 2.47
OE 0.44 (0.270.75)
Tolerant

Typical tolerance to LoF variation

Missense Constraint?
0.73Z-score
OE missense 0.88 (0.800.98)
283 obs / 320.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios?
LoF OE?0.44 (0.270.75)
00.351.4
Missense OE?0.88 (0.800.98)
00.61.4
Synonymous OE?1.09
01.21.6
LoF obs/exp: 10 / 22.7Missense obs/exp: 283 / 320.0Syn Z: -0.82

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PNPLA1 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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