ENPP2

Chr 8

ectonucleotide pyrophosphatase/phosphodiesterase 2

Also known as: ATX, ATX-X, AUTOTAXIN, LysoPLD, NPP2, PD-IALPHA, PDNP2

This gene encodes a secreted lysophospholipase D that hydrolyzes lysophospholipids to produce lysophosphatidic acid (LPA), a signaling molecule involved in cell proliferation, migration, and angiogenesis. Biallelic mutations cause generalized arterial calcification of infancy (GACI), a severe autosomal recessive disorder presenting in early infancy with calcification of large arteries and cardiac complications. The gene shows high tolerance to loss-of-function variants (low pLI score), consistent with its recessive inheritance pattern.

Summary from RefSeq, UniProt
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2
Active trials
22
Pubs (1 yr)
55
P/LP submissions
0%
P/LP missense
0.43
LOEUF
Mechanism
Clinical SummaryENPP2
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.28) despite low pLI — interpret in context.
📋
ClinVar Variants
55 unique Pathogenic / Likely Pathogenic· 131 VUS of 225 total submissions
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.43LOEUF
pLI 0.003
Z-score 4.98
OE 0.28 (0.190.43)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.38Z-score
OE missense 0.83 (0.760.90)
425 obs / 513.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.28 (0.190.43)
00.351.4
Missense OE0.83 (0.760.90)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 16 / 56.4Missense obs/exp: 425 / 513.1Syn Z: -0.07

ClinVar Variant Classifications

225 submitted variants in ClinVar

Classification Summary

Pathogenic55
VUS131
Likely Benign5
Benign5
55
Pathogenic
131
VUS
5
Likely Benign
5
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
55
0
55
Likely Pathogenic
0
0
0
0
0
VUS
0
120
10
1
131
Likely Benign
0
3
0
2
5
Benign
0
2
2
1
5
Total0125674196

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

ENPP2 · protein map & ClinVar variants

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

Clinical Literature
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