ACSL3

Chr 2

acyl-CoA synthetase long chain family member 3

Also known as: ACS3, FACL3, LACS 3, LACS3, PRO2194

ACSL3 encodes a long-chain fatty-acid-coenzyme A ligase that converts free fatty acids into fatty acyl-CoA esters for lipid biosynthesis and fatty acid degradation, with high expression in brain and preference for substrates including myristate and arachidonate. Mutations cause autosomal recessive intellectual disability with onset in infancy. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.55), consistent with its role in brain lipid metabolism.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
59
Pubs (1 yr)
32
P/LP submissions
0%
P/LP missense
0.55
LOEUF
DN
Mechanism· predicted
Clinical SummaryACSL3
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.35) despite low pLI — interpret in context.
📋
ClinVar Variants
32 unique Pathogenic / Likely Pathogenic· 63 VUS of 125 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.55LOEUF
pLI 0.000
Z-score 3.72
OE 0.35 (0.220.55)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.01Z-score
OE missense 0.72 (0.650.79)
283 obs / 395.6 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.35 (0.220.55)
00.351.4
Missense OE0.72 (0.650.79)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 13 / 37.6Missense obs/exp: 283 / 395.6Syn Z: -0.96
DN
0.6842th %ile
GOF
0.5758th %ile
LOF
0.3454th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

125 submitted variants in ClinVar

Classification Summary

Pathogenic31
Likely Pathogenic1
VUS63
Likely Benign5
Benign2
31
Pathogenic
1
Likely Pathogenic
63
VUS
5
Likely Benign
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
31
0
31
Likely Pathogenic
0
0
1
0
1
VUS
0
62
1
0
63
Likely Benign
0
4
0
1
5
Benign
0
0
0
2
2
Total066333102

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

View in ClinVar →

Protein Context — Lollipop Plot

ACSL3 · 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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Clinical Literature
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