ACOT9

Chr X

acyl-CoA thioesterase 9

Also known as: ACATE2, CGI-16, MT-ACT48, MTACT48

This mitochondrial acyl-CoA thioesterase catalyzes the hydrolysis of acyl-CoAs into free fatty acids and coenzyme A, regulating mitochondrial lipid and amino acid metabolism. Mutations cause autosomal recessive infantile-onset spinocerebellar ataxia, affecting the central nervous system with early childhood presentation. The gene is highly intolerant to loss-of-function variants, supporting its essential role in mitochondrial metabolism.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 1.18
Clinical SummaryACOT9
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
96 unique Pathogenic / Likely Pathogenic· 51 VUS of 300 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.18LOEUF
pLI 0.000
Z-score 0.96
OE 0.77 (0.511.18)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.70Z-score
OE missense 0.85 (0.740.97)
144 obs / 169.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.77 (0.511.18)
00.351.4
Missense OE0.85 (0.740.97)
00.61.4
Synonymous OE0.81
01.21.6
LoF obs/exp: 15 / 19.6Missense obs/exp: 144 / 169.7Syn Z: 1.15

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic95
Likely Pathogenic1
VUS51
Likely Benign6
Benign2
95
Pathogenic
1
Likely Pathogenic
51
VUS
6
Likely Benign
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
95
0
95
Likely Pathogenic
0
0
1
0
1
VUS
0
42
9
0
51
Likely Benign
0
3
1
2
6
Benign
0
1
0
1
2
Total0461063155

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

View in ClinVar →

Protein Context — Lollipop Plot

ACOT9 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold

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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