ESCO1

Chr 18

establishment of sister chromatid cohesion N-acetyltransferase 1

Also known as: A930014I12Rik, CTF, ECO1, EFO1, ESO1

The ESCO1 protein is an acetyltransferase that establishes sister chromatid cohesion during DNA replication by acetylating the cohesin component SMC3. Mutations cause Roberts syndrome, a rare autosomal recessive disorder characterized by severe growth retardation, limb malformations, and craniofacial abnormalities. The gene is highly constrained against loss-of-function variation, indicating its critical role in cellular function.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
6
Pubs (1 yr)
1
P/LP submissions
P/LP missense
0.17
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryESCO1
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
1 unique Pathogenic / Likely Pathogenic· 84 VUS of 100 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.17LOEUF
pLI 1.000
Z-score 5.34
OE 0.05 (0.020.17)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
0.43Z-score
OE missense 0.94 (0.871.02)
398 obs / 423.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.05 (0.020.17)
00.351.4
Missense OE0.94 (0.871.02)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 2 / 37.1Missense obs/exp: 398 / 423.0Syn Z: 0.19
DN
0.3197th %ile
GOF
0.1899th %ile
LOF
0.82top 5%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.17

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

100 submitted variants in ClinVar

Classification Summary

Likely Pathogenic1
VUS84
1
Likely Pathogenic
84
VUS

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
0
0
0
Likely Pathogenic
0
0
1
0
1
VUS
0
80
4
0
84
Likely Benign
0
0
0
0
0
Benign
0
0
0
0
0
Total0805085

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

View in ClinVar →

Protein Context — Lollipop Plot

ESCO1 · 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.

Search ClinicalTrials.gov →
Clinical Literature
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