NCOA1

Chr 2

nuclear receptor coactivator 1

Also known as: F-SRC-1, KAT13A, RIP160, SRC1, bHLHe42, bHLHe74

NCOA1 encodes a nuclear receptor coactivator that directly binds steroid and nuclear hormone receptors to stimulate hormone-dependent gene transcription and has histone acetyltransferase activity. Mutations cause autosomal dominant intellectual disability and developmental delay, often with additional features including growth abnormalities and endocrine dysfunction. This gene is extremely intolerant to loss-of-function variants (pLI = 1.0, LOEUF = 0.25), indicating that functional copies are critical for normal development.

Summary from RefSeq, UniProt
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1
Active trials
42
Pubs (1 yr)
20
P/LP submissions
0%
P/LP missense
0.25
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryNCOA1
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.
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ClinVar Variants
20 unique Pathogenic / Likely Pathogenic· 285 VUS of 462 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.25LOEUF
pLI 1.000
Z-score 6.56
OE 0.15 (0.090.25)
Highly constrained

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

Missense Constraint
2.35Z-score
OE missense 0.76 (0.710.82)
599 obs / 784.5 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.15 (0.090.25)
00.351.4
Missense OE0.76 (0.710.82)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 10 / 68.6Missense obs/exp: 599 / 784.5Syn Z: 0.57
DN
0.2798th %ile
GOF
0.1799th %ile
LOF
0.85top 5%

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

LOFprediction above median · LOEUF 0.25

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

462 submitted variants in ClinVar

Classification Summary

Pathogenic19
Likely Pathogenic1
VUS285
Likely Benign121
Benign5
19
Pathogenic
1
Likely Pathogenic
285
VUS
121
Likely Benign
5
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
19
0
19
Likely Pathogenic
0
0
1
0
1
VUS
2
264
17
2
285
Likely Benign
0
5
13
103
121
Benign
0
0
1
4
5
Total226951109431

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

View in ClinVar →

Protein Context — Lollipop Plot

NCOA1 · 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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