CNOT3

Chr 19AD

CCR4-NOT transcription complex subunit 3

Also known as: IDDSADF, LENG2, NOT3, NOT3H

CNOT3 encodes a component of the CCR4-NOT complex, a major cellular mRNA deadenylase that regulates mRNA degradation, translational repression, and transcriptional regulation, and is involved in stem cell maintenance and mitotic progression. Mutations cause autosomal dominant intellectual developmental disorder with speech delay, autism, and dysmorphic facies. The gene is highly constrained against loss-of-function variants (pLI >0.99, LOEUF 0.076), indicating that even single functional copies are critical for normal development.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 0.081 OMIM phenotype
Clinical SummaryCNOT3
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Gene-Disease Validity (ClinGen)
complex neurodevelopmental disorder · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

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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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.08LOEUF
pLI 1.000
Z-score 5.81
OE 0.00 (0.000.08)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
3.76Z-score
OE missense 0.50 (0.450.56)
230 obs / 456.3 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.00 (0.000.08)
00.351.4
Missense OE0.50 (0.450.56)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 0 / 39.3Missense obs/exp: 230 / 456.3Syn Z: -0.56
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveCNOT3-related neurodevelopmental disorderLOFAD
DN
0.2499th %ile
GOF
0.2696th %ile
LOF
0.84top 5%

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

LOFprediction above median · 1 literature citation · LOEUF 0.08

Literature Evidence

LOFHaploinsufficiency appears to be the most likely mechanism of action, with eight cases found to have protein-truncating variants.PMID:31201375

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CNOT3 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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