DCUN1D3

Chr 16

defective in cullin neddylation 1 domain containing 3

Also known as: 44M2.4, DCNL3, SCCRO3

The protein facilitates neddylation of cullin proteins, which regulates SCF ubiquitin E3 ligase complexes involved in cell cycle progression and DNA damage response. Mutations cause autosomal dominant developmental delay with seizures and dysmorphic features, typically presenting in infancy or early childhood. The gene is highly constrained against loss-of-function variants (pLI = 0.87, LOEUF = 0.43), consistent with its essential role in cellular regulation.

OMIMResearchSummary from RefSeq, UniProt
GOFmechanismLOEUF 0.43
Clinical SummaryDCUN1D3
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.87) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
14 unique Pathogenic / Likely Pathogenic· 37 VUS of 58 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.43LOEUF
pLI 0.867
Z-score 2.79
OE 0.09 (0.030.43)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.40Z-score
OE missense 0.70 (0.600.81)
119 obs / 170.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.09 (0.030.43)
00.351.4
Missense OE0.70 (0.600.81)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 1 / 11.0Missense obs/exp: 119 / 170.4Syn Z: 0.57
DN
0.3395th %ile
GOF
0.6443th %ile
LOF
0.60top 25%

The highest-scoring mechanism for this gene is gain-of-function.

GOFprediction 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

58 submitted variants in ClinVar

Classification Summary

Pathogenic14
VUS37
14
Pathogenic
37
VUS

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
14
0
14
Likely Pathogenic
0
0
0
0
0
VUS
0
31
6
0
37
Likely Benign
0
0
0
0
0
Benign
0
0
0
0
0
Total03120051

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

View in ClinVar →

Protein Context — Lollipop Plot

DCUN1D3 · 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
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 1 results · since 2015Search PubMed ↗