UBE3D

Chr 6

ubiquitin protein ligase E3D

Also known as: C6orf157, H10BH, UBE2CBP, YJR141W

The protein is an E3 ubiquitin ligase that transfers ubiquitin to target proteins for proteasome-mediated degradation and also inhibits CPSF3 endonuclease activity. Mutations cause autosomal recessive intellectual disability with developmental delay and behavioral abnormalities. The gene shows minimal constraint against loss-of-function variants, consistent with a recessive inheritance pattern.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 0.99
Clinical SummaryUBE3D
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
14 unique Pathogenic / Likely Pathogenic· 65 VUS of 97 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.99LOEUF
pLI 0.000
Z-score 1.60
OE 0.62 (0.400.99)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.28Z-score
OE missense 0.94 (0.841.06)
196 obs / 207.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.62 (0.400.99)
00.351.4
Missense OE0.94 (0.841.06)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 13 / 20.9Missense obs/exp: 196 / 207.5Syn Z: -0.46

ClinVar Variant Classifications

97 submitted variants in ClinVar

Classification Summary

Pathogenic14
VUS65
Likely Benign8
14
Pathogenic
65
VUS
8
Likely Benign

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
58
7
0
65
Likely Benign
0
8
0
0
8
Benign
0
0
0
0
0
Total06621087

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

View in ClinVar →

Protein Context — Lollipop Plot

UBE3D · 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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