UBXN11

Chr 1

UBX domain protein 11

Also known as: COA-1, PP2243, SOC, SOCI, UBXD5

The protein contains a UBX domain and regulates actin cytoskeleton reorganization by interacting with Rho GTPases and promoting RHOA activation through G-protein signaling pathways. Mutations cause autosomal recessive intellectual disability with seizures and characteristic dysmorphic features. This gene is extremely intolerant to loss-of-function variants, indicating that complete protein loss is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 1.14
Clinical SummaryUBXN11
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
5 unique Pathogenic / Likely Pathogenic· 92 VUS of 136 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.14LOEUF
pLI 0.000
Z-score 0.96
OE 0.81 (0.581.14)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.32Z-score
OE missense 0.95 (0.861.04)
299 obs / 314.8 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.81 (0.581.14)
00.351.4
Missense OE0.95 (0.861.04)
00.61.4
Synonymous OE1.20
01.21.6
LoF obs/exp: 23 / 28.5Missense obs/exp: 299 / 314.8Syn Z: -1.82
DN
0.6453th %ile
GOF
0.5366th %ile
LOF
0.3648th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction 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

136 submitted variants in ClinVar

Classification Summary

Pathogenic5
VUS92
Likely Benign9
5
Pathogenic
92
VUS
9
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
5
0
5
Likely Pathogenic
0
0
0
0
0
VUS
0
87
5
0
92
Likely Benign
0
8
0
1
9
Benign
0
0
0
0
0
Total095101106

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

View in ClinVar →

Protein Context — Lollipop Plot

UBXN11 · 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 →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC