DENND5B

Chr 12

DENN domain containing 5B

The protein functions as a guanine nucleotide exchange factor that activates RAB39A and RAB39B by promoting GDP to GTP exchange. Mutations cause autosomal recessive intellectual disability with onset in infancy or early childhood. This gene is highly constrained against loss-of-function variation (pLI = 1.0, LOEUF = 0.155), indicating that complete loss of protein function is likely not tolerated.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
3
Pubs (1 yr)
34
P/LP submissions
3%
P/LP missense
0.15
LOEUF· LoF intol.
LOF
Mechanism· G2P
Clinical SummaryDENND5B
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.
📋
ClinVar Variants
33 unique Pathogenic / Likely Pathogenic· 167 VUS of 241 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.15LOEUF
pLI 1.000
Z-score 6.65
OE 0.07 (0.030.15)
Highly constrained

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

Missense Constraint
3.39Z-score
OE missense 0.63 (0.580.68)
413 obs / 657.5 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.07 (0.030.15)
00.351.4
Missense OE0.63 (0.580.68)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 4 / 59.2Missense obs/exp: 413 / 657.5Syn Z: 0.48

ClinVar Variant Classifications

241 submitted variants in ClinVar

Classification Summary

Pathogenic29
Likely Pathogenic4
VUS167
Likely Benign21
29
Pathogenic
4
Likely Pathogenic
167
VUS
21
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
29
0
29
Likely Pathogenic
0
1
3
0
4
VUS
4
153
10
0
167
Likely Benign
0
13
0
8
21
Benign
0
0
0
0
0
Total4167428221

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

View in ClinVar →

Protein Context — Lollipop Plot

DENND5B · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC