DENND1A

Chr 9

DENN domain containing 1A

Also known as: FAM31A, KIAA1608

DENND1A encodes a guanine nucleotide exchange factor that activates RAB35 and regulates clathrin-mediated endocytosis of synaptic vesicles and exit from early endosomes. Loss-of-function mutations cause neurodevelopmental disorders with an autosomal dominant inheritance pattern due to haploinsufficiency. The protein's critical role in synaptic vesicle endocytosis explains the neurological phenotype when cellular trafficking is disrupted.

Summary from RefSeq, UniProt, Mechanism
Research Assistant →
0
Active trials
12
Pubs (1 yr)
8
P/LP submissions
0%
P/LP missense
0.23
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryDENND1A
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
8 unique Pathogenic / Likely Pathogenic· 86 VUS of 100 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.23LOEUF
pLI 1.000
Z-score 5.60
OE 0.11 (0.060.23)
Highly constrained

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

Missense Constraint
1.26Z-score
OE missense 0.85 (0.790.92)
493 obs / 578.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.11 (0.060.23)
00.351.4
Missense OE0.85 (0.790.92)
00.61.4
Synonymous OE1.11
01.21.6
LoF obs/exp: 5 / 45.9Missense obs/exp: 493 / 578.3Syn Z: -1.37
DN
0.3892th %ile
GOF
0.5660th %ile
LOF
0.66top 25%

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

LOFprediction above median · LOEUF 0.23

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

100 submitted variants in ClinVar

Classification Summary

Pathogenic8
VUS86
Likely Benign4
Benign2
8
Pathogenic
86
VUS
4
Likely Benign
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
8
0
8
Likely Pathogenic
0
0
0
0
0
VUS
0
84
2
0
86
Likely Benign
0
3
0
1
4
Benign
0
1
0
1
2
Total088102100

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

View in ClinVar →

Protein Context — Lollipop Plot

DENND1A · 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.

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Clinical Literature
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