ARFGEF1

Chr 8AD

ARF guanine nucleotide exchange factor 1

Also known as: ARFGEP1, BIG1, DEDISB, P200

The protein encoded by ARFGEF1 promotes guanine-nucleotide exchange on ARF1 and ARF3 to activate these proteins for vesicular trafficking, maintains Golgi structure, and is required for integrin beta-1 maturation. Mutations cause autosomal dominant developmental delay, impaired speech, and behavioral abnormalities with or without seizures. This gene is highly constrained against loss-of-function variants (pLI >0.99), indicating that functional copies are critical for normal development.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 0.141 OMIM phenotype
Clinical SummaryARFGEF1
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.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.14LOEUF
pLI 1.000
Z-score 8.58
OE 0.08 (0.040.14)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
5.37Z-score
OE missense 0.51 (0.470.55)
479 obs / 942.9 exp
Constrained

Extremely missense-constrained (top ~0.01%)

Observed / Expected Ratios
LoF OE0.08 (0.040.14)
00.351.4
Missense OE0.51 (0.470.55)
00.61.4
Synonymous OE1.03
01.21.6
LoF obs/exp: 8 / 101.1Missense obs/exp: 479 / 942.9Syn Z: -0.38
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongARFGEF1-related intellectual disability and epilepsyLOFAD
DN
0.3693th %ile
GOF
0.5170th %ile
LOF
0.65top 25%

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

LOFprediction above median · LOEUF 0.14

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ARFGEF1 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗