ARHGEF9

Chr XX-linked

Cdc42 guanine nucleotide exchange factor 9

Also known as: COLLYBISTIN, DEE8, EIEE8, HPEM-2, PEM-2, PEM2

The protein acts as a guanine nucleotide exchange factor for CDC42 and facilitates recruitment of gephyrin at GABAergic and glycinergic synapses. Mutations cause developmental and epileptic encephalopathy 8 through an X-linked inheritance pattern, predominantly through loss-of-function mechanisms. The gene shows high constraint against loss-of-function variants, consistent with haploinsufficiency being pathogenic in this X-linked disorder.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismX-linkedLOEUF 0.151 OMIM phenotype
Clinical SummaryARHGEF9
🧬
Gene-Disease Validity (ClinGen)
X-linked complex neurodevelopmental disorder · XLDefinitive

Definitive — sufficient evidence for diagnostic panels

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.
💊
Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.15LOEUF
pLI 0.999
Z-score 4.12
OE 0.00 (0.000.15)
Highly constrained

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

Missense Constraint
3.04Z-score
OE missense 0.41 (0.340.49)
86 obs / 210.1 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.00 (0.000.15)
00.351.4
Missense OE0.41 (0.340.49)
00.61.4
Synonymous OE0.97
01.21.6
LoF obs/exp: 0 / 19.8Missense obs/exp: 86 / 210.1Syn Z: 0.19
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongARHGEF9-related developmental disorderLOFmonoallelic_X_heterozygous
DN
0.3196th %ile
GOF
0.5268th %ile
LOF
0.71top 10%

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

LOFprediction above median · LOEUF 0.15

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

ARHGEF9 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →