SH3BP1

Chr 22

SH3 domain binding protein 1

Also known as: ARHGAP43

The protein functions as a GTPase activating protein that specifically inactivates RAC1 and CDC42, regulating actin cytoskeleton dynamics, cell migration, epithelial junction formation, and angiogenesis. Mutations cause autosomal recessive neurodevelopmental disorders with intellectual disability, developmental delay, and dysmorphic features. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.54), suggesting some tolerance to protein loss.

OMIMResearchSummary from RefSeq, UniProt
GOFmechanismLOEUF 0.54
Clinical SummarySH3BP1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.31) despite low pLI — interpret in context.
📋
ClinVar Variants
20 unique Pathogenic / Likely Pathogenic· 121 VUS of 176 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.54LOEUF
pLI 0.010
Z-score 3.45
OE 0.31 (0.190.54)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.58Z-score
OE missense 0.78 (0.710.85)
305 obs / 393.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.31 (0.190.54)
00.351.4
Missense OE0.78 (0.710.85)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 9 / 29.1Missense obs/exp: 305 / 393.0Syn Z: -0.24
DN
0.6064th %ile
GOF
0.6444th %ile
LOF
0.3842th %ile

The highest-scoring mechanism for this gene is gain-of-function.

GOFprediction 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

176 submitted variants in ClinVar

Classification Summary

Pathogenic19
Likely Pathogenic1
VUS121
Likely Benign4
Benign2
Conflicting1
19
Pathogenic
1
Likely Pathogenic
121
VUS
4
Likely Benign
2
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
19
0
19
Likely Pathogenic
0
0
1
0
1
VUS
0
119
2
0
121
Likely Benign
0
2
1
1
4
Benign
0
0
1
1
2
Conflicting
1
Total0121242148

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

View in ClinVar →

Protein Context — Lollipop Plot

SH3BP1 · 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