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10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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ARHGAP31
Rho GTPase activating protein 31
Chromosome 3 Β· 3q13.32-q13.33
NCBI Gene: 57514Ensembl: ENSG00000031081.12HGNC: HGNC:29216UniProt: A0A8S0MHV1
35PubMed Papers
21Diseases
0Drugs
4Pathogenic Variants
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
protein bindingsmall GTPase-mediated signal transductionlamellipodiumGTPase activator activityAdams-Oliver syndromehypertensioncerebral palsyresponse to vaccine
✦AI Summary

ARHGAP31 encodes a GTPase-activating protein that functions as a negative regulator of CDC42 and RAC1 small GTPases, playing critical roles in cell migration, cytoskeletal organization, and developmental processes 1. The protein is required for proper cell spreading, polarized lamellipodia formation, and regulation of actin cytoskeletal structures through its GAP activity toward CDC42 and RAC1 12. During development, ARHGAP31 expression is restricted to terminal limb buds and craniofacial processes, sites that correspond to the anatomical defects observed in associated disorders 1. Loss-of-function mutations in ARHGAP31 cause Adams-Oliver syndrome, a rare developmental disorder characterized by scalp defects (aplasia cutis congenita) and terminal transverse limb defects 13. Interestingly, pathogenic variants typically result in gain-of-function effects through premature truncation of the C-terminal auto-inhibitory domain, leading to constitutively active ARHGAP31 that disrupts normal CDC42/RAC1 signaling 14. Beyond developmental roles, ARHGAP31 functions in intestinal stem cell responses to Wnt signaling through a DLG1-ARHGAP31-CDC42 regulatory axis 5 and may be involved in cancer-associated fibroblast biology 6. The clinical spectrum of ARHGAP31 mutations extends from isolated limb defects to complete Adams-Oliver syndrome, with variable penetrance observed among carriers 3.

Sources cited
1
ARHGAP31 functions as a GAP for CDC42/RAC1, causes Adams-Oliver syndrome through gain-of-function mutations, and is expressed in terminal limb buds during development
PMID: 21565291
2
ARHGAP31 mutations can cause isolated terminal limb defects with variable penetrance, extending beyond complete Adams-Oliver syndrome
PMID: 24668619
3
ARHGAP31 acts as a negative regulator during endothelial tubulogenesis by inactivating CDC42 and RAC
PMID: 26812085
4
ARHGAP31 participates in a DLG1-ARHGAP31-CDC42 axis essential for intestinal stem cell response to Wnt signaling
PMID: 36640764
5
ARHGAP31 is differentially expressed in cancer-associated fibroblasts and connected to Rho kinase signaling pathways
PMID: 23319410
6
Pathogenic ARHGAP31 variants result in truncated proteins with constitutively activated catalytic sites due to loss of C-terminal auto-inhibitory domain
PMID: 38790165
Disease Associationsβ“˜21
Adams-Oliver syndromeOpen Targets
0.75Strong
hypertensionOpen Targets
0.43Moderate
cerebral palsyOpen Targets
0.27Weak
response to vaccineOpen Targets
0.23Weak
celiac diseaseOpen Targets
0.21Weak
urinary system diseaseOpen Targets
0.21Weak
genetic disorderOpen Targets
0.19Weak
systemic lupus erythematosusOpen Targets
0.16Weak
Intellectual disabilityOpen Targets
0.15Weak
systemic sclerodermaOpen Targets
0.13Weak
autoimmune diseaseOpen Targets
0.12Weak
primary biliary cirrhosisOpen Targets
0.09Suggestive
breast cancerOpen Targets
0.09Suggestive
B-cell acute lymphoblastic leukemiaOpen Targets
0.09Suggestive
prostate cancerOpen Targets
0.07Suggestive
neoplasmOpen Targets
0.07Suggestive
cutaneous lupus erythematosusOpen Targets
0.05Suggestive
lymphatic malformation 11Open Targets
0.05Suggestive
hereditary angioedemaOpen Targets
0.04Suggestive
hereditary angioedema with normal C1InhOpen Targets
0.04Suggestive
Adams-Oliver syndrome 1UniProt
Pathogenic Variants4
NM_020754.4(ARHGAP31):c.1700del (p.Pro567fs)Likely pathogenic
Cerebral palsy
β˜…β˜†β˜†β˜†2021β†’ Residue 567
NM_020754.4(ARHGAP31):c.2182C>T (p.Gln728Ter)Pathogenic
Adams-Oliver syndrome 1
β˜…β˜†β˜†β˜†2017β†’ Residue 728
NM_020754.4(ARHGAP31):c.2047C>T (p.Gln683Ter)Pathogenic
Adams-Oliver syndrome 1
β˜†β˜†β˜†β˜†2011β†’ Residue 683
NM_020754.4(ARHGAP31):c.3260del (p.Lys1087fs)Pathogenic
Adams-Oliver syndrome 1
β˜†β˜†β˜†β˜†2011β†’ Residue 1087
View on ClinVar β†—
Related Genes
ARHGAP9Shared pathway100%MYLKProtein interaction88%KALRNProtein interaction88%CDC42Protein interaction86%ZEB2Protein interaction84%ITSN1Protein interaction75%
Tissue Expression6 tissues
Heart
100%
Lung
67%
Ovary
23%
Brain
22%
Bone Marrow
7%
Liver
6%
Gene Interaction Network
Click a node to explore
ARHGAP31ARHGAP9MYLKKALRNCDC42ZEB2ITSN1
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q2M1Z3
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.38Moderately Constrained
pLIβ“˜
1.00Intolerant
Observed/Expected LoF0.29 [0.22–0.38]
RankingsWhere ARHGAP31 stands among ~20K protein-coding genes
  • #10,912of 20,598
    Most Researched35
  • #3,823of 5,498
    Most Pathogenic Variants4
  • #1,836of 17,882
    Most Constrained (LOEUF)0.38 Β· top quartile
Genes detectedARHGAP31
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Mutations in NOTCH1 cause Adams-Oliver syndrome.
PMID: 25132448
Am J Hum Genet Β· 2014
1.00
2
Novel signatures of cancer-associated fibroblasts.
PMID: 23319410
Int J Cancer Β· 2013
0.90
3
Isolated terminal limb reduction defects: extending the clinical spectrum of Adams-Oliver syndrome and ARHGAP31 mutations.
PMID: 24668619
Am J Med Genet A Β· 2014
0.80
4
Cdc42 and k-Ras Control Endothelial Tubulogenesis through Apical Membrane and Cytoskeletal Polarization: Novel Stimulatory Roles for GTPase Effectors, the Small GTPases, Rac2 and Rap1b, and Inhibitory Influence of Arhgap31 and Rasa1.
PMID: 26812085
PLoS One Β· 2016
0.70
5
Gain-of-function mutations of ARHGAP31, a Cdc42/Rac1 GTPase regulator, cause syndromic cutis aplasia and limb anomalies.
PMID: 21565291
Am J Hum Genet Β· 2011
0.60