RSU1

Chr 10

Ras suppressor protein 1

Also known as: RSP-1

The RSU1 protein functions in Ras signal transduction and suppresses cellular transformation. Mutations cause autosomal recessive intellectual disability with seizures and brain abnormalities. This gene is not highly constrained against loss-of-function variants, consistent with recessive inheritance where one functional copy is insufficient.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
4
Pubs (1 yr)
15
P/LP submissions
0%
P/LP missense
0.95
LOEUF
DN
Mechanism· predicted
Clinical SummaryRSU1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
15 unique Pathogenic / Likely Pathogenic· 46 VUS of 92 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.95LOEUF
pLI 0.001
Z-score 1.69
OE 0.51 (0.290.95)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.46Z-score
OE missense 0.90 (0.781.03)
142 obs / 158.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.51 (0.290.95)
00.351.4
Missense OE0.90 (0.781.03)
00.61.4
Synonymous OE1.17
01.21.6
LoF obs/exp: 7 / 13.8Missense obs/exp: 142 / 158.2Syn Z: -1.12
DN
0.6260th %ile
GOF
0.5169th %ile
LOF
0.48top 25%

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction 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

92 submitted variants in ClinVar

Classification Summary

Pathogenic15
VUS46
Likely Benign3
Benign3
15
Pathogenic
46
VUS
3
Likely Benign
3
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
15
0
15
Likely Pathogenic
0
0
0
0
0
VUS
0
36
10
0
46
Likely Benign
0
0
2
1
3
Benign
0
0
3
0
3
Total03630167

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

View in ClinVar →

Protein Context — Lollipop Plot

RSU1 · 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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