RPUSD1

Chr 16

RNA pseudouridine synthase domain containing 1

Also known as: C16orf40, RLUCL

Predicted to enable pseudouridine synthase activity. Predicted to be involved in enzyme-directed rRNA pseudouridine synthesis. [provided by Alliance of Genome Resources, Jul 2025]

0
Active trials
47
Pathogenic / LP
164
ClinVar variants
2
Pubs (1 yr)
Missense Z
LOEUF
Clinical SummaryRPUSD1
📋
ClinVar Variants
47 Pathogenic / Likely Pathogenic· 107 VUS of 164 total submissions
Some data sources returned errors (2)

gnomad: Error: gnomAD API error: 502

pubtator: Error: PubTator3 HTTP 502

Population Genetics & Constraint

Constraint data not available from gnomAD.

DN
0.5477th %ile
GOF
0.6931th %ile
LOF
0.3841th %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

164 submitted variants in ClinVar

Classification Summary

Pathogenic47
VUS107
Likely Benign7
Benign3
47
Pathogenic
107
VUS
7
Likely Benign
3
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
47
0
47
Likely Pathogenic
0
0
0
0
0
VUS
0
89
18
0
107
Likely Benign
0
5
1
1
7
Benign
0
0
3
0
3
Total094691164

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

View in ClinVar →

Protein Context — Lollipop Plot

RPUSD1 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Landmark / reviewRecent case evidence