CTU2

Chr 16

cytosolic thiouridylase subunit 2

Also known as: C16orf84, MFRG, NCS2, UPF0432

This protein performs 2-thiolation of specific uridine residues at wobble positions in tRNAs, which enhances codon reading accuracy during protein synthesis. Biallelic mutations cause microcephaly, facial dysmorphism, renal agenesis, and ambiguous genitalia syndrome with autosomal recessive inheritance. The gene is not highly constrained against loss-of-function variants, which is consistent with its recessive inheritance pattern.

ResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismLOEUF 1.50
Clinical SummaryCTU2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
10 unique Pathogenic / Likely Pathogenic· 115 VUS of 200 total submissions
Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.50LOEUF
pLI 0.000
Z-score -0.53
OE 1.11 (0.831.50)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-3.95Z-score
OE missense 1.62 (1.511.75)
514 obs / 316.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.11 (0.831.50)
00.351.4
Missense OE1.62 (1.511.75)
00.61.4
Synonymous OE1.79
01.21.6
LoF obs/exp: 31 / 28.0Missense obs/exp: 514 / 316.4Syn Z: -7.23

ClinVar Variant Classifications

200 submitted variants in ClinVar

Classification Summary

Pathogenic10
VUS115
Likely Benign43
Benign3
Conflicting4
10
Pathogenic
115
VUS
43
Likely Benign
3
Benign
4
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
10
0
10
Likely Pathogenic
0
0
0
0
0
VUS
1
105
9
0
115
Likely Benign
1
4
19
19
43
Benign
0
1
1
1
3
Conflicting
4
Total21103920175

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

View in ClinVar →

Protein Context — Lollipop Plot

CTU2 · 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.

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Clinical Literature
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