XRN2

Chr 20

5'-3' exoribonuclease 2

The protein encoded by this gene is a 5'-3' exoribonuclease that promotes transcription termination by RNA polymerase II through degradation of cleaved RNA fragments. Biallelic pathogenic variants cause Chudley-McCullough syndrome, an autosomal recessive disorder characterized by early-onset sensorineural hearing loss and brain malformations including hydrocephalus, agenesis of the corpus callosum, and colpocephaly. The gene is highly constrained against loss-of-function variants (LOEUF 0.355), indicating that such variants are likely to be pathogenic.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
13
Pubs (1 yr)
25
P/LP submissions
0%
P/LP missense
0.35
LOEUF
Mechanism
Clinical SummaryXRN2
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.23) despite low pLI — interpret in context.
📋
ClinVar Variants
25 unique Pathogenic / Likely Pathogenic· 90 VUS of 166 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.35LOEUF
pLI 0.349
Z-score 5.62
OE 0.23 (0.150.35)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.50Z-score
OE missense 0.69 (0.630.75)
359 obs / 519.3 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.23 (0.150.35)
00.351.4
Missense OE0.69 (0.630.75)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 14 / 61.6Missense obs/exp: 359 / 519.3Syn Z: 0.44

ClinVar Variant Classifications

166 submitted variants in ClinVar

Classification Summary

Pathogenic22
Likely Pathogenic3
VUS90
Likely Benign2
Benign1
22
Pathogenic
3
Likely Pathogenic
90
VUS
2
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
22
0
22
Likely Pathogenic
0
0
3
0
3
VUS
0
85
5
0
90
Likely Benign
0
2
0
0
2
Benign
0
1
0
0
1
Total088300118

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

View in ClinVar →

Protein Context — Lollipop Plot

XRN2 · 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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