RNF168

Chr 3AR

ring finger protein 168

Also known as: RIDL, hRNF168

RNF168 encodes an E3 ubiquitin ligase that is required for accumulating DNA repair proteins at sites of double-strand breaks and interstrand cross-links, promoting histone ubiquitination to recruit key repair factors like TP53BP1 and BRCA1. Biallelic mutations cause RIDDLE syndrome, an autosomal recessive disorder characterized by radiosensitivity, immunodeficiency, dysmorphic features, and learning difficulties. The gene is not highly constrained against loss-of-function variants (pLI near zero), consistent with the recessive inheritance pattern where heterozygous carriers are typically unaffected.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

RIDDLE syndromeMIM #611943
AR
0
Active trials
20
Pubs (1 yr)
73
P/LP submissions
0%
P/LP missense
1.20
LOEUF
LOF
Mechanism· G2P
Clinical SummaryRNF168
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Gene-Disease Validity (ClinGen)
RIDDLE syndrome · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
66 unique Pathogenic / Likely Pathogenic· 187 VUS of 410 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.20LOEUF
pLI 0.000
Z-score 0.76
OE 0.84 (0.601.20)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.39Z-score
OE missense 1.06 (0.971.17)
319 obs / 299.9 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.84 (0.601.20)
00.351.4
Missense OE1.06 (0.971.17)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 22 / 26.2Missense obs/exp: 319 / 299.9Syn Z: -0.51

ClinVar Variant Classifications

410 submitted variants in ClinVar

Classification Summary

Pathogenic55
Likely Pathogenic11
VUS187
Likely Benign130
Benign5
Conflicting11
55
Pathogenic
11
Likely Pathogenic
187
VUS
130
Likely Benign
5
Benign
11
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
20
0
35
0
55
Likely Pathogenic
6
0
5
0
11
VUS
4
167
15
1
187
Likely Benign
0
3
40
87
130
Benign
0
2
3
0
5
Conflicting
11
Total301729888399

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

View in ClinVar →

Protein Context — Lollipop Plot

RNF168 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗