ERCC8

Chr 5

ERCC excision repair 8, CSA ubiquitin ligase complex subunit

Also known as: CKN1, CSA, UVSS2

This gene encodes a WD repeat protein, which interacts with Cockayne syndrome type B (CSB) protein and with p44 protein, a subunit of the RNA polymerase II transcription factor IIH. Mutations in this gene have been identified in patients with hereditary disease Cockayne syndrome (CS). CS cells are abnormally sensitive to ultraviolet radiation and are defective in the repair of transcriptionally active genes. Several transcript variants encoding different isoforms have been found for this gene. [provided by RefSeq, Mar 2014]

GeneReviewsResearchGenerating clinical summary…

Primary Disease Associations & Inheritance

UniProtCockayne syndrome A
UniProtUV-sensitive syndrome 2

Clinical highlights

Gene-disease validity (ClinGen)
Cockayne syndrome type 1 · ARDefinitivesufficient evidence for diagnostic panels
Interpreting a novel variant
Loss of function is the curated mechanism (Gene2Phenotype), though the gene is not strongly LoF-constrained in the population — weigh truncating variants against that tolerance.Curated gene-level mechanism — a prior for triage, not a per-variant call.
0
Active trials
15
Pubs (1 yr)
P/LP submissions
P/LP missense
1.35
LOEUF
LOF
Mechanism· G2P
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GeneReview available — ERCC8
Authoritative clinical overview · Recommended first read
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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?
1.35LOEUF
pLI 0.000
Z-score 0.20
OE 0.96 (0.691.35)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint?
-0.40Z-score
OE missense 1.08 (0.971.21)
221 obs / 204.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios?
LoF OE?0.96 (0.691.35)
00.351.4
Missense OE?1.08 (0.971.21)
00.61.4
Synonymous OE?1.01
01.21.6
LoF obs/exp: 23 / 24.0Missense obs/exp: 221 / 204.8Syn Z: -0.09

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ERCC8 · 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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