C18ORF63

Chr 18

chromosome 18 open reading frame 63

Also known as: DKFZP781G0119

The protein encoded by this gene functions as a component of the mitochondrial ribosome, specifically participating in mitochondrial protein synthesis. Mutations cause combined oxidative phosphorylation deficiency, which presents as a severe mitochondrial disorder with early-onset neurodegeneration, developmental delay, and multi-system involvement. The condition follows autosomal recessive inheritance, and the gene shows moderate constraint against loss-of-function variants.

0
Active trials
0
Pubs (1 yr)
153
P/LP submissions
P/LP missense
0.76
LOEUF
Mechanism
Clinical SummaryC18ORF63
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
151 unique Pathogenic / Likely Pathogenic· 17 VUS of 169 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.76LOEUF
pLI 0.000
Z-score 2.51
OE 0.48 (0.310.76)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.99Z-score
OE missense 0.68 (0.610.76)
207 obs / 304.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.48 (0.310.76)
00.351.4
Missense OE0.68 (0.610.76)
00.61.4
Synonymous OE0.69
01.21.6
LoF obs/exp: 13 / 27.1Missense obs/exp: 207 / 304.6Syn Z: 2.45

ClinVar Variant Classifications

169 submitted variants in ClinVar

Classification Summary

Pathogenic145
Likely Pathogenic6
VUS17
145
Pathogenic
6
Likely Pathogenic
17
VUS

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories· variant type breakdown unavailable

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
145
Likely Pathogenic
6
VUS
17
Likely Benign
0
Benign
0
Total168

Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

C18ORF63 · 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
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 1 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC

No open access results found