SCO2

Chr 22

synthesis of cytochrome C oxidase 2

Also known as: CEMCOX1, ECGF1, Gliostatin, MC4DN2, MYP6, PD-ECGF, SCO1L, TP

Cytochrome c oxidase (COX) catalyzes the transfer of electrons from cytochrome c to molecular oxygen, which helps to maintain the proton gradient across the inner mitochondrial membrane that is necessary for aerobic ATP production. Human COX is a multimeric protein complex that requires several assembly factors; this gene encodes one of the COX assembly factors. The encoded protein is a metallochaperone that is involved in the biogenesis of cytochrome c oxidase subunit II. Mutations in this gene are associated with fatal infantile encephalocardiomyopathy and myopia 6. [provided by RefSeq, Oct 2014]

ResearchGenerating clinical summary…
LOFmechanismLOEUF 1.76
Clinical SummarySCO2
🧬
Gene-Disease Validity (ClinGen)
Leigh syndrome · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

3 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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?
1.76LOEUF
pLI 0.000
Z-score -0.21
OE 1.08 (0.651.76)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint?
-1.61Z-score
OE missense 1.36 (1.221.52)
219 obs / 161.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios?
LoF OE?1.08 (0.651.76)
00.351.4
Missense OE?1.36 (1.221.52)
00.61.4
Synonymous OE?1.21
01.21.6
LoF obs/exp: 9 / 8.4Missense obs/exp: 219 / 161.4Syn Z: -1.39
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongSCO2-related myopiaOTHERAD
definitiveSCO2-related fatal infantile cardioencephalomyopathy due to cytochrome c oxidase deficiencyLOFAR

This gene — mechanism propensity

DN
0.6550th %ile
GOF
0.6150th %ile
LOF
0.3549th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction above median · 1 literature citation

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Literature Evidence

DNOverexpression of either wild-type SCO protein in the reciprocal patient background resulted in a dominant-negative phenotype, suggesting a physical interaction between SCO1 and SCO2.1

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

References

  1. 1.PMID 15229189

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

SCO2 · 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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