SCO1

Chr 17AR

synthesis of cytochrome C oxidase 1

The protein functions as a copper metallochaperone essential for assembly of cytochrome c oxidase (complex IV) by facilitating incorporation of subunits 1 and 2 into the holoenzyme within the mitochondrial inner membrane. Mutations cause mitochondrial complex IV deficiency with autosomal recessive inheritance through a predicted dominant-negative mechanism. The low pLI score indicates tolerance to heterozygous loss-of-function variants, consistent with the recessive inheritance pattern.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.991 OMIM phenotype
Clinical SummarySCO1
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Gene-Disease Validity (ClinGen)
mitochondrial disease · 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.
Some data sources returned errors (1)

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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.99LOEUF
pLI 0.000
Z-score 1.59
OE 0.55 (0.320.99)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.20Z-score
OE missense 1.04 (0.921.18)
177 obs / 169.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.55 (0.320.99)
00.351.4
Missense OE1.04 (0.921.18)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 8 / 14.6Missense obs/exp: 177 / 169.8Syn Z: -0.54
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveSCO1-related mitochondrial complex IV deficiencyLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.74top 25%
GOF
0.6053th %ile
LOF
0.2775th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

SCO1 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

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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