ATP5PO

Chr 21AR

ATP synthase peripheral stalk subunit OSCP

Also known as: ATP5O, ATPO, HMC08D05, MC5DN7, OSCP

The protein is a subunit of mitochondrial ATP synthase (Complex V) that helps link the catalytic and membrane domains and maintains structural stability during ATP production from ADP. Mutations cause mitochondrial complex V deficiency with autosomal recessive inheritance, resulting in a mitochondrial disorder that affects cellular energy production. This gene has a GeneReviews entry available for detailed clinical guidance.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismARLOEUF 0.781 OMIM phenotype
Clinical SummaryATP5PO
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Gene-Disease Validity (ClinGen)
mitochondrial disease · ARStrong

Strong evidence — appropriate for clinical testing

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.34) despite low pLI — interpret in context.
📖
GeneReview available — ATP5PO
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.78LOEUF
pLI 0.058
Z-score 2.10
OE 0.34 (0.170.78)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.03Z-score
OE missense 0.99 (0.851.16)
117 obs / 117.8 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.34 (0.170.78)
00.351.4
Missense OE0.99 (0.851.16)
00.61.4
Synonymous OE1.09
01.21.6
LoF obs/exp: 4 / 11.8Missense obs/exp: 117 / 117.8Syn Z: -0.48
DN
0.73top 25%
GOF
0.4777th %ile
LOF
0.2386th %ile

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

DNprediction above median

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.

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ATP5PO · 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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