ATPAF2

Chr 17AR

ATP synthase mitochondrial F1 complex assembly factor 2

Also known as: ATP12, ATP12p, LP3663, MC5DN1

This protein functions as an assembly factor for the F1 component of mitochondrial ATP synthase, binding specifically to the F1 alpha subunit to prevent formation of nonproductive complexes during enzyme assembly. Mutations cause mitochondrial complex V (ATP synthase) deficiency with autosomal recessive inheritance. The gene shows very low constraint against loss-of-function variants (pLI near zero), consistent with recessive disease requiring biallelic mutations.

OMIMResearchSummary from RefSeq, OMIM, UniProt
ARLOEUF 1.441 OMIM phenotype
Clinical SummaryATPAF2
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Gene-Disease Validity (ClinGen)
mitochondrial disease · ARLimited

Limited evidence — not for standalone diagnostic reporting

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
119 unique Pathogenic / Likely Pathogenic· 155 VUS of 386 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.44LOEUF
pLI 0.000
Z-score 0.29
OE 0.92 (0.611.44)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.69Z-score
OE missense 0.85 (0.740.98)
142 obs / 167.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.92 (0.611.44)
00.351.4
Missense OE0.85 (0.740.98)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 14 / 15.2Missense obs/exp: 142 / 167.1Syn Z: 0.60

ClinVar Variant Classifications

386 submitted variants in ClinVar

Classification Summary

Pathogenic116
Likely Pathogenic3
VUS155
Likely Benign73
Benign18
Conflicting13
116
Pathogenic
3
Likely Pathogenic
155
VUS
73
Likely Benign
18
Benign
13
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
116
0
116
Likely Pathogenic
3
0
0
0
3
VUS
7
118
27
3
155
Likely Benign
0
6
37
30
73
Benign
0
1
16
1
18
Conflicting
13
Total1012519634378

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

ATPAF2 · 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.

Search ClinicalTrials.gov →
Clinical Literature
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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC