MT-ATP6

Chr MT

ATP synthase F0 subunit 6

Also known as: ATPase6, MTATP6

This gene encodes ATP synthase F0 subunit 6, a component of Complex V that synthesizes ATP in the mitochondrial respiratory chain. Mutations cause NARP syndrome at 70-90% heteroplasmy levels, maternally inherited Leigh syndrome at >90% heteroplasmy, and Charcot-Marie-Tooth-like neuropathy, with phenotype severity strongly dependent on the degree of heteroplasmy. Inheritance is maternal through the mitochondrial genome.

OMIMResearchSummary from Curated mito context
Multiplemechanism
Clinical SummaryMT-ATP6
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Gene-Disease Validity (ClinGen)
mitochondrial disease · MTDefinitive

Definitive — sufficient evidence for diagnostic panels

2 total gene-disease associations curated

Population Genetics & Constraint

Constraint data not available from gnomAD.

Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveMT-ATP6-related neuropathy, ataxia, retinitis pigmentosa syndromeOTHERmitochondrial
DN
0.87top 5%
GOF
0.81top 10%
LOF
0.1499th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOFprediction 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

MT-ATP6 · 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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Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗