MTFMT

Chr 15AR

mitochondrial methionyl-tRNA formyltransferase

Also known as: COXPD15, FMT1, MC1DN27

The protein catalyzes the formylation of methionyl-tRNA within mitochondria, a critical step in mitochondrial protein synthesis. Mutations cause combined oxidative phosphorylation deficiency 15 and mitochondrial complex I deficiency through autosomal recessive inheritance. The pathogenic mechanism involves de novo mutations that disrupt mitochondrial translation and respiratory chain function.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 1.012 OMIM phenotypes
Clinical SummaryMTFMT
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Gene-Disease Validity (ClinGen)
Leigh syndrome · 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.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.01LOEUF
pLI 0.000
Z-score 1.53
OE 0.64 (0.421.01)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.46Z-score
OE missense 1.09 (0.981.22)
214 obs / 195.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.64 (0.421.01)
00.351.4
Missense OE1.09 (0.981.22)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 14 / 21.7Missense obs/exp: 214 / 195.8Syn Z: -0.38
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveMTFMT-related mitochondrial disease with regression and lactic acidosisLOFAR

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

DN
0.6258th %ile
GOF
0.4973th %ile
LOF
0.3261th %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

MTFMT · 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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