AIFM1

Chr XXLR

apoptosis inducing factor mitochondria associated 1

Also known as: AIF, AUNX1, CMT2D, CMTX4, COWCK, COXPD6, DFNX5, NADMR

AIFM1 encodes a mitochondrial flavoprotein that functions as an NADH oxidoreductase essential for respiratory chain biogenesis and also regulates apoptosis through caspase-independent DNA fragmentation. X-linked recessive mutations cause a spectrum of disorders including combined oxidative phosphorylation deficiency 6 (severe mitochondrial encephalomyopathy), Cowchock syndrome (X-linked Charcot-Marie-Tooth disease with neuropathy, deafness, and cognitive disability), X-linked deafness, and X-linked spondyloepimetaphyseal dysplasia with hypomyelinating leukodystrophy. The pathogenic mechanism involves loss of function, disrupting both mitochondrial respiratory chain function and cellular apoptotic regulation.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismXLRLOEUF 0.234 OMIM phenotypes
Clinical SummaryAIFM1
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Gene-Disease Validity (ClinGen)
Leigh syndrome · XLModerate

Moderate evidence — consider for supplementary testing

2 total gene-disease associations curated

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
10 unique Pathogenic / Likely Pathogenic· 55 VUS of 300 total submissions
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GeneReview available — AIFM1
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.23LOEUF
pLI 0.998
Z-score 4.45
OE 0.07 (0.030.23)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
2.49Z-score
OE missense 0.55 (0.470.63)
130 obs / 238.4 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.07 (0.030.23)
00.351.4
Missense OE0.55 (0.470.63)
00.61.4
Synonymous OE0.92
01.21.6
LoF obs/exp: 2 / 26.9Missense obs/exp: 130 / 238.4Syn Z: 0.57
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongAIFM1-related combined oxidative phosphorylation deficiencyOTHERXLR
DN
0.3992th %ile
GOF
0.4579th %ile
LOF
0.67top 25%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.23

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic4
Likely Pathogenic6
VUS55
Likely Benign37
Benign3
Conflicting1
4
Pathogenic
6
Likely Pathogenic
55
VUS
37
Likely Benign
3
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
4
0
4
Likely Pathogenic
0
5
1
0
6
VUS
0
49
4
2
55
Likely Benign
0
2
17
18
37
Benign
0
0
1
2
3
Conflicting
1
Total0562722106

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

View in ClinVar →

Protein Context — Lollipop Plot

AIFM1 · 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
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗