CHCHD4

Chr 3

coiled-coil-helix-coiled-coil-helix domain containing 4

Also known as: MIA40, TIMM40

The CHCHD4 protein functions as a central component of mitochondrial intermembrane space import machinery, serving as a chaperone that catalyzes disulfide bond formation in substrate proteins required for respiratory chain complex biogenesis. Mutations in CHCHD4 cause autosomal recessive mitochondrial complex IV deficiency, nuclear type 11, which presents as a severe mitochondrial respiratory chain disorder. This gene follows autosomal recessive inheritance and affects mitochondrial energy metabolism through impaired assembly of cytochrome c oxidase and other respiratory complexes.

OMIMResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 0.95
Clinical SummaryCHCHD4
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.30) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.95LOEUF
pLI 0.187
Z-score 1.66
OE 0.30 (0.120.95)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.88Z-score
OE missense 0.73 (0.600.90)
63 obs / 85.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.30 (0.120.95)
00.351.4
Missense OE0.73 (0.600.90)
00.61.4
Synonymous OE1.15
01.21.6
LoF obs/exp: 2 / 6.6Missense obs/exp: 63 / 85.9Syn Z: -0.65
DN
0.6162th %ile
GOF
0.4677th %ile
LOF
0.2581th %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

CHCHD4 · 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 →