PDHX

Chr 11AR

pyruvate dehydrogenase complex component X

Also known as: DLDBP, E3BP, OPDX, PDHXD, PDX1, proX

The PDHX protein serves as the E3 binding protein that anchors dihydrolipoamide dehydrogenase to the core of the mitochondrial pyruvate dehydrogenase complex, which is essential for converting pyruvate to acetyl-CoA and linking glycolysis to the Krebs cycle. Autosomal recessive mutations cause pyruvate dehydrogenase deficiency presenting with neurological dysfunction and lactic acidosis in infancy and early childhood.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.721 OMIM phenotype
Clinical SummaryPDHX
🧬
Gene-Disease Validity (ClinGen)
Leigh syndrome · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

2 total gene-disease associations curated

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
0.72LOEUF
pLI 0.000
Z-score 2.64
OE 0.43 (0.270.72)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.20Z-score
OE missense 1.03 (0.941.14)
289 obs / 279.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.43 (0.270.72)
00.351.4
Missense OE1.03 (0.941.14)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 11 / 25.4Missense obs/exp: 289 / 279.4Syn Z: -0.15
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePDHX-related lacticacidemia due to PDX1 deficiencyLOFAR

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

DN
0.6743th %ile
GOF
0.5758th %ile
LOF
0.4038th %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

PDHX · 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 →