PDK3

Chr XXLD

pyruvate dehydrogenase kinase 3

Also known as: CMTX6, GS1-358P8.4

PDK3 encodes pyruvate dehydrogenase kinase 3, which inhibits the pyruvate dehydrogenase complex by phosphorylating its E1 subunit, thereby regulating the conversion of pyruvate to acetyl-CoA and controlling glucose metabolism. Mutations cause X-linked dominant Charcot-Marie-Tooth disease type 6, a peripheral neuropathy. The gene is predominantly expressed in heart and skeletal muscle and shows moderate constraint against loss-of-function variants (LOEUF 0.546).

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismXLDLOEUF 0.551 OMIM phenotype
Clinical SummaryPDK3
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Gene-Disease Validity (ClinGen)
Charcot-Marie-Tooth disease X-linked dominant 6 · XLDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.21) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.55LOEUF
pLI 0.445
Z-score 2.75
OE 0.21 (0.100.55)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.32Z-score
OE missense 0.46 (0.380.56)
67 obs / 145.8 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.21 (0.100.55)
00.351.4
Missense OE0.46 (0.380.56)
00.61.4
Synonymous OE0.87
01.21.6
LoF obs/exp: 3 / 14.2Missense obs/exp: 67 / 145.8Syn Z: 0.77
DN
0.6357th %ile
GOF
0.6150th %ile
LOF
0.3939th %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

PDK3 · 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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