PDHA2

Chr 4AR

pyruvate dehydrogenase E1 subunit alpha 2

Also known as: PDHAL, SPGF70

This protein forms part of the E1 subunit of the testis-specific pyruvate dehydrogenase complex, which catalyzes the conversion of pyruvate to acetyl-CoA and links glycolysis to the mitochondrial tricarboxylic acid cycle. Mutations cause spermatogenic failure 70, an autosomal recessive condition affecting male fertility. The gene shows low constraint to loss-of-function variants (pLI near zero, LOEUF 1.77), consistent with its tissue-specific expression and reproductive phenotype.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Spermatogenic failure 70MIM #619828
AR
0
Active trials
7
Pubs (1 yr)
18
P/LP submissions
6%
P/LP missense
1.77
LOEUF
DN
Mechanism· predicted
Clinical SummaryPDHA2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
16 unique Pathogenic / Likely Pathogenic· 61 VUS of 79 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.77LOEUF
pLI 0.000
Z-score -0.45
OE 1.15 (0.731.77)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.26Z-score
OE missense 1.05 (0.941.17)
242 obs / 230.9 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.15 (0.731.77)
00.351.4
Missense OE1.05 (0.941.17)
00.61.4
Synonymous OE1.06
01.21.6
LoF obs/exp: 12 / 10.4Missense obs/exp: 242 / 230.9Syn Z: -0.43
DN
0.76top 25%
GOF
0.6248th %ile
LOF
0.3066th %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

79 submitted variants in ClinVar

Classification Summary

Pathogenic13
Likely Pathogenic3
VUS61
Benign2
13
Pathogenic
3
Likely Pathogenic
61
VUS
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
1
12
0
13
Likely Pathogenic
0
0
3
0
3
VUS
0
58
3
0
61
Likely Benign
0
0
0
0
0
Benign
0
2
0
0
2
Total06118079

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

View in ClinVar →

Protein Context — Lollipop Plot

PDHA2 · 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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