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10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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PDHA2
pyruvate dehydrogenase E1 subunit alpha 2
Chromosome 4 Β· 4q22.3
NCBI Gene: 5161Ensembl: ENSG00000163114.6HGNC: HGNC:8807UniProt: P29803
37PubMed Papers
21Diseases
0Drugs
1Pathogenic Variants
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
pyruvate decarboxylation to acetyl-CoApyruvate dehydrogenase (acetyl-transferring) activityprotein bindingmitochondrionspermatogenic failure 70male infertility with azoospermia or oligozoospermia due to single gene mutationazoospermiaspermatogenic failure 1
✦AI Summary

PDHA2 encodes the E1-alpha subunit of the pyruvate dehydrogenase (PDH) complex, a heterotetrameric enzyme that catalyzes the conversion of pyruvate to acetyl-CoA and CO2, linking glycolysis and the mitochondrial TCA cycle 1. The E1 subunit performs thiamine pyrophosphate-dependent decarboxylation of pyruvate and reductive acetylation of lipoyl groups on the E2 subunit 2. Unlike its X-linked somatic paralogue PDHA1, PDHA2 is exclusively expressed in testicular germ cells, regulated by demethylation of a CpG island within its coding region 34. PDHA2 is essential for spermatogenesis, functioning in mitochondrial ATP production required for meiotic double-strand break repair through RAD51 and DMC1 ATPase activity 5. Loss-of-function mutations cause non-obstructive azoospermia (NOA) with meiotic arrest 67, and PDHA2 variants have been identified in consanguineous families with combined digit malformation and male infertility 8. Clinical relevance includes NOA diagnosis and potential therapeutic approaches involving PDHA2 mRNA delivery via lipid nanoparticles, which successfully restored spermatogenesis in knockout mice 7.

Sources cited
1
PDHA2 together with PDHB forms the heterotetrameric E1 subunit of PDH complex in testis
PMID: 14638692
2
E1 subunit catalyzes TPP-dependent decarboxylation and reductive acetylation of lipoyl groups
PMID: 16436377
3
PDHA2 is exclusively expressed in spermatogenic cells; methylation of CpG island in coding region correlates with transcriptional activity
PMID: 20005141
4
Demethylation of PDHA2 coding region triggers gene activation in somatic tissues
PMID: 22675509
5
PDHA2 compensates for silenced PDHA1 during meiosis and is essential for ATP production required for double-strand break repair
PMID: 40679056
6
PDHA2 mutations identified in NOA patients; defects in meiotic genes associated with unsuccessful sperm retrieval
PMID: 35172124
7
LNP-mediated PDHA2 mRNA delivery restores spermatogenesis and fertility in PDHA2 knockout NOA model
PMID: 41082659
8
Homozygous PDHA2 variant associated with male infertility including azoospermia and sperm dysfunction
PMID: 29581481
Disease Associationsβ“˜21
spermatogenic failure 70Open Targets
0.40Weak
male infertility with azoospermia or oligozoospermia due to single gene mutationOpen Targets
0.37Weak
azoospermiaOpen Targets
0.33Weak
spermatogenic failure 1Open Targets
0.33Weak
smoking initiationOpen Targets
0.25Weak
preeclampsiaOpen Targets
0.24Weak
diaphragm diseaseOpen Targets
0.23Weak
strictureOpen Targets
0.23Weak
Genu valgumOpen Targets
0.20Weak
Genu varumOpen Targets
0.20Weak
male infertilityOpen Targets
0.20Weak
cervical carcinomaOpen Targets
0.20Weak
aortic diseaseOpen Targets
0.19Weak
Alzheimer diseaseOpen Targets
0.17Weak
lysosomal storage diseaseOpen Targets
0.17Weak
multiple sclerosisOpen Targets
0.17Weak
neurodegenerative diseaseOpen Targets
0.17Weak
Parkinson diseaseOpen Targets
0.17Weak
bone remodeling diseaseOpen Targets
0.16Weak
bronchopneumoniaOpen Targets
0.14Weak
Spermatogenic failure 70UniProt
Pathogenic Variants1
NM_005390.5(PDHA2):c.679A>G (p.Met227Val)Pathogenic
Oligosynaptic infertility|Azoospermia|Spermatogenic failure 70
β˜†β˜†β˜†β˜†2022β†’ Residue 227
View on ClinVar β†—
Related Genes
PDHA1Shared pathway100%PDHXProtein interaction100%DLSTProtein interaction100%DBTProtein interaction100%TALDO1Protein interaction100%BCKDHBProtein interaction100%
Tissue Expression6 tissues
Bone Marrow
100%
Liver
0%
Brain
0%
Lung
0%
Ovary
0%
Heart
0%
Gene Interaction Network
Click a node to explore
PDHA2PDHA1PDHXDLSTDBTTALDO1BCKDHB
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt P29803
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
1.41LoF Tolerant
pLIβ“˜
0.52Intermediate
Observed/Expected LoF0.00 [0.00–1.41]
RankingsWhere PDHA2 stands among ~20K protein-coding genes
  • #10,666of 20,598
    Most Researched37
  • #5,197of 5,498
    Most Pathogenic Variants1
  • #14,548of 17,882
    Most Constrained (LOEUF)1.41
Genes detectedPDHA2
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Whole-exome sequencing improves the diagnosis and care of men with non-obstructive azoospermia.
PMID: 35172124
Am J Hum Genet Β· 2022
1.00
2
Human testis-specific PDHA2 gene: methylation status of a CpG island in the open reading frame correlates with transcriptional activity.
PMID: 20005141
Mol Genet Metab Β· 2010
0.90
3
Data supporting the co-expression of PDHA1 gene and of its paralogue PDHA2 in somatic cells of a family.
PMID: 27656664
Data Brief Β· 2016
0.80
4
Demethylation of the coding region triggers the activation of the human testis-specific PDHA2 gene in somatic tissues.
PMID: 22675509
PLoS One Β· 2012
0.70
5
Compensation for X-linked Pdha1 silencing by Pdha2 is essential for meiotic double-strand break repair in spermatogenesis.
PMID: 40679056
Development Β· 2025
0.60