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MS
PDHA1 XLD unreviewed

Pyruvate dehydrogenase complex deficiency

Enzyme or protein defect

PDH E1-alpha

Accumulates or becomes deficient

Lactate, alanine, pyruvate

Key features

Congenital lactic acidosis, neurological impairment; worsened by high-carbohydrate diet

Management

Ketogenic diet; thiamine trial; dichloroacetate

Not clinical guidance. Written from standard clinical biochemistry, not from the uploaded documents, and marked unreviewed. Teaching-level reference for study and database seeding only. It must not be used to manage a patient.

Recent literature

Europe PMC · from cache · sorted by publication date

  1. 1
    Next generation AAV-F capsid gene therapy rescues disease pathology in a model of pyruvate dehydrogenase complex deficiency.

    Keegan A, Çetin Ö, Chilcott EM, Diaz JA, Eaton S, Waddington SN, Counsell JR, Rahman S, Karda R. · 2026-06-15

    unreviewed
  2. 2
  3. 3
    From Severe Neonatal Encephalopathy to Slowly Neurologic Progressive Disease: Pyruvate Dehydrogenase Deficiency Related to <i>PDHA1</i> Variants.

    Corbaz S, Pibernus DA, Loos MA, Pérez MM, Buompadre MC, García FM, Rodriguez MP, Rugilo C, Armeno M, Seo GH, … · 2025-12-23

    unreviewed
  4. 4
    Neurological, Neurodevelopmental and Treatment Outcomes in Patients With Pyruvate Dehydrogenase Complex Deficiency.

    Savvidou A, Reilly C, Sofou K, Ygberg S, Eklund EA, Naess K, Darin N. · 2026-08-06

    unreviewed
  5. 5
    Ketogenic diet therapy in pyruvate dehydrogenase deficiency: Global clinical practice from literature and survey data.

    Madsen S, Allender B, Blackford R, Bollard T, Comerford A, Johnson ML, Sinha R, Vanatta L, Whiteley VJ, Schoe… · 2026-06-18

    unreviewed
  6. 6
  7. 7
    The genotypic and phenotypic landscape of PDHA1-related pyruvate dehydrogenase complex deficiency.

    Merkevicius K, Smirnov D, Schlieben LD, Ganetzky R, Feichtinger RG, Jiang H, Fang F, Ebihara T, Murayama K, F… · 2026-07-01

    cited 3× unreviewed
  8. 8
    Neonatal Epilepsy: Beyond Seizures in a Developing Brain—A Narrative Review

    Boscarino G, Cresta E, Leonardi L, Di Chiara M, Spalice A, Terrin G. · 2026-06-01

    open access unreviewed
  9. 9
    From Pharmacodynamic Biomarker to Evaluating Treatment Response: Biomarkers in Primary Mitochondrial Diseases.

    Stern S, Crisamore K, Patton J, Schuck R, Pacanowski M. · 2026-06-01

    open access unreviewed
  10. 10

External claims. These come from an index outside this database and are not checked against it. Treat them as leads.