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Cardiology26 papers

Lipoic acid synthetase deficiency

Last edited: 4/22/2026

Overview

Acid maltase deficiency (AMD), also known as Pompe disease, encompasses a spectrum of presentations including late-onset forms characterized by glycogen accumulation in tissues such as liver, skeletal muscles, and notably, cerebral arteries 12. Late-onset variants can involve severe vascular pathology leading to cerebral aneurysms and neurological complications 12.

Diagnosis

  • Muscle biopsy: Demonstrates acid phosphatase-positive vacuoles filled with PAS-positive material 12.
  • Enzyme assay: Reduced acid maltase activity confirms the diagnosis 12.
  • Neuroimaging: CT and MRI may reveal hypodense and hyperintense white matter areas, indicative of vascular and demyelination changes 1.
  • Postmortem examination: Reveals vascular pathology including vacuolar degeneration and aneurysms in affected individuals 2.
  • Management

  • Supportive care: Focus on managing symptoms and complications, including respiratory support and neurological care 12.
  • Monitoring: Regular assessment for aneurysm formation and neurological deterioration 12.
  • No specific pharmacological treatment mentioned: Current evidence does not specify targeted drug therapies or dosing 12.
  • Special Populations

  • Neurological complications: Increased risk in late-onset forms, particularly concerning cerebral aneurysms and hemorrhages 12.
  • Comorbidities: Vascular fragility complicates management, especially in cases with subarachnoid hemorrhage 2.
  • Key Recommendations

  • Perform muscle biopsy and enzyme assays for definitive diagnosis (Evidence: Moderate 12).
  • Regular neuroimaging to monitor cerebral vascular changes (Evidence: Moderate 1).
  • Aggressive management of neurological symptoms and vascular complications (Evidence: Expert opinion 2).
  • References

    1 Kretzschmar HA, Wagner H, Hübner G, Danek A, Witt TN, Mehraein P. Aneurysms and vacuolar degeneration of cerebral arteries in late-onset acid maltase deficiency. Journal of the neurological sciences 1990. link90258-o) 2 Miyamoto Y, Etoh Y, Joh R, Noda K, Ohya I, Morimatsu M. Adult-onset acid maltase deficiency in siblings. Acta pathologica japonica 1985. link

    Original source

    1. [1]
      Aneurysms and vacuolar degeneration of cerebral arteries in late-onset acid maltase deficiency.Kretzschmar HA, Wagner H, Hübner G, Danek A, Witt TN, Mehraein P Journal of the neurological sciences (1990)
    2. [2]
      Adult-onset acid maltase deficiency in siblings.Miyamoto Y, Etoh Y, Joh R, Noda K, Ohya I, Morimatsu M Acta pathologica japonica (1985)

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