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Global brain dysfunction in an infant with pyridoxine dependency: evaluation with EEG, evoked potentials, MRI, and
J J Shih1, H Kornblum, D A Shewmon
1Neurodiagnostic Laboratory, University of New Mexico Health Sciences Center, Albuquerque 87131-5281, USA.
Insights
Pyridoxine dependency causes intractable seizures in infants. This study reveals diffuse brain abnormalities in an infant, suggesting a generalized disease process from this vitamin B6 deficiency.
Area of Science:
- Neurology
- Biochemistry
- Pediatrics
Background:
- Pyridoxine dependency is a rare genetic disorder.
- It typically manifests as intractable seizures in newborns and infants.
- Early diagnosis and treatment are crucial to prevent neurological damage.
Purpose of the Study:
- To describe the clinical presentation and diagnostic findings in an infant with pyridoxine-dependent seizures.
- To report novel positron emission tomography (PET) and evoked potential results in this condition.
- To correlate neuroimaging (EEG and MRI) findings with the underlying biochemical deficiency.
Main Methods:
- Case report of an infant with confirmed pyridoxine-dependent seizures.
- Utilized electroencephalography (EEG) for seizure activity monitoring.
- Performed magnetic resonance imaging (MRI) for structural brain assessment.
- Conducted positron emission tomography (PET) for functional brain analysis.
- Recorded evoked potentials to assess neural pathway integrity.
Main Results:
- The infant presented with intractable seizures characteristic of pyridoxine dependency.
- EEG demonstrated generalized seizure activity.
- MRI revealed diffuse structural abnormalities in the brain.
- PET scans indicated widespread functional deficits.
- Evoked potential studies showed abnormalities consistent with a generalized neurological process.
Conclusions:
- Pyridoxine dependency can lead to diffuse structural and functional brain abnormalities.
- The findings suggest that this coenzyme deficiency results in a generalized disease process affecting the brain.
- Comprehensive neurodiagnostic evaluation, including PET and evoked potentials, can provide valuable insights into the pathophysiology of pyridoxine dependency.
Abstract:
Pyridoxine dependency often presents with intractable seizures in neonates and infants. We describe an infant with pyridoxine-dependent seizures and report the first PET and evoked potential results and EEG and MRI findings. All studies show either diffuse structural or functional abnormality. Our data suggest this coenzyme deficiency causes a generalized disease process.