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Delayed-onset focal dystonia after diffuse cerebral hypoxia--two case reports
1Department of Neurology, Youngdong Severance Hospital, Yonsei University College of Medicine, Seoul, Korea.
Insights
Delayed-onset focal dystonia, a rare complication of perinatal anoxia, can occur years after the initial hypoxic insult. This condition may result from non-focal brain damage, challenging previous understandings of its cause.
Area of Science:
- Neurology
- Pediatric Neurology
- Neuroscience
Background:
- Delayed-onset focal dystonia is a rare neurological disorder.
- It is often associated with cerebrovascular disease or hypoxic brain damage.
- The exact lesion sites and pathogenesis remain uncertain.
Observation:
- This study describes two children who developed delayed-onset focal dystonia following perinatal anoxia.
- The onset of dystonia occurred 3 and 6 years after the hypoxic event.
- Neither patient had a focal brain lesion; one exhibited scattered white matter lesions, and the other had diffuse frontoparietal atrophy.
Findings:
- Delayed-onset focal dystonia can arise from non-focal brain lesions.
- Diffuse frontoparietal atrophy and cerebral white matter lesions are potential causes.
- A significant delay between hypoxic insult and dystonia onset is possible.
Implications:
- This expands the understanding of dystonia pathogenesis, particularly after perinatal anoxia.
- It highlights the possibility of late-onset neurological complications from non-focal brain injury.
- Clinical evaluation for dystonia should consider perinatal anoxia even with non-focal brain findings.
Abstract:
The delayed-onset focal dystonia is a rare sequela of cerebrovascular disease or diffuse cerebral hypoxic damage. The responsible lesion sites for the dystonia are variable and the pathogenesis is uncertain. We describe two children with delayed-onset focal dystonia as a complication of perinatal anoxia. The intervals between hypoxic insult and onset of dystonia were 6 years in one and 3 in the other cases. Our patients did not have a focal lesion; one had scattered white matter lesion and the other had a diffuse frontoparietal atrophy. Delayed-onset dystonia after perinatal anoxia can be also caused by non-focal lesion such as diffuse frontoparietal atrophy or cerebral white matter lesion with long interval delay.