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[A case of infantile epileptic apnea with congenital brain anomaly]
Tatsuyuki Ohto1, Nobuaki Iwasaki, Miho Takahashi
1Department of Pediatrics, Institute of Clinical Medicine, University of Tsukuba, Tsukuba, Ibaraki. tohto@md.tsukuba.ac.jp
Insights
This study reports a rare case of epileptic apnea in an infant with brain anomalies. The condition, characterized by specific EEG patterns and apnea, was successfully treated with valproate sodium.
Area of Science:
- Neurology
- Pediatrics
- Epileptology
Background:
- Apnea attacks in infants can be challenging to manage, especially when resistant to standard treatments like aminophylline.
- Brain anomalies, such as cortical and cerebellar hypogenesis and agenesis of the corpus callosum, can present with complex neurological symptoms.
Observation:
- A two-month-old infant with psychomotor retardation experienced aminophylline-resistant apnea attacks.
- Video-electroencephalogram (EEG) monitoring revealed left hemispheric rhythmic delta wave bursts preceding apnea episodes.
- During apnea, the EEG showed suppressed basic activity, indicating a link between epileptic activity and respiratory cessation.
Findings:
- The infant was diagnosed with epileptic apnea, a rare condition associated with significant brain malformations.
- Treatment with valproate sodium effectively controlled the apnea attacks.
- The case highlights the potential role of specific EEG abnormalities in the pathophysiology of epileptic apnea.
Implications:
- This case provides valuable insights into the pathomechanism of epileptic apnea, particularly in infants with congenital brain anomalies.
- It underscores the importance of comprehensive neurophysiological investigation, including video-EEG, for diagnosing rare neurological conditions in infants.
- The successful management with valproate sodium suggests its potential efficacy in similar cases, guiding future clinical practice.
Abstract:
Two months-old girl with psychomotor retardation had aminophylline-resistant apnea attacks and was investigated by video-EEG recording. She had hypogenesis of cerebral cortex and cerebellum and complete agenesis of corpus callosum. Left hemispheric 2 Hz rhythmic delta wave burst originating from the posterior temporal area lasted about 20 seconds, and was followed by an apnea attack persisting for 30 seconds. During the apnea attack, the basic activity of EEG was suppressed. The diagnosis of epileptic apnea was made, and the attacks were controlled with valproate sodium. Reports of cases of brain anomaly presenting with epileptic apnea are rare and this interesting case provided a clue to the pathomechanism of this condition.
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