ABSTRACT We report an infant with holoprosencephaly who developed epileptic apnea without tonic posturing at the age of 5 months. Conventional interictal electroencephalography (EEG) yielded no remarkable findings. At the age of 7 months, polygraphic study including video-EEG monitoring revealed ictal rhythmic theta activity in the right frontal area that precedes the onset of the apnea. The apnea was considered to be a symptom caused by epileptogenic discharges and it resolved immediately after the administration of diazepam. In infants with holoprosencephaly, differentiation of epileptic apnea from central apnea is crucial, because medications used to treat central apnea, caffeine and aminophylline, can be harmful to children with epileptic apnea. Consequently, polygraphic study including video-EEG monitoring is indispensable to differential diagnosis.
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