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Chugani et al. [1] reported seizure-free outcome after cortical resection in patients with intractable infantile spasms due to focal cortical dysplasia identified by MRI or PET. We report an infant who had successful epilepsy surgery for intractable infantile spasms in the setting of bilateral periventricular leukomalacia and remote germinal matrix hemorrhage.
Case report.
The patient was conceived after maternal treatment with clomiphene citrate for infertility. During the first trimester, no maternal trauma, illness, x-ray exposure, or drug ingestion occurred, but pregnancy was complicated by cervical incompetence. Early labor was successfully treated at 25.5 weeks gestation with terbutaline, magnesium sulfate, and cervical cerclage. The vaginal birth was uneventful at 38 weeks with high Apgar scores and no perinatal complications. Birth weight was 3.4 kg. The infant was discharged to home on day 2 and did well until 2 months old, when seizures began.
From 2 to 7 months old, partial seizures occurred in clusters several times a day. Seizures variably involved right eyelid twitching, right or bilateral limb stiffening and trembling, labored respiration, facial grimacing, or eye rolling. Many EEG seizures were recorded over the right temporoparietal-occipital region at 2.5 and 4 months. A single EEG seizure over the left temporoparietal-occipital region was recorded at 5 months old. Failed medications included phenobarbital, carbamazepine, phenytoin, valproate, and pyridoxine.
Infantile spasms replaced the partial seizures at 7 months old and occurred in clusters several times a day despite treatment with adrenocorticotrophic hormone, phenobarbital, phenytoin, clonazepam, and felbamate. EEG at 7 and 13 months showed hypsarrhythmia with generalized electrodecrement during spasms Figure 1. Interictal sharp waves were multifocal but tended to be more frequent over the right temporoparietal-occipital region. Sleep spindles were decreased over the right hemisphere.
Figure 1. EEG at the start of an infantile spasm (arrow) showing a diffuse electrodecremental pattern replacing the …
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