Address correspondence to Lino Nobili, “C. Munari” Centre of Epilepsy Surgery, Centre of Sleep Medicine, Niguarda Hospital, Piazza Ospedale Maggiore 3, 20162, Milan, Italy. E-mail: [email protected]

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Summary

Purpose: Sleep-related complex motor seizures are a common feature of nocturnal frontal lobe epilepsy. Nevertheless, recent studies also suggest that sleep-related hypermotor seizures can originate in the insula. The present study describes the electroclinical features of eight drug-resistant epileptic patients with insular-opercular seizures manifesting with nocturnal complex motor seizures.

Methods: Patients underwent a comprehensive presurgical evaluation, which included history, interictal electroencephalography (EEG), scalp video-EEG monitoring, high-resolution magnetic resonance imaging (MRI), and intracerebral recording by stereo-EEG.

Key Findings: Almost all patients reported an initial sensation consisting of viscerosensitive or somatosensory symptoms. Ictal clinical signs were represented by tonic–dystonic asymmetric posturing and/or hyperkinetic automatisms, including bimanual/bipedal activity and ballistic movements. Some patients exhibited dysarthric speech, hypersalivation, and apnea. Interictal and ictal EEG provided lateralizing information in the majority of patients. In three patients, MRI showed a focal anatomical abnormality in the insular-opercular region. Stereo-EEG ictal recordings demonstrated that the epileptic discharge involved simultaneously the insular cortex and the opercular region. Complex motor manifestations appeared when the ictal discharge showed an extrainsular spreading to frontomesial regions (cingulum, superior frontal gyrus, and supplementary motor area) and/or to internal and neocortical temporal lobe structures. Six patients received an insular-opercular cortical resection; three of them are seizure free (minimum follow-up 24 months) and in one a marked reduction in seizure frequency was obtained. Two patients have been operated on recently. Histology revealed a focal cortical dysplasia in three patients. One patient excluded from surgery died for sudden unexpected death in epilepsy during sleep.

Significance: Our data strengthen the concept that sleep-related complex motor attacks can originate in the insula, and provide useful electroclinical information to differentiate this localization from those with similar clinical characteristics. Furthermore, this study indicates that in these drug-resistant patients, surgical treatment represents a highly effective treatment option.

Supporting Information

Video S1. Video recording of complex motor behaviors: hyperkinetic automatisms (first patient, case 1); asymmetric dystonic posturing (second patient, case 6).

Video S2. All models were reconstructed from real anatomic data. Multimodal dataset were co-registered with FMRIB’s Linear Images Registration Tool (FLIRT), the module for linear registrations included in FMRIB Software Library (FSL). Pial surface and thalamus were automatically segmented with Freesurfer, whereas insular gyri were segmented manually. Fiber tracts were modeled with the Gray Scale Model Maker module included in 3-D Slicer. Electrodes were built in Maxon Cinema 4D, and aligned manually in the correct position, having the post-implant Oarm dataset as a guide. The artistic 3-D rendering was done with Maxon Cinema 4D and Adobe Photoshop.

Table S1. Interictal and ictal EEG at scalp video-EEG recordings.

Table S2. Main features of SEEG investigations.

Filename	Description
EPI_3254_sm_Supplementaryfile1.avi4.7 MB	Supporting info item
EPI_3254_sm_supplementaryfile2.avi3.6 MB	Supporting info item
EPI_3254_sm_TableS1.doc31.5 KB	Supporting info item
EPI_3254_sm_TableS2.doc35.5 KB	Supporting info item

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Volume52, Issue10

October 2011

Pages 1781-1791

Insular-opercular seizures manifesting with sleep-related paroxysmal motor behaviors: A stereo-EEG study

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Insular-opercular seizures manifesting with sleep-related paroxysmal motor behaviors: A stereo-EEG study

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