Temporal lobe epilepsy associated with 'triple pathology' of hippocampal sclerosis, focal cortical dysplasia and cavernoma in the ipsilateral frontal lobe

Abstract

The coexistence of hippocampal sclerosis (HS) and extrahippocampal lesions such as focal cortical dysplasia (FCD) and cavernoma (CA) in temporal lobe epilepsies (TLE) is termed 'dual pathology'. We report a case of TLE having 'triple pathology' of HS, FCD and CA in the ipsilateral frontal lobe. Using chronic subdural electrode recording, an interictal electrocorticogram (ECoG) demonstrated that the medial temporal lobe lesion and the FCD lesion exhibited independent paroxysmal discharges, while an ictal ECoG demonstrated that the medial temporal lobe was the ictal onset zone. We postulated that the FCD lesion Case ReportKey words: temporal lobe epilepsy, triple pathology, hippocampal sclerosis, focal cortical dysplasia, cavernoma 35 caused repeated seizures, which in turn, caused secondary hippocampal damage and HS, and eventually medically intractable epilepsy. The coexistence of CA was considered incidental. As treatments, the epileptogenic HS was resected through an anterior temporal lobectomy, and the FCD lesion was biopsied, while no surgical intervention was performed for the CA lesion. The patient achieved good seizure control during 12 months after surgery. Chronic subdural electrode recording is important for the assessment of multiple epileptogenic lesions before epilepsy surgery.

Medial temporal lobe epilepsy associated with cortical dysplasia extending from the medial temporal lobe to the fornix: a case report

“…In other words, "dual pathology" was not noted. Dual pathology does not merely indicate the morphological coexistence of hippocampal sclerosis with an extrahippocampal lesion, but also indicates the pathophysiologi-cal mechanisms such as the phenomena of kindling and secondary hippocampal epileptogenesis [14]. In the present case, we postulate that CD initially caused the repeated seizures and that the hippocampus gained secondary and independent epileptogenesis without morphological changes.…”

“…The seizure semiology and findings of the interictal EEG and functional imaging such as FDG-PET and IMZ-SPECT [6,7] were consistent with those of TLE, although the MR lesions in the temporal lobe extended outside of the temporal lobe. In this situation, invasive video-intracranial EEG monitoring with subdural grids or depth electrodes may be indicated to aid localization of the epileptogenic zone [1][2][3]14]. However, placement of the intracranial electrodes in the deep-seated continuous lesion such as fornix was too invasive, and surgery for the TLE under the guidance of intraoperative ECoG [8] was selected.…”

“…In the present case, we postulate that CD initially caused the repeated seizures and that the hippocampus gained secondary and independent epileptogenesis without morphological changes. For best clinical practice, the necessity of an additional hippocampectomy should be determined by integrated assessment of the clinical findings, MR and functional imaging, as well as interictal and ictal EEG findings, together with invasive or intraoperative ECoG findings [8,14].…”

Triple pathology in patients with temporal lobe epilepsy: A case report and review of the literature

“…The occurrence of ‘triple pathology’ in epilepsy patients was initially proposed by Maciunas et al (9) and Samura et al (10). Samura et al reported a case of TLE with the coexistence of HS, FCD and cavernoma (CA) (10).…”

Coexistent ganglioglioma, focal cortical dysplasia, and hippocampal sclerosis (triple pathology) in chronic epilepsy

“…These most commonly involve combinations of tumor and focal cortical dysplasia or hippocampal sclerosis and focal cortical dysplasia [7][8][9][10][11]. Only rare instances of triple pathology, where 3 potentially etiologic lesions coexist in the same patient, have been reported [12][13][14][15].…”