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- W2034852667 abstract "We read with great interest the paper entitled “Tailored surgery for drug-resistant epilepsy due to temporal pole encephalocele and microdysgenesis” by Giulioni et al.1Giulioni M. Licchetta L. Bisulli F. Rubboli G. Mostacci B. Marucci G. et al.Tailored surgery for drug-resistant epilepsy due to temporal pole encephalocele and microdysgenesis.Seizure. 2014; 23: 164-166Abstract Full Text Full Text PDF PubMed Scopus (25) Google Scholar The authors describe two cases of encephalocele associated with cortical microdysgenesis and assert the necessity to consider an extensive surgical resection in subjects with small encephaloceles, due to the possible concomitance of malformations of cortical development. We recently observed two patients that may represent two opposite situations of patients. The first subject is a woman who experienced a tonic–clonic seizure during sleep at age 14 years, and was started with carbamazepine (CBZ) up to 1200 mg/day. No seizures appeared for 8 years. At age 22, she started to experience brief, weekly complex partial seizures, described as loss of consciousness, gestural and bipedal automatisms, followed by confusional status. Seizures persisted despite treatment with numerous antiepileptic drugs in various combinations. At age 35, when she came to our observation, physical and neurologic examinations were normal. Video-EEG monitoring allowed recording a nocturnal seizure originating from wide area of the right frontal lobe. MRI showed a small encephalocele associated with a linear cortical hyperintensity in T2-weighted sequences, localized in right anterior frontal region (Fig. 1A) . Neuropsychological examination showed impairment in visuospatial memory, constructive apraxia and difficulties in planning actions, consistent with a right fronto-temporal impairment. At age 36, she underwent lesionectomy and resection of adjacent cortex (Fig. 1B). Histologic examination showed encephalocele and an associated type IB focal cortical dysplasia (FCD), extending beyond the resection margin. Surgery was uncomplicated, without any additional post-operative neurologic or neuropsychological deficits, but seizure frequency was not modified (Engel class IV) at 5 years of follow-up. Patient refused further surgical interventions. The second patient is a 20-year-old man who came to our observation at age 14 for rare seizures characterized by déjà-vu, dreaming state and sometimes oral automatisms. Treatment with carbamazepine resulted in seizure freedom for 14 months, then he started experiencing weekly complex partial seizures with cephalic aura, loss of consciousness, oral and gestural automatisms, dystonia and clonic movements of right upper limb, followed by confusion state. Adjunctive treatment with topiramate and, subsequently, lacosamide did not modify seizure frequency. Standard brain MRI was deemed normal. At age 20 he was evaluated for epilepsy surgery. Prolonged video-EEG monitoring allowed detecting three seizures originating from left temporal lobe. High-resolution CT scan (Fig. 1B) showed a small bony defect in left mesial portion of middle cranial fossa. MRI targeted at temporal pole (Fig. 1C) evidenced a tiny encephalocele in the same area, while no other parenchymal signal abnormalities were evident. FDG-PET showed mild hypometabolism in left temporal lobe. Physical and neurological examinations were normal, whereas neuropsychological assessment showed only a mild deficit in long-term verbal memory. Lesionectomy and bone repair were performed during an uncomplicated surgical procedure (see Fig. 1E). Histology showed normal cortical organization and mild chronic inflammation and gliosis. At twenty-month follow-up, the patient was seizure-free (Engel class IA) and neuropsychological testing was normal. This latter case is in line with the three described by Abou-Hamden et al.2Abou-Hamden A. Lau M. Fabinyi G. Berkovic S.F. Jackson G.D. Mitchell L.A. et al.Small temporal pole encephaloceles: a treatable cause of “lesion negative” temporal lobe epilepsy.Epilepsia. 2010; 51: 2199-2202Crossref PubMed Scopus (54) Google Scholar; indeed our subject was initially labeled as having “lesion negative” temporal lobe epilepsy, while further evaluation directed at temporal lobe permitted to reveal a small encephalocele. In our patient #2, clinical, EEG and imaging data, including CT scan, MRI and interictal FDG-PET, indicated a left temporal epileptogenic focus, consistent with the site of encephalocele. No additional findings indicated an involvement of other structures; lesionectomy was sufficient to reach seizure freedom. Conversely, our patient #1, a Type I FCD was found surrounding the encephalocele; resection margins were not free from lesion, and the FCD boundaries were not visible at high resolution MRI as it may be the case in a high proportion of Type I FCD3Tassi L. Garbelli R. Colombo N. Bramerio M. Lo Russo G. Deleo F. et al.Type I focal cortical dysplasia: surgical outcome is related to histopathology.Epileptic Disord. 2010; 12: 181-191PubMed Google Scholar; neuropathology may thus explaining why seizure freedom was not achieved in this patient, despite a surgical resection exceeding the limits of the encephalocele. In this patient, two clues addressed toward the existence of an associated pathology: a discrete, but visible signal abnormality adjacent to encephalocele, and the presence of multiple cognitive deficits. Of the nearly 40 cases published so far, most of which were reviewed by Faulkner et al.,4Faulkner H.J. Sandeman D.R. Love S. Likeman M.J. Nunez D.A. Lhatoo S.D. Epilepsy surgery for refractory epilepsy due to encephalocele: a case report and review of the literature.Epileptic Disord. 2010; 12: 160-166PubMed Google Scholar about 20 comprised a description of the surgical procedure. Of these, half underwent lesionectomy and the remaining lobectomy. Lesionectomy was realized in all cases of non-temporal encephalocele, while lobectomy was preferred in two thirds of subjects with temporal encephalocele. Both lesionectomy and lobectomy resulted in excellent outcome, without any difference between the two surgical approaches. This supports the vision that encephalocele itself may act as epileptogenic focus, probably by consequence of an irritative effect of mechanical traction, with secondary inflammation and gliosis.4Faulkner H.J. Sandeman D.R. Love S. Likeman M.J. Nunez D.A. Lhatoo S.D. Epilepsy surgery for refractory epilepsy due to encephalocele: a case report and review of the literature.Epileptic Disord. 2010; 12: 160-166PubMed Google Scholar The two patients described by Giulioni et al.,1Giulioni M. Licchetta L. Bisulli F. Rubboli G. Mostacci B. Marucci G. et al.Tailored surgery for drug-resistant epilepsy due to temporal pole encephalocele and microdysgenesis.Seizure. 2014; 23: 164-166Abstract Full Text Full Text PDF PubMed Scopus (25) Google Scholar as well as our patient #1, point out how the presence of additional signal abnormalities at MRI should prompt to consider wider surgical resections and, in selected extratemporal cases, an invasive pre-surgical assessment to better “tailor” the surgical procedure. In our patient#1, pre-operative video-EEG results and neuropsychological assessment suggested the existence of a wider lesion, but available non-invasive data were not sufficient for planning a radical resection of the encephalocele-associated FCD. Unfortunately, Giulioni et al.1Giulioni M. Licchetta L. Bisulli F. Rubboli G. Mostacci B. Marucci G. et al.Tailored surgery for drug-resistant epilepsy due to temporal pole encephalocele and microdysgenesis.Seizure. 2014; 23: 164-166Abstract Full Text Full Text PDF PubMed Scopus (25) Google Scholar reported partial data concerning their patient #1 only. In conclusion, when clinical, neuropsychological, electrophysiological and imaging data point toward a well-localized epileptogenic focus corresponding with encephalocele (as exemplified by our patient #2), lesionectomy may be sufficient to control seizures. On the contrary, when all data suggest a wider epileptogenic zone (patient #1), lesionectomy limited to encephalocele may not be adequate to get seizure freedom. The authors declare that there are no conflicts of interest." @default.
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- W2034852667 title "Refractory epilepsy and encephalocele: Lesionectomy or tailored surgery?" @default.
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