Surgical management of cortical dysplasia in infancy and early childhood

Otsuki, Taisuke; Honda, Ryoko; Takahashi, Akio; Kaido, Toshimi; Kaneko, Yu; Nakai, Tetsuji; Saito, Yuko; Itoh, Masayuki; Nakagawa, Eiji; Sugai, Kenji; Sasaki, Masayuki

doi:10.1016/j.braindev.2013.04.008

Cited by 25 publications

(27 citation statements)

References 47 publications

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“…In a recent literature review, Hauptman and Mathern reported an overall seizure-free rate of 62 % in patients with FCD, the best predictor being complete removal of the dysplastic tissue [11]. Otsuki et al reported on a pediatric population comparable to our own, with children younger than 6 years at the time of surgery for FCD [14]. Only 12 % had been invasively explored with chronic subdural recordings.…”

Section: Discussionmentioning

confidence: 81%

Outcome of surgery in children with focal cortical dysplasia younger than 5 years explored by stereo-electroencephalography

et al. 2014

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Section: Discussionmentioning

confidence: 81%

Outcome of surgery in children with focal cortical dysplasia younger than 5 years explored by stereo-electroencephalography

et al. 2014

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“…In a retrospective, crosssectional study by Otsuki et al on 56 children with different forms of MCD, 66% were seizure-free at last follow-up. The follow-up period ranged from 1 to 11 years and 55% had been seizure-free since surgery [20].…”

Section: Discussionmentioning

confidence: 99%

Long-term follow-up after epilepsy surgery in infancy and early childhood – A prospective population based observational study

Reinholdson

Olssøn

Edelvik

et al. 2015

Seizure

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This is the first prospective, population based, longitudinal study to show that a favourable seizure outcome is achievable in a majority of infants and young children undergoing resective epilepsy surgery and that the improvements are consistent over time. Many can also stop taking AEDs. The findings emphasise the importance of early referral to epilepsy surgery evaluation in cases of medically intractable epilepsy in infants and young children.

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“…1 Among them, hemimegalencephaly (HME) and focal cortical dysplasia (FCD) are relatively frequent in childhood epilepsy and often can be treated by surgery. 2 The histopathologic process in HME and FCD brains is characterized by the features of cortical dyslamination, dysmorphic immature neurons, and neuronal heterotopia, indicating primary defects in neuroglial differentiation, migration, and cellular growth. Interestingly, this pathologic disorder shares similarities with tuberous sclerosis complex (TSC), having multiple cortical tubers and showing infantile spasm and various mental retardations.…”

mentioning

confidence: 99%

Pathologic Active mTOR Mutation in Brain Malformation with Intractable Epilepsy Leads to Cell-Autonomous Migration Delay

Hanai

Sukigara

Dai

et al. 2017

The American Journal of Pathology

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The activation of phosphatidylinositol 3-kinase-AKTs-mammalian target of rapamycin cell signaling pathway leads to cell overgrowth and abnormal migration and results in various types of cortical malformations, such as hemimegalencephaly (HME), focal cortical dysplasia, and tuberous sclerosis complex. However, the pathomechanism underlying abnormal cell migration remains unknown. With the use of fetal mouse brain, we performed causative gene analysis of the resected brain tissues from a patient with HME and investigated the pathogenesis. We obtained a novel somatic mutation of the MTOR gene, having approximately 11% and 7% mutation frequency in the resected brain tissues. Moreover, we revealed that the MTOR mutation resulted in hyperphosphorylation of its downstream molecules, S6 and 4E-binding protein 1, and delayed cell migration on the radial glial fiber and did not affect other cells. We suspect cell-autonomous migration arrest on the radial glial foot by the active MTOR mutation and offer potential explanations for why this may lead to cortical malformations such as HME.

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Surgical management of cortical dysplasia in infancy and early childhood

Cited by 25 publications

References 47 publications

Outcome of surgery in children with focal cortical dysplasia younger than 5 years explored by stereo-electroencephalography

Outcome of surgery in children with focal cortical dysplasia younger than 5 years explored by stereo-electroencephalography

Long-term follow-up after epilepsy surgery in infancy and early childhood – A prospective population based observational study

Pathologic Active mTOR Mutation in Brain Malformation with Intractable Epilepsy Leads to Cell-Autonomous Migration Delay

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