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OBJECTIVE: To investigate the local cortical morphology and individual-based morphological brain networks (MBNs) changes in children with Rolandic epilepsy (RE). METHODS: Based on the structural MRI data of 56 children with RE and 56 healthy controls (HC), we constructed four types of individual-based MBNs using morphological indices (cortical thickness [CT], fractal dimension [FD], gyrification index [GI], and sulcal depth [SD]). The global and nodal properties of the brain networks were analyzed using graph theory. The between-group difference in local morphology and network topology was estimated, and partial correlation analysis was further analyzed. RESULTS: Compared with the HC, children with RE showed regional GI increases in the right posterior cingulate gyrus and SD increases in the right anterior cingulate gyrus and medial prefrontal cortex. Regarding the network level, RE exhibited increased characteristic path length in CT-based and FD-based networks, while decreased FD-based network node efficiency in the right inferior frontal gyrus. No significant correlation between altered morphological features and clinical variables was found in RE. CONCLUSIONS: These findings indicated that children with RE have disrupted morphological brain network organization beyond local morphology changes. SIGNIFICANCE: The present study could provide more theoretical basis for exploring the neuropathological mechanisms in RE.
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Epilepsia Rolândica , Imageamento por Ressonância Magnética , Rede Nervosa , Humanos , Epilepsia Rolândica/fisiopatologia , Epilepsia Rolândica/diagnóstico por imagem , Criança , Masculino , Feminino , Rede Nervosa/diagnóstico por imagem , Rede Nervosa/fisiopatologia , Rede Nervosa/patologia , Encéfalo/diagnóstico por imagem , Encéfalo/patologia , Encéfalo/fisiopatologia , AdolescenteRESUMO
BACKGROUND: Benign childhood epilepsy with centrotemporal spikes (BECTS), a common pediatric epilepsy, may lead to cognitive decline when compounded by Electrical Status Epilepticus during Sleep (ESES). Emerging evidence suggests that disruptions in the Salience Network (SN) contribute significantly to the cognitive deficits observed in BECTS-ESES. Our study rigorously investigates the dynamic functional connectivity (dFC) within the SN and its correlation with cognitive impairments in BECTS-ESES, employing advanced neuroimaging and neuropsychological assessments. METHODS: In this research, 45 patients diagnosed with BECTS-ESES and 55 age-matched healthy controls (HCs) participated. We utilized resting-state functional magnetic resonance imaging (fMRI) and Independent Component Analysis (ICA) to identify three fundamental SN nodes: the right Anterior Insula (rAI), left Anterior Insula (lAI), and the Anterior Cingulate Cortex (ACC). A two-sample t-test facilitated the comparison of dFC between these pivotal regions and other brain areas. RESULTS: Significantly, the BECTS-ESES group demonstrated increased dFC, particularly between the ACC and the right Middle Occipital Gyrus, and from the rAI to the right Superior Parietal Gyrus and Cerebellum, and from the lAI to the left Postcentral Gyrus. Such dFC augmentations provide neural insights potentially explaining the neuropsychological deficits in BECTS-ESES children. Employing comprehensive neuropsychological evaluations, we mapped these dFC disruptions to specific cognitive impairments encompassing memory, executive functioning, language, and attention. Through multiple regression analysis and path analysis, a preliminary but compelling association was discovered linking dFC disturbances directly to cognitive impairments. These findings underscore the critical role of SN disruptions in BECTS-ESES cognitive dysfunctions. LIMITATION: Our cross-sectional design and analytic methods preclude definitive mediation models and causal inferences, leaving the precise nature of dFC's mediating role and its direct impact by BECTS-ESES partially unresolved. Future longitudinal and confirmatory studies are needed to comprehensively delineate these associations. CONCLUSION: Our study heralds dFC within the SN as a vital biomarker for cognitive impairment in pediatric epilepsy, advocating for targeted cognitive-specific interventions in managing BECTS-ESES. The preliminary nature of our findings invites further studies to substantiate these associations, offering profound implications for the prognosis and therapeutic strategies in BECTS-ESES, thereby underlining the importance of this research in the field of pediatric neurology and epilepsy management.
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Epilepsia Rolândica , Imageamento por Ressonância Magnética , Humanos , Masculino , Feminino , Criança , Adolescente , Epilepsia Rolândica/fisiopatologia , Epilepsia Rolândica/diagnóstico por imagem , Cognição/fisiologia , Encéfalo/diagnóstico por imagem , Encéfalo/fisiopatologia , Testes Neuropsicológicos , Disfunção Cognitiva/fisiopatologia , Disfunção Cognitiva/diagnóstico por imagem , Estado Epiléptico/fisiopatologia , Estado Epiléptico/diagnóstico por imagem , Giro do Cíngulo/diagnóstico por imagem , Giro do Cíngulo/fisiopatologia , Rede Nervosa/diagnóstico por imagem , Rede Nervosa/fisiopatologiaRESUMO
OBJECTIVE: Drowsiness has been implicated in the modulation of centro-temporal spikes (CTS) in Self-limited epilepsy with Centro-Temporal Spikes (SeLECTS). Here, we explore this relationship and whether fluctuations in wakefulness influence the brain networks involved in CTS generation. METHODS: Functional MRI (fMRI) and electroencephalography (EEG) was simultaneously acquired in 25 SeLECTS. A multispectral EEG index quantified drowsiness ('EWI': EEG Wakefulness Index). EEG (Pearson Correlation, Cross Correlation, Trend Estimation, Granger Causality) and fMRI (PPI: psychophysiological interactions) analytic approaches were adopted to explore respectively: (a) the relationship between EWI and changes in CTS frequency and (b) the functional connectivity of the networks involved in CTS generation and wakefulness oscillations. EEG analyses were repeated on a sample of routine EEG from the same patient's cohort. RESULTS: No correlation was found between EWI fluctuations and CTS density during the EEG-fMRI recordings, while they showed an anticorrelated trend when drowsiness was followed by proper sleep in routine EEG traces. According to PPI findings, EWI fluctuations modulate the connectivity between the brain networks engaged by CTS and the left frontal operculum. CONCLUSIONS: While CTS frequency per se seems unrelated to drowsiness, wakefulness oscillations modulate the connectivity between CTS generators and key regions of the language circuitry, a cognitive function often impaired in SeLECTS. SIGNIFICANCE: This work advances our understanding of (a) interaction between CTS occurrence and vigilance fluctuations and (b) possible mechanisms responsible for language disruption in SeLECTS.
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Encéfalo , Eletroencefalografia , Imageamento por Ressonância Magnética , Rede Nervosa , Vigília , Humanos , Vigília/fisiologia , Masculino , Feminino , Eletroencefalografia/métodos , Rede Nervosa/diagnóstico por imagem , Rede Nervosa/fisiologia , Encéfalo/fisiologia , Encéfalo/diagnóstico por imagem , Adolescente , Adulto , Epilepsia Rolândica/fisiopatologia , Fases do Sono/fisiologia , Adulto Jovem , CriançaRESUMO
OBJECTIVE: The study aimed to determine the prevalence of attention deficit hyperactivity disorder (ADHD) in patients with self-limiting epilepsy with centrotemporal spike wave (SeLECTS), as well as the electroclinical features associated with this comorbid condition and the neurocognitive effects using psychometric tests. Additionally, we analysed the electrophysiological findings and neurocognitive status of patients with ADHD to estimate the prevalence of epilepsy and neurocognitive effects in the ADHD population and evaluate their clinical features. METHOD: The study included patients diagnosed with SeLECTS and ADHD who were matched for age and gender. Electrophysiological tests, psychometric tests, demographic and clinical characteristics of SeLECTS patients aged 7-13 years and ADHD patients of similar age were analysed. The study examined electrophysiological and psychometric tests, as well as demographic and clinical characteristics. Both groups underwent testing using the Wechsler Intelligence Scale for Children (WISC-R), Stroop Colour and Word Test (SCWT), and EEG (Electroencephalogram). The SeLECTS group also underwent the Bender Visual-Motor Gestalt Test. RESULTS: No significant relationship was found between the SeLECTS and ADHD groups in terms of age and gender. The rate of epileptiform discharge in EEG findings without a diagnosis of epilepsy was 5.6 % (n = 2) in the ADHD group. The rate of ADHD in the SeLECTS group was 28 % (n = 11). Although all subsections of the WISCR test were higher in the ADHD patient group than in the SeLECTS patient group, only verbal IQ and total IQ showed a significant difference. No significant differences were found between the completion times, error rates, and correction averages of the SCWT sections in both groups. There was no significant correlation found between the performance IQ, verbal IQ, and total intelligence scores in either the isolated SeLECTS patient group or the SeLECTS + ADHD patient group (p > 0.05). However, it is worth noting that verbal IQ was below normal in both groups and slightly lower in the SeLECT + ADHD group. Additionally, the mean SCWT completion time was significantly longer in the SeLECT + ADHD group than in the isolated SeLECTS group. However, no significant difference was found in the Bender Gestalt Visual Motor Perception Test. In the psychometric analyses comparing the isolated SeLECTS, SeLECT + ADHD, and ADHD patient groups, the SCWT completion times were significantly longer in the SeLECT + ADHD group than in the other two groups. The verbal IQ score was significantly higher in the ADHD group than in the other two groups. CONCLUSION: In conclusion, although SeLECTS is commonly considered a benign form of epilepsy, our study found a high rate of comorbidity with ADHD. This condition has a negative impact on verbal intelligence and sustained attention, highlighting the importance of a complete neuropsychological evaluation at the stage of epilepsy diagnosis. It is crucial not to overlook the possibility of an ADHD diagnosis.
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Transtorno do Deficit de Atenção com Hiperatividade , Eletroencefalografia , Epilepsia Rolândica , Testes Neuropsicológicos , Humanos , Transtorno do Deficit de Atenção com Hiperatividade/fisiopatologia , Transtorno do Deficit de Atenção com Hiperatividade/diagnóstico , Transtorno do Deficit de Atenção com Hiperatividade/epidemiologia , Masculino , Feminino , Criança , Adolescente , Estudos Prospectivos , Epilepsia Rolândica/fisiopatologia , Epilepsia Rolândica/complicações , Atenção Terciária à Saúde , Psicometria , Escalas de WechslerRESUMO
OBJECTIVE: Benign childhood epilepsy with centrotemporal spikes (BECTS) affects brain network hierarchy and cognitive function; however, itremainsunclearhowhierarchical changeaffectscognition in patients with BECTS. A major aim of this study was to examine changes in the macro-network function hierarchy in BECTS and its potential contribution to cognitive function. METHODS: Overall, the study included 50 children with BECTS and 69 healthy controls. Connectome gradient analysis was used to determine the brain network hierarchy of each group. By comparing gradient scores at each voxel level and network between groups, we assessed changes in whole-brain voxel-level and network hierarchy. Functional connectivity was used to detect the functional reorganization of epilepsy caused by these abnormal brain regions based on these aberrant gradients. Lastly, we explored the relationships between the change gradient and functional connectivity values and clinical variables and further predicted the cognitive function associated with BECTS gradient changes. RESULTS: In children with BECTS, the gradient was extended at different network and voxel levels. The gradient scores frontoparietal network was increased in the principal gradient of patients with BECTS. The left precentral gyrus (PCG) and right angular gyrus gradient scores were significantly increased in the principal gradient of children with BECTS. Moreover, in regions of the brain with abnormal principal gradients, functional connectivity was disrupted. The left PCG gradient score of children with BECTS was correlated with the verbal intelligence quotient (VIQ), and the disruption of functional connectivity in brain regions with abnormal principal gradients was closely related to cognitive function. VIQ was significantly predicted by the principal gradient map of patients. SIGNIFICANCE: The results indicate connectome gradient disruption in children with BECTS and its relationship to cognitive function, thereby increasing our understanding of the functional connectome hierarchy and providing potential biomarkers for cognitive function of children with BECTS.
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Cognição , Conectoma , Epilepsia Rolândica , Imageamento por Ressonância Magnética , Humanos , Criança , Epilepsia Rolândica/fisiopatologia , Epilepsia Rolândica/diagnóstico por imagem , Masculino , Conectoma/métodos , Feminino , Cognição/fisiologia , Rede Nervosa/diagnóstico por imagem , Rede Nervosa/fisiopatologia , Encéfalo/fisiopatologia , Encéfalo/diagnóstico por imagem , AdolescenteRESUMO
PURPOSE: To investigate glymphatic function by Virchow-Robin space (VRS) counts and volume in patients with newly diagnosed self-limited epilepsy with centrotemporal spikes (SeLECTS) and evaluate its relationship with structural connectivity and cognitive impairment. METHODS: Thirty-two children with SeLECTS and thirty-two age- and sex-matched typically developing (TD) children were enrolled in this study. VRS counts and volume were quantified. Structural networks were constructed and the topological metrics were analyzed. Wechsler Intelligence Scale (WISC) was used to assess cognitive function in all participants. Correlation analysis assessed the association between VRS counts and volume, network connectivity, and cognitive impairment. Mediation effects of topological metrics of the structural networks on the relationship between glymphatic function and cognitive impairment were explored. RESULTS: Patients with SeLECTS showed a higher VRS counts, VRS volume, and global shortest path length (Lp); they also showed a lower global efficiency (Eg). VRS counts and volume were significantly correlated with full-scale intelligence quotient (FIQ) (r_VRS counts = -0.520, r_VRS volume = -0.639), performance intelligence quotient (PIQ) (r_VRS counts = -0.693, r_VRS volume = -0.597), verbal intelligence quotient (VIQ) (r_VRS counts = -0.713, r_VRS volume = -0.699), Eg (r_VRS counts = -0.499, r_VRS volume = -0.490), and Lp (r_VRS volume = 0.671) in patients with SeLECTS. Eg mediated 24.59% of the effects for the relationship between VRS volume and FIQ. CONCLUSION: Glymphatic function may be impaired in SeLECTS reflected by VRS counts and volume. Glymphatic dysfunction may result in cognitive impairment by disrupting structural connectivity in SeLECTS.
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Epilepsia Rolândica , Sistema Glinfático , Humanos , Masculino , Feminino , Criança , Epilepsia Rolândica/fisiopatologia , Sistema Glinfático/fisiopatologia , Imageamento por Ressonância Magnética , Disfunção Cognitiva/fisiopatologia , Disfunção Cognitiva/etiologia , Encéfalo/fisiopatologia , Encéfalo/diagnóstico por imagem , Cognição/fisiologia , Escalas de Wechsler , AdolescenteRESUMO
BACKGROUND: The decision to treat children with benign epilepsy with centrotemporal spikes (BECTS) using antiseizure medications (ASM) is controversial. Our goal is to compare the effect of ASM treatment on the alteration of electroencephalographic (EEG) functional connectivity and power across four frequency bands in children with BECTS. METHODS: Children with BECTS with two-year follow-up were retrospectively divided into ASM versus non-ASM groups. The network properties of the EEGs as based on network-based statistic and graph theory were evaluated by the following indices: global efficiency, clustering coefficient, betweenness centrality, and nodal strength in four frequency bands (delta, theta, alpha, and beta). EEG power including absolute power (AP) and relative power (RP) was analyzed in four frequency bands. RESULTS: In children with BECTS with ASM treatment, there was no significant change in EEG connectivity across all bands before and after two years of ASM. In children with BECTS without ASM treatment, there was a significant increase of global efficiency, clustering coefficient, and nodal strength but not the betweenness centrality in the delta band after two years of follow-up. A decrease in AP in the delta and theta bands and a decrease in RP in the theta band were found in the ASM group after two years of treatment. CONCLUSIONS: Our results suggest that ASM may play a role in modulating the development of increasing overall brain connectivity and in downregulating overt synaptic activity, but not intrinsic focal connectivity, in the early years of BECTS. The changes in the EEG power indicate that ASM significantly normalized slow-wave band power.
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Anticonvulsivantes , Eletroencefalografia , Epilepsia Rolândica , Humanos , Epilepsia Rolândica/tratamento farmacológico , Epilepsia Rolândica/fisiopatologia , Feminino , Criança , Masculino , Anticonvulsivantes/farmacologia , Estudos Retrospectivos , Pré-Escolar , Seguimentos , Ondas Encefálicas/efeitos dos fármacos , Ondas Encefálicas/fisiologia , Rede Nervosa/efeitos dos fármacos , Rede Nervosa/fisiopatologia , Encéfalo/fisiopatologia , Encéfalo/efeitos dos fármacosRESUMO
PURPOSE: In brain development, Myelination is the characteristic feature of white matter maturation, which plays an important role in efficient information transmitting. The white matter abnormality has been reported to be associated with self-limited epilepsy with centrotemporal spikes (SeLECTS). This study aimed to detect the altered white matter region in the SeLECTS patients by the combination of diffusion tensor imaging (DTI) and quantitative susceptibility mapping (QSM) technique. METHODS: 27 children with SeLECTS and 23 age- and gender-matched healthy children were enrolled. All participants were scanned with 3.0-T MRI to acquire the structure, diffusion and susceptibility-weighted data. The susceptibility and diffusion weighted data were processed to obtain quantitative susceptibility map and fraction anisotropy (FA) map. Then voxel-wise tract-based spatial statistics (TBSS) were used to analyze quantitative susceptibility and FA data. RESULTS: Both DTI and QSM revealed extensive white matter alterations in the frontal, parietal, and temporal lobes in SeLECTS patients. The overlapped region of DTI and QSM analyses was located in the fiber tracts of the corona radiata. The FA values in this overlapped region were negatively correlated with the magnetic susceptibility values. CONCLUSION: Our results suggest that TBSS-based QSM can be employed as a novel approach for characterizing alterations in white matter in SeLECTS. And the combination of QSM and DTI can provide a more comprehensive evaluation of white matter integrity by utilizing different biophysical features.
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Imagem de Tensor de Difusão , Substância Branca , Humanos , Imagem de Tensor de Difusão/métodos , Feminino , Masculino , Substância Branca/diagnóstico por imagem , Substância Branca/patologia , Criança , Epilepsia Rolândica/diagnóstico por imagem , Epilepsia Rolândica/fisiopatologia , Estudos de Casos e Controles , Anisotropia , Mapeamento Encefálico/métodos , Pré-Escolar , AdolescenteRESUMO
In this case report, we describe a 14-year-old patient with a novel RyR2 gene mutation (c.6577G > T/p.Val2193Leu), identified through a comprehensive review of medical history, examination findings, and follow-up data. The pathogenic potential of this mutation, which results in the loss of some interatomic forces and compromises the closure of the RyR2 protein pore leading to calcium leakage, was analyzed using the I-TASSER Suite to predict the structural changes in the protein. This mutation manifested clinically as co-morbid catecholaminergic polymorphic ventricular tachycardia (CPVT) and benign epilepsy with centrotemporal spikes (BECTS), a combination not previously documented in the same patient. While seizures were successfully managed with levetiracetam, the patient's exercise-induced syncope episodes could not be controlled with metoprolol, highlighting the complexity and challenge in managing CPVT associated with this novel RyR2 variation.
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Mutação , Canal de Liberação de Cálcio do Receptor de Rianodina , Taquicardia Ventricular , Humanos , Canal de Liberação de Cálcio do Receptor de Rianodina/genética , Taquicardia Ventricular/genética , Taquicardia Ventricular/tratamento farmacológico , Adolescente , Masculino , Epilepsia Rolândica/genética , Epilepsia Rolândica/tratamento farmacológico , EletrocardiografiaRESUMO
BACKGROUND: Benign epilepsy with centrotemporal spikes (BECTS) is a common childhood epilepsy syndrome, accompanied by behavioral problems and cognitive impairments. Previous studies of BECTS-related brain structures applied univariate analysis and showed inconsistent results. And neurotransmitter patterns associated with brain structural alterations were still unclear. METHODS: Structural images of twenty-one drug-naïve children with BECTS and thirty-five healthy controls (HCs) were scanned. Segmented gray matter volume (GMV) images were decomposed into independent components (ICs) using the source-based morphometry method. Then spatial correlation analyses were applied to examine possible relationships between GMV changes and neurotransmitter systems. RESULTS: Compared with HCs, drug-naïve children with BECTS showed increased volume in one GMV component (IC7), including bilateral precentral gyrus, bilateral supplementary motor area, left superior frontal cortex, bilateral middle/ inferior frontal cortex and bilateral anterior/ middle cingulate cortex. A positive correlation was observed between one GMV component (IC6) and seizure frequency. There were significantly positive correlations between abnormal GMV in IC7 and serotonergic, GABAergic and glutamatergic systems. CONCLUSION: These findings provided further evidence of changed GMV in drug-naïve children with BECTS related to their behavioral problems and cognitive impairments, and associated neurotransmitters which could help to better understand neurobiological mechanisms and underlying molecular mechanisms of BECTS. IMPACT: The article provides further evidence of changed gray matter volume in drug-naïve children with BECTS related to their behavioral problems and cognitive impairments as well as associated neurotransmitters. Most literature to date has applied univariate analysis and showed inconsistent results, and neurotransmitter patterns associated with brain structural alterations were still unclear. Therefore, this article uses multivariate method and JuSpace toolbox to fill the gap. Significantly increased gray matter volume was found in drug-naïve children with BECTS compared with healthy controls. Abnormal gray matter volume was significantly correlated with clinical data and specific neurotransmitters.
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Encéfalo , Epilepsia Rolândica , Substância Cinzenta , Imageamento por Ressonância Magnética , Humanos , Criança , Epilepsia Rolândica/fisiopatologia , Epilepsia Rolândica/diagnóstico por imagem , Feminino , Masculino , Substância Cinzenta/diagnóstico por imagem , Substância Cinzenta/patologia , Encéfalo/diagnóstico por imagem , Encéfalo/patologia , Estudos de Casos e Controles , Neurotransmissores/metabolismo , AdolescenteRESUMO
PURPOSE: Our aim was to assess intelligence, visual perception and working memory in children with new-onset Rolandic epilepsy (RE) and children with Rolandic discharges without seizures (RD). METHODS: The participants in the study were 12 children with RE and 26 children with RD aged 4 to 10 years (all without medication and shortly after diagnosis) and 31 healthy controls. Their cognitive performance was assessed using the German versions of the Wechsler Preschool and Primary Scale of Intelligence (WPPSI-III), the Wechsler Intelligence Scale for Children (WISC-IV), the Developmental Test of Visual Perception-2 (DTVP-2), the Developmental Test of Visual Perception-Adolescent and Adult (DTVP-A) (each according to age) and the Word Order, Hand Movements and Spatial Memory subtests of the German version of the Kaufman Assessment Battery for Children (K-ABC). RESULTS: The comparison of the entire group of children with RE/RD and the control group conducted in the first step of our analysis revealed a weaker performance of the children with RE/RD in all cognitive domains. Significant deficits, however, were found exclusively in the RD group. Compared to the controls, they performed significantly weaker regarding IQ (full scale IQ: p < 0.001; verbal IQ: p < 0.001; performance IQ: p = 0.002; processing speed: p = 0.005), visual perception (general visual perception: p = 0.005; visual-motor integration: p = 0.002) and working memory (WISC working memory: p = 0.002 and K-ABC Word Order (p = 0.010) and Hand Movements (p = 0.001) subtests. Also, the children without seizures scored significantly lower than those with seizures on the WISC Working Memory Index (p = 0.010) and on the K-ABC Word Order (p = 0.021) and Hand Movements (p = 0.027) subtests. Further analysis of our data demonstrated the particular importance of the family context for child development. Significant cognitive deficits were found only in children with RD from parents with lower educational levels. This group consistently scored lower compared to the control group regarding IQ (full scale IQ: p < 0.001; verbal IQ: p < 0.001; performance IQ: p = 0.012; processing speed: p = 0.034), visual perception (general visual perception: p = 0.018; visual-motor integration: p = 0.010) and auditory working memory (WISC working memory: p = 0.014). Furthermore, compared to the children with RE, they performed significantly weaker on verbal IQ (p = 0.020), auditory working memory consistently (WISC working memory: p = 0.027; K-ABC: Word Order: p = 0.046) as well as in one of the K-ABC spatial working memory subtests (Hand Movements: p = 0.029). Although we did not find significant deficits in children with new-onset RE compared to healthy controls, the performance of this group tended to be weaker more often. No statistically significant associations were observed between selected clinical markers (focus types: centrotemporal/other foci/laterality of foci and spread of Rolandic discharges) and cognitive test results. Except for spatial working memory, we also found no evidence that the age of our patients at the time of study participation was of significant importance to their cognitive performance. CONCLUSIONS: Our study provides some evidence that children with Rolandic discharges, with and without seizures, may be at higher risk of cognitive impairment. In addition to medical care, we emphasise early differentiated psychosocial diagnostics to provide these children and their families with targeted support if developmental problems are present.
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Epilepsia Rolândica , Memória de Curto Prazo , Criança , Pré-Escolar , Humanos , Cognição , Eletroencefalografia , Epilepsia Rolândica/complicações , Epilepsia Rolândica/psicologia , Inteligência , Testes Neuropsicológicos , Convulsões , Percepção VisualRESUMO
OBJECTIVE: Children with self-limited epilepsy characterized by centrotemporal spikes (SeLECTS) exhibit cognitive deficits in memory during the active phase, but there is currently a lack of studies and techniques to assess their memory development after well-controlled seizures. In this study, we employed eye-tracking techniques to investigate visual memory and its association with clinical factors and global intellectual ability, aiming to identify potential risk factors by examining encoding and recognition processes. METHODS: A total of 26 recruited patients diagnosed with SeLECTS who had been seizure-free for at least 2 years, along with 24 control subjects, underwent Wechsler cognitive assessment and an eye-movement-based memory task while video-electroencephalographic (EEG) data were recorded. Fixation and pupil data related to eye movements were utilized to detect distinct memory processes and subsequently to compare the cognitive performance of patients exhibiting different regression patterns on EEG. RESULTS: The findings revealed persistent impairments in visual memory among children with SeLECTS after being well controlled, primarily observed in the recognition stage rather than the encoding phase. Furthermore, the age at onset, frequency of seizures, and interictal epileptiform discharges exhibited significant correlations with eye movement data. SIGNIFICANCE: Children with SeLECTS exhibit persistent recognition memory impairment after being well controlled for the disease. Controlling the frequency of seizures and reducing prolonged epileptiform activity may improve memory cognitive development. The application of the eye-tracking technique may provide novel insights into exploring memory cognition as well as underlying mechanisms associated with pediatric epilepsy.
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Epilepsia Rolândica , Tecnologia de Rastreamento Ocular , Humanos , Criança , Convulsões/diagnóstico , Cognição , Eletroencefalografia/métodos , Transtornos da Memória/etiologia , Transtornos da Memória/complicações , Epilepsia Rolândica/complicações , Epilepsia Rolândica/psicologiaRESUMO
Although the remission of self-limited epilepsy with centrotemporal spikes (SeLECTS) usually occurs by adolescence, deficits in cognition and behavior are not uncommon. Several functional magnetic resonance imaging (fMRI) studies have revealed connectivity disturbances in patients with SeLECTS associated with cognitive impairment. However, the disadvantages of fMRI are expensive, time-consuming, and motion sensitive. In the current study, we used a partial directed coherence (PDC) method to analyze electroencephalogram (EEG) for exploring brain connectivity in patients with SeLECTS. This study enrolled 38 participants (19 patients with SeLECTS and 19 healthy controls) for PDC analysis. Our results demonstrated that the controls had significantly higher PDC inflow connectivity in the F7, T3, FP1, and F8 channels than patients with SeLECTS. By contrast, the patients with SeLECTS demonstrated significantly higher PDC inflow connectivity than did the controls in the T5, Pz, and P4 channels. We also compared the PDC connectivity in different Brodmann areas between the patients with SeLECTS and the controls. The results revealed that the inflow connectivity in the BA9_46_L area was significantly higher in the controls than in the patients with SeLECTS, whereas the inflow connectivity in the MIF_L area 4 was significantly higher in the patients with SeLECTS than in the controls. Our proposed approach of combining EEG with PDC provides a convenient and useful tool for investigating functional connectivity in patients with SeLECTS. This approach is time-saving and inexpensive compared with fMRI, but it achieves similar results to fMRI.
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Epilepsia Rolândica , Epilepsia , Adolescente , Humanos , Eletroencefalografia/métodos , Encéfalo , Córtex Cerebral , Mapeamento Encefálico/métodos , Imageamento por Ressonância Magnética/métodos , Epilepsia Rolândica/patologiaRESUMO
Objective. To investigate the prevalence and risk factors for electrical status epilepticus during slow-wave sleep (ESES) in patients with self-limited epilepsy with centrotemporal spikes (SeLECTS). Methods. The clinical and follow-up data of children with SeLECTS were collected between 2017 and 2021. Patients were divided into typical ESES, atypical ESES, and non-ESES groups according to spike-wave indices (SWI). Clinical and electroencephalography characteristics were retrospectively analyzed. Logistic regression was used to identify risk factors for ESES. Results. A total of 95 patients with SeLECTS were enrolled. Seven patients (7.4%) developed typical ESES, 30 (31.6%) developed atypical ESES, 25 (26.3%) developed ESES at the first visit, and 12 (12.6%) developed ESES during treatment and follow-up. Multivariate logistic regression analysis showed that the risk factors for SeLECTS combined with ESES were Rolandic double or multiple spikes (OR = 8.626, 95% CI: 2.644-28.147, P < .001) and Rolandic slow waves (OR = 53.550, 95% CI: 6.339-452.368, P < .001). There were no significant differences in seizure characteristics, electroencephalogram (EEG) findings, or cognitive impairment between the atypical and typical ESES groups. Conclusion. More than one-third of the SeLECTS patients combined with ESES. Both atypical and typical ESES scores can affect cognitive function. On electroencephalography, interictal Rolandic double/multiple spikes and slow-wave abnormalities may indicate SeLECTS with ESES.
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Epilepsia Rolândica , Epilepsia , Sono de Ondas Lentas , Estado Epiléptico , Criança , Humanos , Sono , Estudos Retrospectivos , Prevalência , Eletroencefalografia , Fatores de RiscoRESUMO
OBJECTIVE: With research progress on Rolandic epilepsy (RE), its "benign" nature has been phased out. Clinicians are exhibiting an increasing tendency toward a more assertive treatment approach for RE. Nonetheless, in clinical practice, delayed treatment remains common because of the "self-limiting" nature of RE. Therefore, this study aimed to identify an imaging marker to aid treatment decisions and select a more appropriate time for initiating therapy for RE. METHODS: We followed up with children newly diagnosed with RE, classified them into medicated and non-medicated groups according to the follow-up results, and compared them with matched healthy controls. Before beginning follow-up visits, interictal magnetic data were collected using magnetoencephalography in treatment-naïve recently diagnosed patients. The spectral power of the whole brain during initial diagnosis was determined using minimum normative estimation combined with the Welch technique. RESULTS: A difference was observed in the magnetic source intensity within the left caudal anterior cingulate and precentral and postcentral gyri in the delta band between the medicated and non-medicated groups. The results revealed good discriminatory ability within the receiver operator characteristic curve. In the medicated group, there was a specific change in the frontotemporal magnetic source intensity, which shifted from high to low frequencies, compared with the healthy control group. SIGNIFICANCE: The intensity of the precentral gyrus magnetic source within the delta band showed good specificity. Considering the rigor of initial treatment, the intensity of the precentral gyrus magnetic source can provide some help as an imaging marker for initial RE treatment, particularly for the timing of treatment initiation.
Assuntos
Epilepsia Rolândica , Criança , Humanos , Epilepsia Rolândica/tratamento farmacológico , Eletroencefalografia/métodos , Magnetoencefalografia/métodos , Encéfalo/diagnóstico por imagem , Fenômenos MagnéticosRESUMO
PURPOSE: Benign Epilepsy with Centro-Temporal Spikes (BECTS) is a pediatric epilepsy with typically good seizure control. Although BECTS may increase patients' risk of developing neurological comorbidities, their clinical care and short-term outcomes are poorly quantified. METHODS: We retrospectively assessed adherence to National Institute for Health and Care Excellence (NICE) guidelines relating to specialist referral, electroencephalogram (EEG) conduct and annual review in the care of patients with BECTS, and measured their seizure, neurodevelopmental and learning outcomes at three years post-diagnosis. RESULTS: Across ten centers in England, we identified 124 patients (74 male) diagnosed with BECTS between 2015 and 2017. Patients had a mean age at diagnosis of 8.0 (95% CI = 7.6-8.4) years. 24/95 (25%) patients were seen by a specialist within two weeks of presentation; 59/100 (59%) received an EEG within two weeks of request; and 59/114 (52%) were reviewed annually. At three years post-diagnosis, 32/114 (28%) experienced ongoing seizures; 26/114 (23%) had reported poor school progress; 15/114 (13%) were diagnosed with a neurodevelopmental disorder (six autism spectrum disorder, six attention-deficit/hyperactivity disorder); and 10/114 (8.8%) were diagnosed with a learning difficulty (three processing deficit, three dyslexia). Center-level random effects models estimated neurodevelopmental diagnoses in 9% (95% CI: 2-16%) of patients and learning difficulty diagnoses in 7% (95% CI: 2-12%). CONCLUSIONS: In this multicenter work, we found variable adherence to NICE guidelines in the care of patients with BECTS and identified a notable level of neurological comorbidity. Patients with BECTS may benefit from enhanced cognitive and behavioral assessment and monitoring.
Assuntos
Transtorno do Espectro Autista , Epilepsia Rolândica , Humanos , Criança , Masculino , Epilepsia Rolândica/diagnóstico , Epilepsia Rolândica/epidemiologia , Epilepsia Rolândica/psicologia , Estudos Retrospectivos , Transtorno do Espectro Autista/complicações , Transtorno do Espectro Autista/diagnóstico , Transtorno do Espectro Autista/epidemiologia , Convulsões , EletroencefalografiaRESUMO
Self-limited epilepsy with centrotemporal spikes (SeLECTS) is one of the most frequent epilepsies in childhood, characterised by typical clinical presentation with characteristic EEG findings. This review investigates the existing knowledge regarding cognitive function, the potential effect of anti-seizure medicines on cognitive development as well as prognosis of SeLECTS based on recent studies. There is evidence supporting that SeLECTS may not be as benign as previously assumed due to the possible neurocognitive comorbidities.
Assuntos
Eletroencefalografia , Epilepsia Rolândica , Humanos , Cognição , PrognósticoRESUMO
Self-limited epilepsy with centrotemporal spikes (SeLECTS) is one of the most frequent epilepsies in childhood, characterised by typical clinical presentation with characteristic EEG findings. This review investigates the existing knowledge regarding cognitive function, the potential effect of anti-seizure medicines on cognitive development as well as prognosis of SeLECTS based on recent studies. There is evidence supporting that SeLECTS may not be as benign as previously assumed due to the possible neurocognitive comorbidities.
Assuntos
Eletroencefalografia , Epilepsia Rolândica , Humanos , Tratamento Conservador , Lábio , CogniçãoRESUMO
OBJECTIVE: Self-limiting Rolandic epilepsy (RE) is the most common epilepsy in school-age children. Seizures are generally infrequent, but cognitive, language, and motor coordination problems can significantly impact the child's life. To better understand brain structure and function changes in RE, we longitudinally assessed neurocognition, cortical thickness, and subcortical volumes. METHODS: At baseline, we recruited 30 participants diagnosed with RE and 24-healthy controls and followed up for 4.94 ± 0.8 years when the participants with RE were in seizure remission. Measures included were as follows: T1-weighted magnetic resonance brain imaging (MRI) with FreeSurfer analysis and detailed neuropsychological assessments. MRI and neuropsychological data were compared between baseline and follow-up in seizure remission. RESULTS: Longitudinal MRI revealed excess cortical thinning in the left-orbitofrontal (p = 0.0001) and pre-central gyrus (p = 0.044). There is a significant association (p = 0.003) between a reduction in cortical thickness in the left-orbitofrontal cluster and improved processing of filtered words. Longitudinal neuropsychology revealed significant improvements in the symptoms of developmental coordination disorder (DCD, p = 0.005) in seizure remission. CONCLUSIONS: There is evidence for altered development of neocortical regions between active seizure state and seizure remission in RE within two clusters maximal in the left-orbitofrontal and pre-central gyrus. There is significant evidence for improvement in motor coordination between active seizures and seizure remission and suggestive evidence for a decline in fluid intelligence and gains in auditory processing.
Assuntos
Epilepsia Rolândica , Criança , Humanos , Epilepsia Rolândica/diagnóstico por imagem , Estudos Prospectivos , Estudos Longitudinais , Convulsões/diagnóstico por imagem , Encéfalo/diagnóstico por imagem , Imageamento por Ressonância MagnéticaRESUMO
Functional connectivity hierarchy is an important principle in the process of brain functional organization and an important feature reflecting brain development. However, atypical brain network hierarchy organization in Rolandic epilepsy have not been systematically investigated. We examined connectivity alteration with age and its relation to epileptic incidence, cognition, or underlying genetic factors in 162 cases of Rolandic epilepsy and 117 typically developing children, by measuring fMRI multi-axis functional connectivity gradients. Rolandic epilepsy is characterized by contracting and slowing expansion of the functional connectivity gradients, highlighting the atypical age-related change of the connectivity hierarchy in segregation properties. The gradient alterations are relevant to seizure incidence, cognition, and connectivity deficit, and development-associated genetic basis. Collectively, our approach provides converging evidence for atypical connectivity hierarchy as a system-level substrate of Rolandic epilepsy, suggesting this is a disorder of information processing across multiple functional domains, and established a framework for large-scale brain hierarchical research.