Faculty Profiles - AKIYAMA Tomoyuki

Papers - AKIYAMA Tomoyuki

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CSF/plasma ratios of amino acids: Reference data and transports in children Reviewed

Tomoyuki Akiyama, Katsuhiro Kobayashi, Akihito Higashikage, Junko Sato, Harumi Yoshinaga

BRAIN & DEVELOPMENT 36 ( 1 ) 3 - 9 2014.1

Language：English Publishing type：Research paper (scientific journal) Publisher：ELSEVIER SCIENCE BV

Objective: We intended to investigate the effects of age, gender, and medications on amino acid cerebrospinal fluid (CSF)/plasma ratios in children, and to determine whether amino acid transports across the blood CSF barrier in children differ from those in adults. Patients and methods: Amino acid concentrations measured by ion-exchange high-performance liquid chromatography were used (CSF from 99 children, simultaneously collected plasma from 76 children). Influence of age, gender, and medications on the amino acid CSF concentrations and CSF/plasma ratios were analyzed by linear multiple regression. Interactions of amino acid transports were analyzed by correlation analysis of CSF/plasma ratios. Results: CSF/plasma ratios of serine, valine, histidine, and arginine were higher in younger children. The glutamate CSF/plasma ratio was higher in older children. Serine, alanine, threonine, valine, and histidine CSF/plasma ratios were lower in females. Glutamine, methionine, tyrosine, and phenylalanine CSF/plasma ratios were elevated with valproate therapy. Serine, threonine, valine, leucine, and tyrosine CSF/plasma ratios were lower with clobazam therapy. The asparagine CSF/plasma ratio was elevated with pyridoxal phosphate therapy. Transports of most essential neutral amino acids interacted with each other, as did neutral amino acids with low molecular weights. Cationic amino acids interacted with each other and some essential neutral amino acids. Acidic amino acids had no interactions with other amino acids. Conclusions: Age, gender, and anti-epileptic drugs affect amino acid CSF/plasma ratios in children. Transport interactions between amino acids in children showed no remarkable difference from those of adults and generally followed the substrate specificities of multiple amino acid transport systems. (C) 2012 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

DOI： 10.1016/j.braindev.2012.12.001

A Japanese adult case of guanidinoacetate methyltransferase deficiency. Reviewed International journal

Akiyama T, Osaka H, Shimbo H, Nakajiri T, Kobayashi K, Oka M, Endoh F, Yoshinaga H

JIMD reports 12 65 - 69 2014

Language：English Publishing type：Research paper (scientific journal)

Guanidinoacetate methyltransferase (GAMT) deficiency is a rare disorder of creatine synthesis resulting in cerebral creatine depletion. We present a 38-year-old patient, the first Japanese case of GAMT deficiency. Developmental delay started after a few months of age with a marked delay in language, which resulted in severe intellectual deficit. She showed hyperactivity and trichotillomania from childhood. Epileptic seizures appeared at 18 months and she had multiple types of seizures including epileptic spasms, brief tonic seizures, atypical absences, complex partial seizures with secondary generalization, and "drop" seizures. They have been refractory to multiple antiepileptic drugs. Although there have been no involuntary movements, magnetic resonance imaging revealed T2 hyperintense lesions in bilateral globus pallidi. Motor regression started around 30 years of age and the patient is now able to walk for only short periods. Very low serum creatinine levels measured by enzymatic method raised a suspicion of GAMT deficiency, which was confirmed by proton magnetic resonance spectroscopy and urinary guanidinoacetate assay. GAMT gene analysis revealed that the patient is a compound heterozygote of c.578A>G, p.Gln193Arg and splice site mutation, c.391G>C, p.Gly131Arg, neither of which have been reported in the literature. We also identified two aberrant splice products from the patient's cDNA analysis. The patient was recently started on supplementation of high-dose creatine and ornithine, the effects of which are currently under evaluation. Although rare, patients with developmental delay, epilepsy, behavioral problems, and movement disorders should be vigorously screened for GAMT deficiency, as it is a treatable disorder.

DOI： 10.1007/8904_2013_245

Neocortical pathological high-frequency oscillations are associated with frequency-dependent alterations in functional network topology Reviewed

George M. Ibrahim, Ryan Anderson, Tomoyuki Akiyama, Ayako Ochi, Hiroshi Otsubo, Gabrielle Singh-Cadieux, Elizabeth Donner, James T. Rutka, O. Carter Snead, Sam M. Doesburg

JOURNAL OF NEUROPHYSIOLOGY 110 ( 10 ) 2475 - 2483 2013.11

Language：English Publishing type：Research paper (scientific journal) Publisher：AMER PHYSIOLOGICAL SOC

Synchronization of neural oscillations is thought to integrate distributed neural populations into functional cell assemblies. Epilepsy is widely regarded as a disorder of neural synchrony. Knowledge is scant, however, regarding whether ictal changes in synchrony involving epileptogenic cortex are expressed similarly across various frequency ranges. Cortical regions involved in epileptic networks also exhibit pathological high-frequency oscillations (pHFOs, >80 Hz), which are increasingly utilized as biomarkers of epileptogenic tissue. It is uncertain how pHFO amplitudes are related to epileptic network connectivity. By calculating phase-locking values among intracranial electrodes implanted in children with intractable epilepsy, we constructed ictal connectivity networks and performed graph theoretical analysis to characterize their network properties at distinct frequency bands. Ictal data from 17 children were analyzed with a hierarchical mixed-effects model adjusting for patient-level covariates. Epileptogenic cortex was defined in two ways: 1) a hypothesis-driven method using the visually defined seizure-onset zone and 2) a data-agnostic method using the high-frequency amplitude of each electrode. Epileptogenic cortex exhibited a logarithmic decrease in interregional functional connectivity at high frequencies (>30 Hz) during seizure initiation and propagation but not at termination. At slower frequencies, conversely, epileptogenic cortex expressed a relative increase in functional connectivity. Our findings suggest that pHFOs reflect epileptogenic network interactions, yielding theoretical support for their utility in the presurgical evaluation of intractable epilepsy. The view that abnormal network synchronization plays a critical role in ictogenesis and seizure dynamics is supported by the observation that functional isolation of epileptogenic cortex at high frequencies is absent at seizure termination.

DOI： 10.1152/jn.00034.2013

乳児期早期に皮質性ミオクローヌスを認めたDravet症候群の一例

小林由典, 秋山倫之, 中尻智史, 柴田敬, 井上拓志, 小林勝弘, 大守伊織, 大内田守, 吉永治美

臨床神経生理学 41 ( 5 ) 490 - 490 2013.10

Cortical contribution to scalp EEG gamma rhythms associated with epileptic spasms Reviewed

Katsuhiro Kobayashi, Kazushi Miya, Tomoyuki Akiyama, Fumika Endoh, Makio Oka, Harumi Yoshinaga, Yoko Ohtsuka

BRAIN & DEVELOPMENT 35 ( 8 ) 762 - 770 2013.9

Language：English Publishing type：Research paper (scientific journal) Publisher：ELSEVIER SCIENCE BV

The cortical contribution for the generation of gamma rhythms detected from scalp ictal EEG was studied in unique cases of epileptic spasms and a review of the related literature was conducted. Ictal scalp gamma rhythms were investigated through time frequency analysis in two cases with a combination of focal seizures and spasms and another case with spasms associated with cortical dysplasia. In the two patients with combined seizures, the scalp distribution of ictal gamma rhythms was related to that of focal seizure activity. In the third patient, an asymmetric distribution of the ictal scalp gamma rhythms was transiently revealed in correspondence to the dysplasic cortex during hormonal treatment. Therefore, the dominant region of scalp gamma rhythms may correspond to the epileptogenic cortical area. The current findings have reinforced the possibility of the cortical generation of ictal scalp gamma rhythms associated with spasms The detection of high frequencies through scalp EEG is a technical challenge, however, and the clinical significance of scalp gamma rhythms may not be the same as that of invasively recorded high frequencies. Further studies on the pathophysiological mechanisms related to the generation of spasms involving high frequencies are necessary in the future, and the development of animal models of spasms will play an important role in this regard. (C) 2013 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

DOI： 10.1016/j.braindev.2012.12.012

West症候群を発症した46,XY,der(16)t(2;16)(p22;p13.3)の一家系

柴田敬, 岡牧郎, 兵頭勇紀, 秋山倫之, 吉永治美, 小林勝弘

てんかん研究 31 ( 2 ) 474 - 474 2013.9

後方1/4離断術が奏功した大田原症候群の一例

上利崇, 近藤聡彦, 秋山倫之, 林裕美子, 桃木恵美子, 井上拓志, 岡牧郎, 小林勝弘, 佐々木達也, 金恭平, 伊達勲

てんかん研究 31 ( 1 ) 90 - 91 2013.6

小児てんかんの診療実態に関する疫学調査(第1報)

花岡義行, 柴田敬, 岡牧郎, 秋山倫之, 小林勝弘, 吉永治美

脳と発達 45 ( Suppl. ) S236 - S236 2013.5

Clinical implications of preceding positive spikes in patients with benign partial epilepsy and febrile seizures Reviewed

Harumi Yoshinaga, Katsuhiro Kobayashi, Tomoyuki Akiyama, Takashi Shibata, Fumika Endoh, Yoko Ohtsuka

BRAIN & DEVELOPMENT 35 ( 4 ) 299 - 306 2013.4

Language：English Publishing type：Research paper (scientific journal) Publisher：ELSEVIER SCIENCE BV

Purpose: To clarify the clinical implications of the preceding positive spikes (PPSs) observed primarily in rolandic spikes, we analyzed PPSs in the rolandic and occipital spikes observed in the electroencephalograms (EEGs) of patients with two types of benign partial epilepsies (benign childhood epilepsy with centro-temporal spikes [BECT] and Panayiotopoulos syndrome [PS]) and febrile seizures (FS). Subjects and methods: We identified patients from our outpatient EEG database that were seen between 2006 and 2008 that had BECT, PS, and FS with rolandic or occipital spikes. We generated an averaged spike for each patient from the rolandic and occipital spikes that were detected using an automatic spike detection and clustering system. We compared the presence rate of the averaged spikes with the PPS among the three groups (BECT vs. PS vs. FS) using sequential mapping. Results: We identified 25 BECT, 18 PS, and 15 FS patients with rolandic spikes. Fifteen BECT and nine PS patients exhibited a PPS in their averaged rolandic spikes, whereas only four FS patients did. Three of these four FS patients later developed afebrile seizures, and one of them was diagnosed as having PS. We analyzed eight PS and six FS patients with occipital spikes. Five PS patients exhibited a PPS in their averaged occipital spikes, whereas only one FS patient did. This FS patient later developed prolonged autonomic febrile seizures. Conclusion: PPSs are observed not only in rolandic spikes associated with BECT that is related strictly to sylvian seizures, but also in rolandic and occipital spikes associated with PS. Although PPSs are rare in such spikes observed in FS, patients with FS and PPSs may have an increased risk of developing afebrile seizures or prolonged autonomic febrile seizures. Further studies are warranted to determine the diagnostic utility of PPSs as a marker of the future development of epilepsy when they are observed in FS patients. (C) 2012 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

DOI： 10.1016/j.braindev.2012.06.006

Disruption of Rolandic Gamma-Band Functional Connectivity by Seizures is Associated with Motor Impairments in Children with Epilepsy Reviewed

George M. Ibrahim, Tomoyuki Akiyama, Ayako Ochi, Hiroshi Otsubo, Mary Lou Smith, Margot J. Taylor, Elizabeth Donner, James T. Rutka, O. Carter Snead, Sam M. Doesburg

PLOS ONE 7 ( 6 ) e39326 2012.6

Language：English Publishing type：Research paper (scientific journal) Publisher：PUBLIC LIBRARY SCIENCE

Although children with epilepsy exhibit numerous neurological and cognitive deficits, the mechanisms underlying these impairments remain unclear. Synchronization of oscillatory neural activity in the gamma frequency range (>30 Hz) is purported to be a mechanism mediating functional integration within neuronal networks supporting cognition, perception and action. Here, we tested the hypothesis that seizure-induced alterations in gamma synchronization are associated with functional deficits. By calculating synchrony among electrodes and performing graph theoretical analysis, we assessed functional connectivity and local network structure of the hand motor area of children with focal epilepsy from intracranial electroencephalographic recordings. A local decrease in inter-electrode phase synchrony in the gamma bands during ictal periods, relative to interictal periods, within the motor cortex was strongly associated with clinical motor weakness. Gamma-band ictal desychronization was a stronger predictor of deficits than the presence of the seizure-onset zone or lesion within the motor cortex. There was a positive correlation between the magnitude of ictal desychronization and impairment of motor dexterity in the contralateral, but not ipsilateral hand. There was no association between ictal desynchronization within the hand motor area and non-motor deficits. This study uniquely demonstrates that seizure-induced disturbances in cortical functional connectivity are associated with network-specific neurological deficits.

DOI： 10.1371/journal.pone.0039326

Occipital lobe epilepsy in children: Characterization, evaluation and surgical outcomes Reviewed

George M. Ibrahim, Aria Fallah, Gregory W. Albert, Teresa Withers, Hiroshi Otsubo, Ayako Ochi, Tomoyuki Akiyama, Elizabeth J. Donner, Shelly Weiss, O. Carter Snead, James M. Drake, James T. Rutka

EPILEPSY RESEARCH 99 ( 3 ) 335 - 345 2012.5

Language：English Publishing type：Research paper (scientific journal) Publisher：ELSEVIER SCIENCE BV

Introduction: Occipital lobe epilepsy (OLE) poses a diagnostic challenge to clinicians. Here, we present our experience in the surgical management of OLE in children using magnetoencephalography (MEG) in the pre-operative evaluation.
Methods: Retrospective chart review was performed from 2000 to 2010 to identify patients with OLE. Patients were analyzed in two categories: isolated OLE (11 patients) and extended OLE (parietooccipital, temporooccipital, and temporoparietooccipital; 30 patients). Survival analysis and multivariate Cox proportional hazards regression were used to identify independent predictors of seizure outcome.
Results: Forty-one patients with a mean follow-up of 3.1 years were identified with an overall 68% rate of satisfactory seizure outcome. Patients with extended OLE had younger ages at seizure onset and different seizure semiologies compared with those with isolated OLE. None of the latter underwent insertion of subdural grid electrodes for localization of the epileptogenic zone compared with 77% of the former (p < 0.001). On multivariate analysis, the strongest independent predictor of unsatisfactory outcome was MEG dipoles in the occipital lobe contralateral to resection.
Conclusion: Here, we find similar seizure outcomes for isolated and extended OLE foci despite the use of less invasive strategies for the former. Furthermore, we describe the role of MEG in evaluation, surgical planning and prognostication of children with OLE. (C) 2012 Elsevier B.V. All rights reserved.

DOI： 10.1016/j.eplepsyres.2011.12.015

Histopathology of cortex and white matter in pediatric epileptic spasms: Comparison with those of partial seizures Reviewed

Yukiko Inage, William C. Halliday, Cristina Go, Ayako Ochi, Tomoyuki Akiyama, Mari Akiyama, Elysa Widjaja, Hiroshi Otsubo

BRAIN & DEVELOPMENT 34 ( 2 ) 118 - 123 2012.2

Language：English Publishing type：Research paper (scientific journal) Publisher：ELSEVIER SCIENCE BV

Epileptic spasms in older children have increasingly been recognized as a distinct seizure type and subset of these patients are considered for surgical resection. This study compares histopathology and magnetic resonance imaging (MRI), especially focusing the difference between the cortical grey matter and the subcortical white matter to understand the extensive epileptic brain in patients with epileptic spasms. We examined 22 patients consisting of 11 patients with epileptic spasms and 11 with partial seizures. Scalp video electroencephalography (EEG) showed interictal generalized epileptiform discharges (9 patients with epileptic spasms vs. 1 with partial seizures) and ictal generalized epileptiform discharges (10 vs. 3). We found MRI abnormalities in a single lobe (6 vs. 7) and multiple lobes (2 vs. 1). Surgical resections were performed across multiple lobes (9 vs. 2), comparing within a single lobe (2 vs. 9), (p < 0.001). Histopathology showed abnormal cortical organizations as FCD (2 vs. 5) and microdysgenesis (4 vs. 4), normal (4 vs. 1). Two patients with epileptic spasms showed hyaline proteoplasmic astrocytopathy. There were heterotopic neurons (10 vs. 10), cluster of oligodendroglia (8 vs. 7), balloon cells (2 vs. 5) and blurred myelination (1 vs. 4), in the white matter. Seizure-free outcomes were seen in seven patients with epileptic spasms (64%) and four with partial seizures (36%). The multilobar epileptogenic zones existed in patients with epileptic spasms, compared with the focal epileptogenic zone in patients with partial seizures. There was no difference of MRI and histopathology findings in cortex and subcortical white matter between two groups. (C) 2011 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

DOI： 10.1016/j.braindev.2011.03.009

High kurtosis of intracranial electroencephalogram as a marker of ictogenicity in pediatric epilepsy surgery Reviewed

Tomoyuki Akiyama, Makoto Osada, Masahide Isowa, Cristina Y. Go, Ayako Ochi, Irene M. Elliott, Mari Akiyama, O. Carter Snead, James T. Rutka, James M. Drake, Hiroshi Otsubo

CLINICAL NEUROPHYSIOLOGY 123 ( 1 ) 93 - 99 2012.1

Language：English Publishing type：Research paper (scientific journal) Publisher：ELSEVIER IRELAND LTD

Objective: We determined whether kurtosis analysis of intracranial electroencephalogram (EEG) can estimate the localization of the epileptogenic zone.
Methods: We analyzed 29 pediatric epilepsy patients who underwent intracranial EEG before focal resective surgery. We localized the brain regions with high kurtosis, the seizure onset zone (SOZ) and the regions with high-rate, high-amplitude and long-duration interictal paroxysms >= 20 Hz. We tested correlations between the surgical resection of those regions and post-surgical seizure outcome, and correlations between kurtosis and the rate/amplitude/duration of interictal paroxysms.
Results: The resection of the regions with high kurtosis correlated with 1-year post-surgical seizure outcome (p = 0.028) but not with 2-year outcome. Kurtosis showed more significant correlation with 1-year seizure outcome than the SOZ and the rate/amplitude/duration of interictal paroxysms. Kurtosis showed positive, independent correlations with the amplitude and duration of interictal paroxysms (p < 0.0001) but not with the rate (p = 0.4).
Conclusions: The regions with high kurtosis provide more reliable information to predict seizure outcome than the SOZ and the regions with high-rate/amplitude and long-duration interictal paroxysms. Kurtosis reflects combined effects of the amplitude and duration of the interictal paroxysms.
Significance: High kurtosis suggests the regions with acquired ictogenicity within the irritative zone. (C) 2011 International Federation of Clinical Neurophysiology. Published by Elsevier Ireland Ltd. All rights reserved.

DOI： 10.1016/j.clinph.2011.05.026

Lateralized interictal epileptiform discharges during rapid eye movement sleep correlate with epileptogenic hemisphere in children with intractable epilepsy secondary to tuberous sclerosis complex Reviewed

Ayako Ochi, Ryan Hung, Shelly Weiss, Elysa Widjaja, Tonia To, Yoshihiro Nawa, Taiki Shima, Cristina Go, Tomoyuki Akiyama, Elizabeth Donner, James Drake, James T. Rutka, O. Carter Snead, Hiroshi Otsubo

EPILEPSIA 52 ( 11 ) 1986 - 1994 2011.11

Language：English Publishing type：Research paper (scientific journal) Publisher：WILEY-BLACKWELL

Purpose: We assessed lateralization of interictal epileptiform discharges (IEDs) in children with intractable epilepsy secondary to tuberous sclerosis complexes (TSCs) during rapid eye movement sleep (R), compared with non-rapid eye movement sleep (NR) and wakefulness (W), to determine epileptogenicity of R-IEDs.
Methods: We retrospectively studied 23 children with TSC, who underwent prolonged scalp video-electroencephalography (EEG) and magnetic resonance imaging (MRI). We determined the lateralization of ictal EEG, clinical semiology, and the largest tuber on MRI. We analyzed a minimum of 100 IEDs during R, NR, and W to classify right/left/generalized spikes to compare the lateralization with ictal EEG, clinical semiology, and MRI.
Key Findings: R-IEDs were lateralized in 22 patients (96%), W-IEDs in 19 (83%), and NR-IEDs in 16 (70%). Ictal EEGs were lateralized in 15 patients (65%) and clinical semiology in 5 (22%). Ictal EEG lateralization was concordant to R-IEDs in 14 patients (93%), W-IEDs in 11 (73%), and NR-IEDs in 10 (67%). The lateralization of clinical semiology was concordant with R-IEDs in 4 (80%), W-IEDs in 3 (60%), and NR-IEDs in 2 (40%). MRI lateralization of the largest tuber was concordant with R-IEDs in 16 (70%), W-IEDs in 12 (52%), and NR-IEDs in 11 (48%). Thirteen patients (57%) underwent resective surgery, the hemisphere of which was concordant with R-IEDs in 13 (100%), W-IEDs in 8 (62%), and NR-IEDs in 8 (62%). Nine (69%) of them achieved good seizure control after surgery.
Significance: R provoked the most lateralized IEDs compared to NR and W in children with TSC. Lateralization of R-IEDs corresponded with the hemisphere of ictal EEG and largest tuber, and can be used to identify surgical candidacy in TSC children with intractable epilepsy.

DOI： 10.1111/j.1528-1167.2011.03198.x

Fast activity during EEG seizures in neonates Reviewed

Lakshmi Nagarajan, Soumya Ghosh, Linda Palumbo, Tomoyuki Akiyama, Hiroshi Otsubo

EPILEPSY RESEARCH 97 ( 1-2 ) 162 - 169 2011.11

Language：English Publishing type：Research paper (scientific journal) Publisher：ELSEVIER SCIENCE BV

Introduction: Paroxysmal fast activity (FA) has been proposed as a marker for epileptic networks. We explore the presence and significance of ictal FA on scalp video EEG seizures (EEG Sz) recorded in neonates.
Methodology: Forty two babies had 159 EEG Szs. The seizures were analysed for ictal FA, using a low frequency filter of 30 Hz. We explored the relationship of ictal FA to the occurrence of electroclinical and electrographic seizures, the use of phenobarbitone and to neurodevelopmental outcomes.
Results: Ictal FA occurred in 62 (39%) of the 159 EEG Szs. In the 62 seizures with ictal FA, 34 (55%) were electroclinical seizures (ECSz) and 28 (45%) were electrographic (ESz). In the remaining 97 seizures without ictal FA, 27 (28%) were ECSz and 70 (72%) ESz. There was a significant correlation (p=0.0006) between ictal FA and electroclinical seizures. There was no relationship between phenobarbitone and presence of ictal FA. There was no significant difference in the background EEG, neuroimaging abnormalities, neurodevelopmental impairment or post neonatal seizures between those with and without ictal FA.
Conclusions: Ictal FA is highly correlated to the occurrence of clinical features during an EEG seizure. The presence of ictal FA does not appear to influence neurodevelopmental outcomes. (C) 2011 Elsevier B.V. All rights reserved.

DOI： 10.1016/j.eplepsyres.2011.08.003

Focal resection of fast ripples on extraoperative intracranial EEG improves seizure outcome in pediatric epilepsy Reviewed

Tomoyuki Akiyama, Blathnaid McCoy, Cristina Y. Go, Ayako Ochi, Irene M. Elliott, Mari Akiyama, Elizabeth J. Donner, Shelly K. Weiss, O. Carter Snead, James T. Rutka, James M. Drake, Hiroshi Otsubo

EPILEPSIA 52 ( 10 ) 1802 - 1811 2011.10

Language：English Publishing type：Research paper (scientific journal) Publisher：WILEY-BLACKWELL

Purpose: High-frequency oscillations (HFOs), termed ripples at 80-200 Hz and fast ripples (FRs) at >200/250 Hz, recorded by intracranial electroencephalography (EEG), may be a valuable surrogate marker for the localization of the epileptogenic zone. We evaluated the relationship of the resection of focal brain regions containing high-rate interictal HFOs and the seizure-onset zone (SOZ) determined by visual EEG analysis with the postsurgical seizure outcome, using extraoperative intracranial EEG monitoring in pediatric patients and automated HFO detection.
Methods: We retrospectively analyzed 28 pediatric epilepsy patients who underwent extraoperative intracranial video-EEG monitoring prior to focal resection. Utilizing the automated analysis, we identified interictal HFOs during 20 min of sleep EEG and determined the brain regions containing high-rate HFOs. We investigated spatial relationships between regions with high-rate HFOs and SOZs. We compared the size of these regions, the surgical resection, and the amount of the regions with high-rate HFOs/SOZs within the resection area with seizure outcome.
Key Findings: Ten patients were completely seizure-free and 18 were not at 2 years after surgery. The brain regions with high-rate ripples were larger than those with high-rate FRs (p = 0.0011) with partial overlap. More complete resection of the regions with high-rate FRs significantly correlated with a better seizure outcome (p = 0.046). More complete resection of the regions with high-rate ripples tended to improve seizure outcome (p = 0.091); however, the resection of SOZ did not influence seizure outcome (p = 0.18). The size of surgical resection was not associated with seizure outcome (p = 0.22-0.39).
Significance: The interictal high-rate FRs are a possible surrogate marker of the epileptogenic zone. Interictal ripples are not as specific a marker of the epileptogenic zone as interictal FRs. Resection of the brain regions with high-rate interictal FRs in addition to the SOZ may achieve a better seizure outcome.

DOI： 10.1111/j.1528-1167.2011.03199.x

非定型良性小児部分てんかん診断における脳磁図検査の有用性 Reviewed

白石秀明, 萩野谷和裕, 中川栄二, 香坂忍, 金子裕, 須貝研司, 植松貢, 柿坂庸介, 中里信和, 花谷亮典, 秋山倫之, 齋藤伸治, 大坪宏

てんかん研究 29 ( 2 ) 337 - 337 2011.9

Jeavons syndrome existing as occipital cortex initiating generalized epilepsy Reviewed

Sorawit Viravan, Cristina Go, Ayako Ochi, Tomoyuki Akiyama, O. Carter Snead, Hiroshi Otsubo

EPILEPSIA 52 ( 7 ) 1273 - 1279 2011.7

Language：English Publishing type：Research paper (scientific journal) Publisher：WILEY-BLACKWELL

Purpose: Jeavons syndrome (JS) is one of the underreported epileptic syndromes characterized by eyelid myoclonia (EM), eye closure-induced seizures/electroencephalography (EEG) paroxysms, and photosensitivity. JS has been proposed as idiopathic generalized epilepsy (IGE) because of normal posterior dominant background activity and paroxysmal generalized ictal epileptiform discharges (EDs). However, we noticed subtle occipital EDs preceding EM and interictal posterior EDs using digital video-EEG. We studied clinical and EEG findings in JS to determine the specific occipital lobe relation to this "eye closure-induced'' reflex IGE.
Methods: We identified 12 children who met the diagnostic criteria of JS from January 2004 to April 2009 at the Hospital for Sick Children, Toronto, Canada. All patients had EM captured by video-EEG. We reviewed and described ictal EEG patterns, interictal abnormalities, and demographics, clinical, and neuroimaging findings.
Key Findings: All patients but one were female (92%). Age at seizure onset ranged from 1.5 to 9 years, with a mean age of 4.9 years. Six patients (50%) were previously diagnosed as having absence epilepsy and 10 patients were on antiepileptic medications. All 12 patients had normal posterior dominant alpha rhythm, reactive to eye opening and closure. Spiky posterior alpha activity was noted with sustained eye closure in six patients (50%). Interictally, there were generalized EDs found in 10 patients (83%); four of them also had focal interictal EDs over the posterior head region. Eleven patients (92%) had evidence of focal posterior ictal EDs. EM and/or paroxysmal EDs were induced by photic stimulation in 9 (75%) and hyperventilation in 7 (58%).
Significance: We observed two neurophysiologic findings in JS: (1) focal interictal EDs from posterior head region; and (2) predominant focal posterior ictal EDs preceding generalized EDs. Further clinical observations of seizures induced by eye closure, photic stimulation, and hyperventilation along with EEG paroxysms would raise the possibility of the occipital cortex initiating generalized epilepsy network involving the brainstem, and thalamocortical and transcortical pathways in JS.

DOI： 10.1111/j.1528-1167.2011.03068.x

Focal-onset myoclonic seizures and secondary bilateral synchrony Reviewed

Tomoyuki Akiyama, Elizabeth J. Donner, Cristina Y. Go, Ayako Ochi, O. Carter Snead, James T. Rutka, Hiroshi Otsubo

EPILEPSY RESEARCH 95 ( 1-2 ) 168 - 172 2011.6

Language：English Publishing type：Research paper (scientific journal) Publisher：ELSEVIER SCIENCE BV

We present a child with myoclonic seizures, epileptic spasms and generalized epileptiform discharges on scalp EEG. Magnetoencephalography and coherence-phase analysis for scalp EEG suggested focal onset in the left hemisphere. Intracranial video EEG confirmed seizure onset in the left frontal lobe with ictal high-frequency oscillations. Left frontal lobectomy suppressed his seizures. Subsets of myoclonic seizures can be focal in origin despite bilaterally synchronized epileptiform discharges on scalp EEG. (c) 2011 Elsevier B.V. All rights reserved.

DOI： 10.1016/j.eplepsyres.2011.02.006

Autoimmune Limbic Encephalitis as an Emerging Pediatric Condition: Case Report and Review of the Literature Reviewed

Blathnaid McCoy, Tomoyuki Akiyama, Elysa Widjaja, Cristina Go

JOURNAL OF CHILD NEUROLOGY 26 ( 2 ) 218 - 222 2011.2

Language：English Publisher：SAGE PUBLICATIONS INC

Limbic encephalitis, first described in the 1960s as a paraneoplastic condition, has emerged as an autoimmune condition, occurring often without evidence of an underlying tumor. Many novel autoantibodies have been identified, and this diagnosis is increasingly being made in the pediatric population. This article reports the case of a 16-year-old boy who presented following gastrointestinal illness with subacute evolution of neuropsychiatric symptoms. Brain magnetic resonance imaging revealed progressive hippocampal signal abnormality and swelling. N-methyl-D-aspartate (NMDA) receptor antibody was detected in serum. The patient responded well to pharmacological immunotherapy but has residual cognitive deficits. The available literature on this condition is reviewed. Limbic encephalitis should be considered in the differential diagnosis in children presenting with encephalopathy, particularly with neuropsychiatric manifestations. Long-term surveillance and close follow-up are required to accurately clarify tumor risk and natural history of this condition in children and balance these factors with risks of radiation exposure through imaging.

DOI： 10.1177/0883073810378536

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