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Authorship：Lead author,　Corresponding author   Language：English   Publishing type：Research paper (scientific journal)  

OBJECTIVE: This cross-sectional observational study aimed to assess gait performance, its correlation with physical functions, and its dual-task costs in children with Down syndrome (DS), to investigate their gait adaptations. METHODS: Gait performance with or without movie-watching tasks was evaluated in 17 children with DS (age, 6-12 years) and 51 age- and sex-matched controls, using three-dimensional gait analysis. We compared participants' demographics, physical functions, and gait performance without tasks between the two groups. In the DS group, correlations between physical functions, the intelligence quotient, and gait variables were assessed. Dual-task costs for gait variables were also compared between the two groups. RESULTS: Children with DS showed poorer balance function and muscle strength and lower gait quality than the control group. In the DS group, there was a significant positive correlation between gait speed, step length, and intelligence quotient. There were no correlations between the balance function, muscle strength, intelligence quotient, and gait quality. Dual-task costs for gait speed, step length, and cadence were greater in the DS group; however, there was no significant difference in dual-task costs for gait quality between the two groups. CONCLUSION: These findings highlight the importance of providing appropriate interventions for motor functions in school-aged children with DS based on their gait performance in single- and dual-task conditions, as well as on their intelligence quotient.

DOI： 10.1016/j.braindev.2022.11.001

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Authorship：Corresponding author   Language：English   Publishing type：Research paper (scientific journal)  

OBJECTIVES: Ataxic-rigid gait is a characteristic gait pathology in patients with Rett syndrome (RTT). In the present study, we aimed to quantitatively evaluate gait pathology in patients with RTT using three-dimensional gait analysis (3DGA). METHODS: We performed 3DGA in 11 patients with RTT ranging from 5 to 18 years (median age, 9 years) and in 33 age-matched healthy female controls. We compared the results of 3DGA, including spatiotemporal gait parameters and comprehensive indices of gait kinematics, such as the Gait Deviation Index (GDI) and Gait Profile Score (GPS), between the two groups. The GPS consists of nine sub-indices called Gait Variable Scores (GVSs). Decline in GDI or elevation of GPS and GVS indicated greater abnormal gait pathology. RESULTS: The patients demonstrated significantly slower walking speed, lower step length/length of the lower extremities, lower cadence, wider step width, and higher coefficient of variation of step length than the controls. Moreover, the patients had a lower GDI and higher GPS than the controls. The patients also exhibited higher GVSs for eight out of nine gait kinematics, particularly the sagittal plane in the pelvis, hip, knee, and ankle joint; coronal plane in the pelvis and hip joint; and horizontal plane in the pelvis than the controls. CONCLUSIONS: Quantitative evaluation of gait pathology in patients with RTT is possible using 3DGA. We found that in addition to ataxic-rigid gait, abnormalities in the coronal plane of the pelvis and hip joint and the horizontal plane of the pelvis were prominent.

DOI： 10.1016/j.ejpn.2022.11.010

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BACKGROUND AND PURPOSE: West syndrome is a developmental and epileptic encephalopathy characterized by epileptic spasms, neurodevelopmental regression, and a specific EEG pattern called hypsarrhythmia. Our aim was to investigate the brain activities related to hypsarrhythmia at onset and focal epileptiform discharges in the remote period in children with West syndrome using simultaneous electroencephalography and fMRI recordings. MATERIALS AND METHODS: Fourteen children with West syndrome underwent simultaneous electroencephalography and fMRI at the onset of West syndrome. Statistically significant blood oxygen level-dependent responses related to hypsarrhythmia were analyzed using an event-related design of 4 hemodynamic response functions with peaks at 3, 5, 7, and 9 seconds after the onset of each event. Six of 14 children had focal epileptiform discharges after treatment and underwent simultaneous electroencephalography and fMRI from 12 to 25 months of age. RESULTS: At onset, positive blood oxygen level-dependent responses were seen in the brainstem (14/14 patients), thalami (13/14), basal ganglia (13/14), and hippocampi (13/14), in addition to multiple cerebral cortices. Group analysis using hemodynamic response functions with peaks at 3, 5, and 7 seconds showed positive blood oxygen level-dependent responses in the brainstem, thalamus, and hippocampus, while positive blood oxygen level-dependent responses in multiple cerebral cortices were seen using hemodynamic response functions with peaks at 5 and 7 seconds. In the remote period, 3 of 6 children had focal epileptiform discharge-related positive blood oxygen level-dependent responses in the thalamus, hippocampus, and brainstem. CONCLUSIONS: Positive blood oxygen level-dependent responses with hypsarrhythmia appeared in the brainstem, thalamus, and hippocampus on earlier hemodynamic response functions than the cerebral cortices, suggesting the propagation of epileptogenic activities from the deep brain structures to the neocortices. Activation of the hippocampus, thalamus, and brainstem was still seen in half of the patients with focal epileptiform discharges after adrenocorticotropic hormone therapy.

DOI： 10.3174/ajnr.A7646

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Authorship：Corresponding author   Language：English   Publishing type：Research paper (scientific journal)  

BACKGROUND: Of the patients with glucose transporter 1 deficiency syndrome (GLUT1-DS), 90% have a pathologic gait. Ataxic-spastic and ataxic gaits are seen in 35% of patients each. A ketogenic diet and modified Atkins diet (MAD) are effective therapy in GLUT1-DS in terms of both the seizures and movement disorder. A three-dimensional gait analysis (3DGA) system can be used to evaluate gait quantitatively using spatiotemporal data and gait kinematics. We performed 3DGA in three ambulatory patients with GLUT1-DS to evaluate the characteristics of their gait pathology, and we compared the gait variables before and after enhancing the MAD in one patient. METHODS: After examination by pediatric neurologists and pediatric orthopedic surgeons, 3DGA was performed. We assessed walking speed, step length, step width, gait variability, Gait Deviation Index (GDI), Gait Profile Score (GPS), and Gait Variable Score (GVS). RESULTS: All three patients had a low GDI and high GPS, comprehensive indices of gait pathology. The unstable gait pattern featured a wide step width in one patient and high gait variability in two patients. In the sagittal plane, the patients had increased GVSs in the knee and ankle joints due to excessive knee flexion or extension and excessive ankle plantarflexion. In the horizontal plane, the patients had increased GVSs in the pelvis, hips, and foot due to excessive rotation during walking. After enhancing the MAD, GDI, GPS, and GVSs improved. CONCLUSIONS: 3DGA has potential for quantifying the characteristics of gait pathology and its improvement with dietary therapy in patients with GLUT1-DS.

DOI： 10.1016/j.pediatrneurol.2022.04.012

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Authorship：Lead author,　Corresponding author   Language：English   Publishing type：Research paper (scientific journal)  

BACKGROUND: We aimed to investigate electroencephalography (EEG)-functional magnetic resonance imaging (fMRI) findings to elucidate the interictal epileptiform discharge (IED)-related functional alterations in deep brain structures and the neocortex in childhood epilepsy with centrotemporal spikes (CECTS). METHODS: Ten children with CECTS (median age 8.2 years), referred to our hospital within a year of onset, were eligible for inclusion. They underwent EEG-fMRI recording during sleep. Llongitudinal evaluations, including medical examinations, intelligence tests, and questionnaires about developmental disabilities, were performed. The initial evaluation was performed at the same time as the EEG-fMRI, and the second evaluation was performed over 2 years after the initial evaluation. RESULTS: Three children were unable to maintain sleep during the EEG-fMRI recording, and the remaining seven children were eligible for further assessment. All patients showed unilateral-dominant centrotemporal spikes during scans. One patient had only positive hemodynamic responses, while the others had both positive and negative hemodynamic responses. All patients showed IED-related hemodynamic responses in the bilateral neocortex. For deep brain structures, IED-related hemodynamic responses were observed in the cingulate gyrus (n = 4), basal ganglia (n = 3), thalamus (n = 2), and default mode network (n = 1). Seizure frequencies at the second evaluation were infrequent or absent, and the longitudinal results of intelligence tests and questionnaires were within normal ranges. CONCLUSIONS: We demonstrated that IEDs affect broad brain areas, including deep brain structures such as the cingulate gyrus, basal ganglia, and thalamus. Deep brain structures may play an important role in the pathophysiology of CECTS.

DOI： 10.1111/ped.15001

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Authorship：Lead author,　Corresponding author   Publishing type：Research paper (scientific journal)   Publisher：Elsevier BV  

DOI： 10.1016/j.earlhumdev.2021.105478

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BACKGROUND: Although previous studies have identified asymmetry in gait parameters in children with developmental coordination disorder (DCD), they have not investigated whether these findings coexist with asymmetry in gait quality, as represented by the gait deviation index (GDI). RESEARCH QUESTION: This study sought to measure gait symmetry in children with traits of DCD (DCD trait) to elucidate the characteristic gait pattern. METHODS: This study included 172 (82 girls, 90 boys) participants with and without DCD trait (age: 6-12 years), as assessed using the Japanese version of the Developmental Coordination Disorder Questionnaire (DCDQ-J), which consists of three subscales. Three-dimensional gait analysis data were obtained during the gait trials. GDI, step length, and step time data were recorded for both the right and left legs, and symmetry ratios were calculated. RESULTS: Participants in the DCD trait group (n = 30) had a significantly lower GDI score (p < 0.0001) and a higher GDI symmetry ratio (p = 0.004) than typically developing children. Furthermore, DCD trait was related to the GDI symmetry ratio (odds ratio, 1.2; p = 0.001; 95 % confidence interval, 1.062-1.289). The control during movement, which was a DCDQ-J subscale, was negatively correlated with the GDI symmetry ratio (r=-0.257, p<0.001). SIGNIFICANCE: This study's results lend support to the hypothesis that an increase in the GDI symmetry ratio is associated with the DCD trait. A quantitative assessment of gait performance is important to help identify gait asymmetry, including gait quality, in children with a DCD trait. Children with a DCD trait have a poor ability to control the interaction of lower leg movements, which may cause bilateral asymmetry in gait quality.

DOI： 10.1016/j.gaitpost.2021.05.027

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OBJECTIVE: The authors recently reported a novel subsecond analysis method of analyzing EEG-functional MRI (fMRI) to improve the detection rate of epileptic focus. This study aims to validate the utility of this method for presurgical evaluation in pharmacoresistant focal epilepsy. METHODS: Among 13 patients with focal epilepsy undergoing presurgical examinations including simultaneous EEG-fMRI at 3T, 11 patients had interictal epileptiform discharges (IEDs) during fMRI. The authors used the sequence of topographic maps during the IEDs as a reference to obtain subsecond fMRI activation maps with the same temporal resolution as the EEG data, and constructed "spike-and-slow-wave-activation-summary" (SSWAS) maps that showed the activation frequency of voxels during IEDs. Clusters were defined by thresholding the SSWAS maps (voxel value > 10), and those containing voxels with the top 3 highest activation frequencies were considered significant. Significant hemodynamic responses using conventional event-related (ER) analysis and SSWAS maps were compared with the resection areas and surgical outcomes at 1 year after surgery. RESULTS: Using ER analysis, 4 (36%) of 11 patients had significant hemodynamic responses. One of 4 patients had significant hemodynamic responses in the resection area and good surgical outcome. Using SSWAS maps, 10 (91%) of 11 patients had significant hemodynamic responses. Six of 10 patients had significant hemodynamic responses in the resection area, and 5 of the 6 patients achieved good surgical outcomes. The remaining 4 patients had significant hemodynamic responses distant from the resection area, and only 1 of the 4 patients achieved good surgical outcomes. The sensitivity, specificity, positive predictive value, and negative predictive value of SSWAS maps were 83.3%, 75.0%, 83.3%, and 75.0%, respectively. CONCLUSIONS: This study demonstrated the clinical utility of SSWAS maps for presurgical evaluation of pharmacoresistant focal epilepsy. The findings indicated that subsecond EEG-fMRI analysis may help surgeons choose the resection areas that could lead to good surgical outcomes.

DOI： 10.3171/2020.1.JNS192567

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Williams syndrome (WS) is a genetically based neurodevelopmental disorder characterized by intellectual disability and impaired visuospatial recognition. The aim of this study was to analyze the gait characteristics of WS children with impaired visuospatial recognition using a three-dimensional gait analysis (3DGA) to clarify the gait adaptation needed to compensate for it. 3DGA was performed in 8 WS children with impaired visuospatial recognition (mean age, 11.8 years) and 9 age-, sex-, height-, and weight-matched controls. Clinical data, fundamental motor tests, and gait variables while walking on a flat surface and walking up a mat were compared between the two groups, and the correlations between variables were analyzed in the WS children. WS children showed impairment of balance function without muscle weakness. In walking on a flat surface, the WS group showed reduced walking speed, short step length, increased variability of step length, increased knee flexion throughout the stance phase, increased horizontal pelvic range of motion (ROM), and a low Gait Deviation Index and a high Gait Profile Score, which are indices of gait quality. In walking up a mat, the WS group showed further reduced walking speed and decreased sagittal hip flexion and ankle dorsiflexion ROM in the swing phase. Impaired balance function was significantly correlated with increased variability of step length and decreased sagittal ankle dorsiflexion ROM in the swing phase. The detailed gait pattern of WS children with impaired visuospatial recognition was presented. These findings show that impaired visuospatial recognition and balance function contribute to gait adaptation.

DOI： 10.1007/s00221-020-05946-0

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Authorship：Lead author,　Corresponding author   Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1111/ped.13793

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

OBJECTIVE: Epileptic spikes are associated with rapidly changing brain activation involving the epileptic foci and other brain regions in the "epileptic network". We aim to resolve these activation changes using simultaneous electroencephalography (EEG) and functional magnetic resonance imaging (fMRI) recordings. METHODS: Simultaneous EEG-fMRI recordings from 9 patients with epilepsy were used in the analysis. Our method employed the whole scalp EEG data to generate regressors for the analysis of fMRI data using the general linear model. RESULTS: We were able to resolve, with milliseconds temporal resolution, changes in activation patterns involving suspected epileptic foci and other brain regions in the epileptic network during spike and slow wave. Using summary maps (called SSWAS maps) which show the activation frequency of voxels, we found that suspected epileptic foci tend to be significantly active during this interval. SSWAS maps also enabled the detection of the epileptic foci in 4 of 5 patients where the conventional event-timing-based analysis failed to identify. CONCLUSION: These findings demonstrated the efficacy of the method and the potential application of SSWAS maps to identify epileptic foci. SIGNIFICANCE: The method could help resolve activation changes during epileptic spike and could provide insights into the underlying pathophysiology of these changes.

DOI： 10.1016/j.clinph.2017.11.018

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Authorship：Lead author,　Corresponding author   Language：English   Publishing type：Research paper (scientific journal)  

OBJECTIVE: The aim of this study was to clarify the characteristics of paroxysmal nonepileptic events (PNEs) suspected as being epileptic seizures by families of children with epilepsy. METHODS: The video-EEG (vEEG) recordings of habitual paroxysmal events in children with epilepsy at Nagoya University Hospital between October 2006 and January 2016 were reviewed. Based on the doctor's suspicion before the vEEG, the PNEs were divided into two groups that included PNEs suspected as epileptic seizures and PNEs suspected as PNEs. PNEs in the former group were classified based on the suspected seizure type. RESULTS: Of 886 habitual paroxysmal events, vEEG confirmed that 83 events (68 children) were PNEs. The median age of the 68 children was 3.2 years. Concurrent epilepsies included focal epilepsies (n=33), infantile spasms (n=16), and other types (n=19). The most common types of PNEs were sleep myoclonus (n=11), followed by stereotypies (n=9), awake myoclonus (n=8), paroxysmal ocular deviations (PODs, n=8), and tonic posturing (n=8). Even after direct observation or video viewing, the doctors suspected epileptic seizures in all three of the PODs and two of the tonic posturing children. Before the vEEG, however, the accurate visual information led to the speculation that the four psychogenic and two sleep myoclonus events were all PNEs. Myoclonus, stereotypies, and head drops were often misdiagnosed as epileptic spasms, while PODs and tonic posturing were often misdiagnosed as focal seizures with motor components. Additionally, staring and motion arrest during a drowsy state were often misdiagnosed as focal dyscognitive seizures. Seven of eight patients with PODs had epileptic spasms that were concurrent with epileptic seizures. A diffuse cerebral lesion or reduced visual acuity was seen in seven patients with PODs. CONCLUSION: We re-emphasize that vEEG is essential for accurate diagnosis and provides evidence for listing POD in the differential diagnosis of oculomotor paroxysmal events.

DOI： 10.1016/j.eplepsyres.2017.02.009

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OBJECTIVE: The aim of this study was to clarify characteristics of post-encephalopathic epilepsy (PEE) in children after acute encephalopathy with biphasic seizures and late reduced diffusion (AESD), paying particular attention to precise diagnosis of seizure types. METHODS: Among 262 children with acute encephalopathy/encephalitis registered in a database of the Tokai Pediatric Neurology Society between 2005 and 2012, 44 were diagnosed with AESD according to the clinical course and magnetic resonance imaging (MRI) findings and were included in this study. Medical records were reviewed to investigate clinical data, MRI findings, neurologic outcomes, and presence or absence of PEE. Seizure types of PEE were determined by both clinical observation by pediatric neurologists and ictal video-electroencephalography (EEG) recordings. RESULTS: Of the 44 patients after AESD, 10 (23%) had PEE. The period between the onset of encephalopathy and PEE ranged from 2 to 39 months (median 8.5 months). Cognitive impairment was more severe in patients with PEE than in those without. Biphasic seizures and status epilepticus during the acute phase of encephalopathy did not influence the risk of PEE. The most common seizure type of PEE on clinical observation was focal seizures (n = 5), followed by epileptic spasms (n = 4), myoclonic seizures (n = 3), and tonic seizures (n = 2). In six patients with PEE, seizures were induced by sudden unexpected sounds. Seizure types confirmed by ictal video-EEG recordings were epileptic spasms and focal seizures with frontal onset, and all focal seizures were startle seizures induced by sudden acoustic stimulation. Intractable daily seizures remain in six patients with PEE. SIGNIFICANCE: We demonstrate seizure characteristics of PEE in children after AESD. Epileptic spasms and startle focal seizures are common seizure types. The specific seizure types may be determined by the pattern of diffuse subcortical white matter injury in AESD and age-dependent reorganization of the brain network.

DOI： 10.1111/epi.13068

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Publishing type：Research paper (scientific journal)   Publisher：Elsevier BV  

DOI： 10.1016/j.braindev.2025.104348

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Publishing type：Research paper (scientific journal)   Publisher：Informa UK Limited  

DOI： 10.1080/21679169.2024.2356832

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STUDY OBJECTIVES: The brains of preterm infants exhibit altered functional connectivity (FC) networks, but the potential variation in sleep states and the impact of breathing patterns on FC networks are unclear. This study explores the evolution of resting-state FC from preterm to term, focusing on breathing patterns and distinguishing between active sleep (AS) and quiet sleep (QS). METHODS: We recruited 63 preterm infants and 44 healthy-term infants and performed simultaneous electroencephalography and functional near-infrared spectroscopy. FC was calculated using oxy- and deoxyhemoglobin signals across eight channels. First, FC was compared between periodic breathing (PB) and non-PB segments. Then sleep state-dependent FC development was explored. FC was compared between AS and QS segments and between preterm infants at term and term-born infants in each sleep state. Finally, associations between FC at term, clinical characteristics, and neurodevelopmental outcomes in late infancy were assessed in preterm infants. RESULTS: In total, 148 records from preterm infants and 44 from term-born infants were analyzed. PB inflated FC values. After excluding PB segments, FC was found to be elevated during AS compared to QS, particularly in connections involving occipital regions. Preterm infants had significantly higher FC in both sleep states compared to term-born infants. Furthermore, stronger FC in specific connections during AS at term was associated with unfavorable neurodevelopment in preterm infants. CONCLUSIONS: Sleep states play a critical role in FC development and preterm infants show observable changes in FC.

DOI： 10.1093/sleep/zsae225

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BACKGROUND: Quantitative EEG is frequently used to monitor children affected by acute encephalopathy (AE), with the expectation of providing comprehensive insights into continuous EEG monitoring. However, the potential of quantitative EEG for estimating outcomes in this context remains unclear. We sought reliable prognostic markers within the color density spectral array (CDSA) of the continuous EEG for AE-affected children undergoing therapeutic hypothermia (TH). METHODS: This retrospective study analyzed CDSA data from eight scalp electrodes of 15 AE-affected children undergoing TH. Two CDSA features were investigated-high-frequency lines (HFLs) and periodic elevation in the low frequency band (PLFB)-along with the corresponding EEG characteristics. The inter-rater reliability for CDSA was assessed by four pediatric neurologists. Outcomes were grouped into either no/mild or severe decline in motor and cognitive functions, then compared with CDSA features. RESULTS: The median EEG recording time was 114 (81-151) h per child. While at least 41 % of HFLs corresponded to typical sleep spindles, 94 % of PLFB aligned with cyclic changes in the amplitude of delta/theta waves on the raw EEG. Inter-rater reliability was higher for HFLs than for PLFB (kappa values: 0.69 vs. 0.46). HFLs were significantly more prevalent in children with no/mild decline than in children with severe decline (p = 0.017), whereas PLFB did not differ significantly (p = 0.33). CONCLUSIONS: This study provides preliminary evidence that reduced HFLs on CDSA predict unfavorable outcomes in AE-affected children undergoing TH. This suggests that maintaining high-frequency waves is critical for optimal brain function.

DOI： 10.1016/j.braindev.2024.08.002

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Publishing type：Research paper (scientific journal)   Publisher：MDPI AG  

Background/Objectives: During the COVID-19 pandemic, children in Japan were restricted from sports and outdoor activities. Regular physical activity is essential for healthy growth and development in children, with recommendations advocating for activities such as strength training. However, the long-term effects of the restrictions have not been fully investigated. This study aimed to evaluate the effectiveness of an “exercise class”, specifically a dance program, designed to improve motor function in elementary school children affected by the coronavirus disease 2019 (COVID-19) pandemic, which may have compromised their motor function. Methods: The dance program was developed by a sports science teacher, a professional dancer, and a physical therapist, and it was evaluated in a community-based participatory study. Trained dance instructors conducted one-hour dance sessions in a medical center, held once a week for two months from June to August 2023. A physical therapist and dance instructors led the elementary school children. Outcome measures included grip strength, lower extremity muscle strength, back muscle strength, dynamic balance function during movement, physical activity time, and body fat percentages. Descriptive statistics were used for analysis. Results: Twenty-four children aged 6–9 years participated in the exercise class over a two-month period. Improvement was observed in grip strength, lower extremity muscle strength, and dynamic balance function during movement. However, physical activity time, body fat percentages, and back muscle strength did not change. Conclusions: The results indicated a significant improvement in motor skills. Dance is an inexpensive program that elementary school children can enjoy while improving their motor skills.

DOI： 10.3390/children11091128

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BACKGROUND: Various factors contribute to the development of infection-related acute encephalopathy (AE) in children, such as infectious agents and chronic underlying disorders. We studied underlying disorders in children with AE to identify predisposing factors of AE. METHODS: We investigated underlying disorders or past histories in patients with two types of AE from the database in the Tokai area of Japan between 2009 and 2022: 204 patients with AE with reduced subcortical diffusion (AED) and 137 with clinically mild encephalopathy with a reversible splenial lesion (MERS). We compared them with 89 patients with acute disseminated encephalomyelitis (ADEM) to clarify the specific disorders in the two AE types. RESULTS: The prevalence of underlying disorders in AED (34%, 70 patients) was significantly higher than that in ADEM (12%, 11 patients) (P < 0.01). The prevalence of underlying disorders in MERS was 23% (32 patients). The underlying disorders included seizure disorders, premature birth, genetic/congenital disorders, and endocrine/renal diseases. In patients with seizure disorders in AED, five patients (18%) had Dravet syndrome and four (15%) had West syndrome, whereas none with MERS had these syndromes. Twenty-five (12%) of 204 patients with AED, three (2%) with MERS, and one (1%) with ADEM were preterm or low birth weight. CONCLUSIONS: The high prevalence of seizure disorders suggests that seizure susceptibility is an important predisposing factor in AED. Premature birth also has an impact on the development of AED. Caution is required regarding the development of AE in patients with chronic seizure disorders or premature birth.

DOI： 10.1016/j.pediatrneurol.2024.03.017

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BACKGROUND: GTP-cyclohydrolase 1-deficient dopa-responsive dystonia (GTPCH1-deficient DRD) typically presents in childhood with dystonic posture of the lower extremities, gait impairment, and a significant response to levodopa. We performed three-dimensional gait analysis (3DGA) to quantitatively assess the gait characteristics and changes associated with levodopa treatment in patients with GTPCH1-deficient DRD. METHODS: Three levodopa-treated patients with GTPCH1-deficient DRD underwent 3DGA twice, longitudinally. Changes were evaluated for cadence; gait speed; step length; gait deviation index; kinematic data of the pelvis, hip, knee, and ankle joints; and foot progression angle. RESULTS: Levodopa treatment increased the cadence and gait speed in one of three patients and increased the gait deviation index in two of three patients. The kinematic data for each joint exhibited different characteristics, with some improvement observed in each of the three patients. There was consistent marked improvement in the abnormal foot progression angle; one patient had excessive external rotation of one foot, another had excessive bilateral internal rotation, and the other had excessive internal rotation of one foot and excessive external rotation of the opposite foot, all of which improved. CONCLUSION: The 3DGA findings demonstrate that the gait pathology and recovery process in GTPCH1-deficient DRD vary from case to case. Changes in the foot progression angle and gait deviation index can enable the effects of treatment to be more easily evaluated.

DOI： 10.1016/j.pediatrneurol.2024.03.006

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Exercise improves physical health and positively impacts physical functions in children. Additionally, the habitual exercise established during childhood often continues into adulthood. Therefore, childhood is an important period for establishing healthy habits. Investigating the relationship between the presence or absence of daily exercise and physical functions may provide important information to confirm poor physical function in children with poor exercise habits. Therefore, this study examined the relationship between exercise habits as defined by the Japanese Ministry of Health, Labor, and Welfare and physical function in children aged 9-12 years. Approximately 239 children were included in this study. Participants answered a questionnaire about their exercise habits. We evaluated the skeletal muscle mass index, grip strength, standing broad jump, one-leg standing time, and gait deviation index. Logistic regression analyses were performed to assess exercise habit association with skeletal muscle mass index, physical function, and gait deviation index after adjusting for sex. Of the 239 children, 75.5% (n = 178) had exercise habits. A significant association was noted between exercise habits and skeletal muscle mass index and standing broad jump (skeletal muscle mass index: OR, 1.84; 95% CI, 1.01-3.36 and p<0.05; standing long jump: OR, 1.02; 95% CI, 1.00-1.04 and p<0.05). In conclusion, exercise regardless of exercise level for at least 30 min per day, 2 days per week, for at least 1 year is important for skeletal muscle mass and instantaneous lower limb muscular strength development in school-aged children.

DOI： 10.18999/nagjms.86.1.24

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Delta brushes are spontaneous neural activities observed in preterm electroencephalograms (EEGs) and are thought to reflect the activities of subplate neurons in the developing brain. We investigated the haemodynamic responses associated with delta brushes in five preterm infants at two time points (at 33 or 34, and 36 weeks of postmenstrual age), using simultaneous EEG-functional near-infrared spectroscopy (NIRS). An automated detection algorithm was developed to identify the brush components of delta brushes in the bipolar EEG envelope; we placed eight EEG electrodes. An eight-channel NIRS device was placed around the head of each infant to measure changes in oxy- and deoxy-haemoglobin (Hb) concentrations. Haemodynamic grand averages were calculated for local brushes in each NIRS channel. We classified the responses into five patterns based on changes in oxy- and deoxy-Hb signals (positive in-phase/anti-phase, negative in-phase/anti-phase, and unclassified) and evaluated the relationship between the locations of NIRS measurements and those of brushes, as well as the haemodynamic response patterns and infant age at the time of recording. In all the 10 recordings, we found that positive responses (oxy-Hb increases) predominated, not only in the corresponding areas but also in remote areas. Particularly, notable responses were observed in the bilateral temporal areas. Among the positive responses, the proportion exhibiting an anti-phase pattern was 12% at 33 to 34 weeks of postmenstrual age and 54% at 36 weeks of postmenstrual age. Our unexpected finding of remarkable temporal responses to localised neuronal activity supports the hypothesis that the insula is the most strongly interconnected hub in the developing brain.

DOI： 10.1007/978-3-031-67458-7_16

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Language：Japanese   Publisher：(NPO)愛知県理学療法学会  

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Low-back pain is common among school-aged children. Decreased trunk flexibility in childhood influences low-back pain in adulthood. Previous studies examining the association between low-back pain and trunk flexibility in children are insufficient. Examining this association among elementary school children may help to better understand trunk flexibility in children with low-back pain and to modify the management of inflexibility. Therefore, this study aimed to identify the prevalence of low-back pain and its relationship with physical function among elementary school students. School-aged children aged 6-12 years were recruited in Japan between May 2018 and March 2023. Fingertip-to-floor distance, back muscle strength, pelvic tilt angle during gait, and the visual analog scale for low-back pain were measured. In addition, factors independently related to low-back pain were determined through logistic regression analysis. Low-back pain was reported in 9.6% of the 394 participants (boys, 191; girls, 203). All children with low-back pain presented with back pain when they moved; however, the pain was non-specific. Logistic regression analysis showed that the fingertip-to-floor distance was an independent risk factor for low-back pain (odds ratio, 0.921; p = 0.007). The odds ratios calculated in the logistic regression analysis confirmed that low-back pain frequency increased as the fingertip-to-floor distance decreased. The risk of low-back pain was associated with inflexibility, regardless of sex and muscle strength. These findings suggest that children with low-back pain must increase their trunk and lower extremity flexibility.

DOI： 10.1371/journal.pone.0293408

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OBJECTIVE: To compare walking efficiency, and associated physical function, between children, with and without developmental coordination disorder trait. DESIGN: Cross-sectional study. SETTING: The study was conducted in a Medical and Rehabilitation Center for Developmental Disabilities in Okazaki, Japan. SUBJECTS: The study included 286 children, 6-12 years of age, with and without developmental coordination disorder trait. Developmental coordination disorder trait was defined by a score below the -1 standard deviation on the Japanese version of the Developmental Coordination Disorder Questionnaire. Based on this criterion, 45 children (28 boys) were classified into the developmental coordination disorder trait group and the other 241 (115 boys) in the control group. MEASURES: The following physical function outcomes were measured: single leg stance test; step length variability; and walking efficiency, measured using the 2-min walk test. Logistic regression analysis was used to determine the association between developmental coordination disorder trait and lower walking efficiency and physical function. RESULTS: Compared to the control group, the developmental coordination disorder group showed lower walking efficiency (P  =  0.026), higher step length variability (P  =  0.047), and shorter single leg stance test (P  =  0.001). Developmental coordination disorder was independently associated with walking efficiency (odds ratio, 1.03; 95% confidence interval [CI], 1.001-1.061; P  =  0.041) and single leg stance test (odds ratio, 0.99; 95% CI, 0.978-0.996; P  =  0.006), but not to step length variability, age, and sex. CONCLUSIONS: Lower walking efficiency increases energy expenditure in children with developmental coordination disorder trait. Interventions to improve walking efficiency could improve the physical function and participation of these children.

DOI： 10.1177/02692155221150385

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Language：Japanese   Publisher：コ・メディカル形態機能学会  

児童において、靴底の摩耗部位における足角の違いが明らかになれば、足関節捻挫や足部外傷のリハビリテーションに役立つ情報となると考え、本研究では児童を対象とした靴底の摩耗部位における歩行時の足角の違いを検討することを目的とした。児童運動器健診に参加した普通学級に通う小学校1年生から6年生の児童172名を対象とした。それらを踵部の靴底の摩耗部位の評価によって、正常群、内側群、外側群の3群に分け、群間比較を行った。評価指標として、三次元動作解析装置を用いて算出した時空間パラメータ、初期接地時と立脚期の足角の値を算出した。また、整形外科医による視診及び問診、触診を基に足部異常の有無を指標に加えた。靴底の内側が摩耗している児は、初期接地時、立脚期の足角が内旋偏位しており、足部異常を有している割合が高かった。このことから、靴底の摩耗の評価に加えて、三次元歩行分析による足角の評価も行うことの重要性が示唆された。(著者抄録)

DOI： 10.11172/keitaikinou.21.32

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The response of muscle oxygen saturation, which is an index for the energy metabolism of muscles during walking in children, and its relationship to the physiological cost index, which indicates walking efficiency, are unknown. This study aimed to evaluate muscle oxygen saturation in lower extremity muscles during walking in children, its changes with age, and the relationship between the physiological cost index. The oxygen saturation was measured by the amount of change during a two-minute walk, and the physiological cost index was calculated from the change in heart rate before and after exercise and walking speed. Results were compared for each muscle, and the correlation between the two was examined. Changes in muscle oxygen saturation were greater in the lower leg muscles, significantly greater in the tibialis anterior at six to seven years, and in the gastrocnemius medial head at eight to ten years. The physiological cost index was significantly correlated with changes in muscle oxygen saturation in the tibialis anterior (r = 0.44, p &lt; 0.001). The lower leg muscles were metabolically active in children's gait, and their response varied with age. Moreover, the muscle oxygenation dynamics of the tibialis anterior may influence walking efficiency.

DOI： 10.3390/healthcare11020221

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Children's exercise habits have changed during the COVID-19 pandemic. This study aimed to examine the physical function and physical activity of preadolescent children before and during the COVID-19 pandemic. This cross-sectional study compared time spent in moderate-to-vigorous physical activity (MVPA), grip strength, single-leg standing time, and two-step tests of healthy children aged 10 to 12 years, enrolled from January 2018 to January 2020 (pre-COVID-19 group, n = 177) and from January 2021 to September 2022 (during-COVID-19 group, n = 69). The during-COVID-19 group had weaker grip strength (median: 14.4 vs. 15.8 kg; p = 0.012), worse performance on the two-step test (mean: 1.56 vs. 1.60; p = 0.013), and less MVPA (median: 4 vs. 7 h per week; p = 0.004). Logistic regression showed that the during-COVID-19 group was significantly related to weaker grip strength (odds ratio: 0.904, 95% CI: 0.829-0.986; p = 0.022) and worse performance in the two-step test (odds ratio: 0.976, 95% CI: 0.955-0.997; p = 0.028). The COVID-19 pandemic decreased exercise opportunities for preadolescent children, which may have had a negative impact on muscle strength and balance. It is essential to increase the amount of MVPA among preadolescent children.

DOI： 10.3390/healthcare10122553

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One major impact of the COVID-19 pandemic on children's lifestyles is the lack of exercise owing to activity restrictions. However, information regarding the way in which physical functions among children decline under these circumstances remains scarce. In this study, we compared the physical functions and life habits among children before and during the COVID-19 pandemic in Japan. The participants involved 40 children aged between 9-15 years (23 girls and 17 boys) who were examined medically both before and during the pandemic. The compared variables included muscle strength, static and dynamic balance functions, gait speed, body fat percentage, screen and sleep times, quality of life, and physical activity time. During the pandemic, compared to before the pandemic, children had lower levels of dynamic balance functions (p = 0.039), increased body fat percentages (p &lt; 0.0001), longer screen time per day (p = 0.002), and shorter sleep time per day (p &lt; 0.0001). Between the two periods, there were no significant differences in muscle strength, static balance functions, gait speed, quality of life, and physical activity time. The activity restrictions imposed as a result of the COVID-19 pandemic negatively affect dynamic balance functions, body-fat levels, and life habits among children.

DOI： 10.3390/ijerph191811513

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We aimed to develop gait standards for gait parameters in school-aged Japanese children and assess age-related differences in gait patterns and parameters. Children aged 6-12 years (n = 424) were recruited from two elementary schools. An instrumented three-dimensional gait analysis system was used to record each child's gait kinematics, kinetics, and spatiotemporal parameters. Participants were subdivided into three age groups (Group A, 6-8 years; Group B, 9-10 years; and Group C, 11-12 years). LMS Chartmaker, version 2.54, was used to create a developmental chart for the gait pattern. The non-normalized step and stride lengths were significantly longer, and the cadence was lower in older children; however, the opposite outcome occurred when analyzing normalized data. Ankle moment differed significantly by age, and the maximum ankle moment was higher in older children than that in younger children. Furthermore, the hip and knee flexion angles during gait and the normalized spatiotemporal parameters of Japanese children aged 6-12 years differed by age and from those of children from other countries. The centile chart of the gait pattern is a useful tool for clinicians to assess developmental changes in the gait pattern and detect gait abnormalities in children.

DOI： 10.1038/s41598-022-11906-1

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BACKGROUND: Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is a common type of acute encephalopathy in Japan; the condition is clinically characterized by prolonged seizures as the initial neurological symptom, followed by late seizures 4-6 days later. It is difficult to differentiate AESD from prolonged febrile seizures (PFSs). Here, we explored the use of electroencephalography to differentiate AESD from PFSs. METHODS: We studied the electroencephalograms (EEGs) of children <6 years of age diagnosed with AESD or PFSs; all EEGs were recorded within 48 h of seizure onset (i.e., before the late seizures of AESD). Two pediatric neurologists evaluated all EEGs, focusing on the basic rhythm, slowing during wakefulness/arousal by stimuli, spindles, fast waves, and slowing during sleep. RESULTS: The EEGs of 14 children with AESD and 31 children with PFSs were evaluated. Spindles were more commonly reduced or absent in children with AESD than in those with PFSs (71% vs. 31%, p = 0.021). Fast waves were also more commonly reduced or absent in children with AESD (21% vs. 0%, p = 0.030). The rates of all types of slowing did not differ between children with AESD and those with PFSs, but continuous or frequent slowing during sleep was more common in the former (50% vs. 17%, p = 0.035). CONCLUSIONS: EEG findings may usefully differentiate AESD from PFSs. Reduced or absent spindles/fast waves and continuous or frequent slowing during sleep are suggestive of AESD in children with prolonged seizures associated with fever.

DOI： 10.1016/j.braindev.2021.11.003

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Children with behavioral problems have a high risk of impaired motor performance. However, the characteristics of balance functions and their associations with behavioral traits are unclear in this population. This study aimed to evaluate balance functions and their relationships with the degree of behavioral problems in school-aged children. A total of 209 children, aged 6-10 years, were divided into two groups, those with and those without behavioral problems, using the Strengths and Difficulties Questionnaire (SDQ). Physical assessments included the one-leg standing test (OLST), the two-step test, and the five-times-sit-to-stand test. We compared the data between groups and assessed for correlations in terms of total difficulties and the SDQ subscale scores. Children with behavioral problems showed significantly reduced the OLST results (p < 0.001) and the two-step test results (p = 0.008). The five-times-sit-to-stand test results did not show significant differences between groups. The OLST results were significantly correlated with emotional symptoms (r = -0.22, p < 0.001), hyperactivity/inattention (r = -0.29, p < 0.001), peer relationship problems (r = -0.22, p < 0.001), and total difficulties (r = -0.32, p < 0.001). Meanwhile, the two-step test results showed no significant correlation with the SDQ scores. Children with behavioral problems have poor balance function, thereby increasing the risk for instability. This suggests that the balance function of children with behavioral problems needs to be considered.

DOI： 10.3390/brainsci12010117

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Publishing type：Research paper (scientific journal)   Publisher：MDPI AG  

The relationship between moderate-to-vigorous physical activity (MVPA) performance time and body muscle mass and stiffness index in pre-puberty school-aged girls has not been fully elucidated. The effect of sexual maturity on bone mass is more pronounced in girls. This study aimed to clarify the relationship between MVPA performance time and the above-mentioned factors. This was a prospective, population-based cohort study of 111 girls aged 7–10 years. Data were collected via medical examination, clinical measurements, and questionnaires. Spearman’s ρ analysis was used to determine the association between skeletal muscle mass index (SMI) and stiffness index, muscle strength, and MVPA performance time. Participants who met the recommended MVPA level accounted for only 24.3% (n = 27) of all participating girls (n = 111). The following factors were significantly positively correlated with MVPA level at spearman’s ρ analysis: SMI (r = 0.303, p = 0.001), stiffness index (r = 0.229, p = 0.015), grip strength (r = 0.283, p = 0.003), back muscle strength (r = 0.197, p = 0.038), and standing long jump distance (r = 0.288, p = 0.002). Multiple regression analysis’s results revealed that SMI (β = 0.237; p = 0.024) was associated with MVPA performance time. These results can help school-aged girls to pay adequate attention to having healthy physical activity habits to prevent the decline of skeletal muscle mass, stiffness index, and body muscle strength.

DOI： 10.3390/healthcare10020197

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The relationship of locomotive syndrome with other physical characteristics and lifestyle habits in children has not been fully elucidated. The aim of this study was to assess the prevalence of children's locomotive syndrome, and to determine its relationship with the above-mentioned factors. This was a cross-sectional study of 285 elementary school children who volunteered to participate in a medical checkup for physical function. Data was collected via medical examination, clinical measurements, and questionnaires. A multivariable logistic regression model was used to determine the relationship (odds ratios; ORs) of participants' characteristics, physical functions, and other outcomes determined by questionnaire on locomotive syndrome. The following factors were related to locomotive syndrome: older age (OR = 1.421, 95% confidence interval [CI] [1.039, 1.945]), male sex (OR = 4.011, 95% CI [2.189, 7.347]), and more time spent watching television per day (OR = 1.281, 95% CI [1.001, 1.640]). These results may assist in the encouragement of children to perform appropriate physical activities and avoid unhealthy lifestyle habits, reducing the occurrence of locomotive syndrome.

DOI： 10.3390/healthcare9111595

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DOI： 10.3390/ijerph18189620

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Language：Japanese   Publisher：(一社)日本体力医学会  

DOI： 10.7600/jspfsm.70.193

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Other Link： https://search.jamas.or.jp/index.php?module=Default&action=Link&pub_year=2021&ichushi_jid=J00797&link_issn=&doc_id=20210604410001&doc_link_id=10.7600%2Fjspfsm.70.193&url=https%3A%2F%2Fdoi.org%2F10.7600%2Fjspfsm.70.193&type=J-STAGE&icon=https%3A%2F%2Fjk04.jamas.or.jp%2Ficon%2F00007_3.gif

Language：Japanese   Publisher：(一社)日本臨床神経生理学会  

脳波を患者の検査として用いる場合に,臨床研究と臨床検査の境界は明確ではない場合がある。汎用性を持って施行可能なのか,診断や治療方針の決定などに用いられるのかが研究か診療かの指標になるだろう。本編では,小児の脳波検査において研究からclinical toolとして確立されてきた,または確立していく可能性のある手法について解説する。amplitude-integrated EEG(aEEG)は新生児領域で広く発作検出の臨床目的で使われており,小児のICU脳波モニタリングでも用いられている。高周波律動も,デジタル脳波の普及に伴い用いられるようになった手法で,頭皮脳波でも評価されている。脳波を他のモダリティと組み合わせることも脳波の新しい活用法である。脳波-機能的MRI同時記録は,脳波活動に伴う血流変化をMRIで可視化し,てんかん焦点,ネットワークの解明に用いられている。(著者抄録)

DOI： 10.11422/jscn.49.141

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Other Link： https://search.jamas.or.jp/index.php?module=Default&action=Link&pub_year=2021&ichushi_jid=J03414&link_issn=&doc_id=20210607260004&doc_link_id=%2Fff2rinsy%2F2021%2F004903%2F005%2F0141-0144%26dl%3D0&url=https%3A%2F%2Fwww.medicalonline.jp%2Fjamas.php%3FGoodsID%3D%2Fff2rinsy%2F2021%2F004903%2F005%2F0141-0144%26dl%3D0&type=MedicalOnline&icon=https%3A%2F%2Fjk04.jamas.or.jp%2Ficon%2F00004_2.gif

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Regular physical activity is an important component of physical health of children and has been associated with increasing skeletal muscle mass and muscle strength. Children with low levels of physical activity may experience health problems, such as loss of muscle mass, later in life. Thus, it may be valuable to identify declining physical function in children who do not perform the recommended amount of physical activity. Therefore, we aimed to evaluate the relationship between the amount of physical activity performed for ≥60 min per day for ≥5 days per week and the skeletal muscle mass index and physical function in young children. In total, 340 typically developing children aged 6-12 years (175 girls; average age, 9.5±1.9 years) were included in this cross-sectional study. We evaluated the proportion of children performing the recommended minimum of 60 min of daily moderate-to-vigorous physical activity at least 5 days per week. The skeletal muscle mass and Gait Deviation Index scores, gait speed, grip strength, Five Times Sit-to-Stand test results, Timed Up-and-Go test results, one-leg standing time, and gait efficiency were evaluated. Multiple logistic regression analyses were performed to assess the association of moderate-to-vigorous physical activity with the skeletal muscle mass index, percent body fat, and physical function, after controlling for confounding factors (age and sex). A logistic regression analysis revealed that the skeletal muscle mass index was independently associated with moderate-to-vigorous physical activity (odds ratio, 2.34; 95% confidence interval, 1.17-4.71; P = 0.017). Performance of moderate-to-vigorous physical activity for ≥5 days per week for ≥60 min per day was associated with the skeletal muscle mass index score of Japanese children. Our findings highlighted the importance of performing moderate-to-vigorous physical activity for the development of skeletal muscle mass in children.

DOI： 10.1371/journal.pone.0251025

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BACKGROUND AND PURPOSE: Bottom shuffling is a locomotion strategy that precedes independent walking in some infants. Shuffling babies are generally considered to have favorable outcomes. The aim of the present study was to reveal clinical features and neurodevelopmental outcomes of shuffling babies who visited a child developmental center. METHODS: We studied 48 shuffling babies who visited Toyota Municipal Child Development Center from April 2007 to March 2015. We excluded patients with cerebral palsy, Down syndrome, or congenital disorders. In 2018, we retrospectively reviewed the clinical charts of the enrolled children. We investigated family history, neurological findings, and the developmental outcome during the follow-up period. RESULTS: During the follow-up period, 20 children (42%) were diagnosed with ASD. Gross motor development in infancy was not different between infants with and without ASD. The rate of poor eye contact at the first visit and a delay in the first word speech were significantly higher in infants with ASD than in infants without ASD. A family history of bottom shuffling was significantly less frequent in infants with ASD (10%) than in those without (39%). CONCLUSION: Some of bottom shufflers may represent ASD during follow-up. Paying attention to social and cognitive functions in shuffling babies is important.

DOI： 10.1016/j.braindev.2020.08.007

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PURPOSE: To establish an objective method of [18F]-fluorodeoxyglucose positron emission tomography (FDG-PET) that can assist in the diagnosis of glucose transporter 1 deficiency syndrome (GLUT1-DS). METHODS: FDG-PET was performed in 8 patients with a mean age of 12.5 years (range, 2-22 years) with GLUT1-DS. Their PET findings were compared with those of 45 controls with a mean age of 11.2 years (range, 2-21 years) by statistical parametric mapping (SPM12, Welcome Neurological Institute). The controls had epilepsy of unknown etiology and normal MRI findings. The age-adjusted ratios of mean radioactivities in regions of interest (ROIs) of bilateral lenticular nuclei, thalami, and the whole cerebral cortex were also measured. The sensitivities and specificities of the ratios for the differential diagnosis of GLUT1-DS were also determined. RESULTS: SPM showed significantly decreased uptake in bilateral thalami and increased uptake in bilateral lenticular nuclei in patients with GLUT1-DS. There were no areas in the cerebral cortex with significant differences between patients and controls. On ROI analysis, by setting the cut-off value of the age-adjusted lenticular nuclei/thalami radioactivity ratio to 1.54, patients with GLUT1-DS were differentiated from controls with sensitivity of 1.00 and specificity of 0.98. CONCLUSION: The age-adjusted lenticular nuclei/thalami radioactivity ratio on PET can distinguish patients with GLUT1-DS from patients with epilepsy of unknown etiology with high sensitivity and specificity. It is important to pay attention to the metabolism of the lenticular nuclei and thalami on PET for the diagnosis of GLUT1-DS.

DOI： 10.1016/j.braindev.2020.07.001

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BACKGROUND: Advances in hematopoietic stem cell transplantation have improved the survival rate of malignant diseases and congenital immunodeficiencies. It has become important to assess long-term complications in survivors. To assess neurological abnormalities in children treated by transplantation, diffusion tensor imaging was performed. METHODS: Forty children who underwent head diffusion tensor imaging before and after their first transplantation were enrolled. Patients with brain lesions on conventional MRI were excluded. Fractional anisotropy and mean diffusivity were compared between patients and 28 control subjects using tract-based spatial statistics. The Strengths and Difficulties Questionnaire was administered as a behavioral evaluation after transplantation, and diffusion tensor images of patients with and without behavioral abnormalities were compared. RESULTS: The age of patients and controls was 0 to 19 years and 0 to 16 years, respectively. The date of diffusion tensor imaging was 10 to 57 days before and 40 to 153 days after transplantation. Tract-based spatial statistics showed fractional anisotropy reduction in widespread white matter in patients before and after transplantation. Mean diffusivity was high before transplantation and normalized after transplantation. Analysis comparing before and after hematopoietic stem cell transplantation shows no difference in fractional anisotropy and a higher mean diffusivity before hematopoietic stem cell transplantation. In patients with behavioral abnormalities, low fractional anisotropy and high mean diffusivity remained after transplantation. CONCLUSIONS: Longitudinal diffusion tensor imaging showed white matter abnormalities in children without conventional MRI abnormalities, which were related to behavioral problems after transplantation. Diffusion tensor imaging is useful for behavioral assessment in children undergoing transplantation.

DOI： 10.1016/j.pediatrneurol.2020.06.008

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OBJECTIVE: To investigate clinical risk factors for acute magnetic resonance imaging (MRI) abnormalities in patients with benign convulsions with mild gastroenteritis or benign infantile epilepsy. STUDY DESIGN: We investigated clinical and diffusion-weighted imaging findings in 32 patients with benign convulsions with mild gastroenteritis and 22 patients with benign infantile epilepsy who underwent MRI within seven days of seizure onset between 2010 and 2015. RESULTS: Diffusion-weighted imaging showed signal hyperintensity in the splenium of the corpus callosum in seven patients with benign convulsions with mild gastroenteritis, but no abnormalities in patients with benign infantile epilepsy. Patients with benign convulsions with mild gastroenteritis with splenial lesions showed a higher rate of rotavirus detection from feces (P = 0.006), higher serum level of C-reactive protein (P = 0.04), and shorter interval between seizure onset and MRI (P = 0.002) than patients with benign convulsions with mild gastroenteritis without splenial lesions. Multivariate analysis revealed rotavirus infection as a significant risk factor for splenial lesions on diffusion-weighted imaging in patients with benign convulsions with mild gastroenteritis (P = 0.02). CONCLUSIONS: Splenial lesions are often seen during acute period in patients with benign convulsions with mild gastroenteritis. Rotavirus infection is a risk factor for splenial lesions in patients with benign convulsions with mild gastroenteritis, suggesting the role of rotavirus to cause edema in the corpus callosum. From our observations, benign convulsions with mild gastroenteritis with a splenial lesion on diffusion-weighted imaging suggests good outcomes, and extensive evaluation of these patients may be unnecessary.

DOI： 10.1016/j.pediatrneurol.2019.05.002

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OBJECTIVE: The objective of this study was to describe a novel amplitude-integrated electroencephalography (aEEG) pattern in infants with hypoxic-ischemic encephalopathy (HIE) and to assess the clinical significance. METHODS: The aEEG traces of infants with HIE who were treated with therapeutic hypothermia (TH) from 2012 to 2017 were analyzed. A pseudo-sawtooth (PST) pattern was defined as a periodic increase of the upper and/or lower margin of the trace on aEEG without showing seizure activities on conventional EEG (CEEG). RESULTS: Of the 46 infants, 6 (13%) had the PST pattern. The PST pattern appeared following a flat trace or a continuous low-voltage pattern and was followed by a burst-suppression pattern. On CEEG, the PST pattern consists of alternating cycles of low-voltage irregular activities and almost flat tracing. The PST pattern was associated with neuroimaging abnormalities and with various degrees of neurodevelopmental outcomes. Positive predictive values of the PST or worse pattern for adverse outcomes were high at 12 h after birth. CONCLUSION: A novel aEEG background pattern in infants with HIE was reported. The PST pattern likely indicates a suppressed background pattern and may be linked to unfavorable outcomes. Further multicenter validation study is needed to clarify its clinical significance.

DOI： 10.1038/s41390-019-0567-5

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The authors wish to make the following corrections to this paper [...].

DOI： 10.3390/jcm9020569

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This study examined the association between Gait Deviation Index (GDI) and the five-times-sit-to-stand test (FTSST) or gait speed results, which represent mobility and muscle strength of the lower extremities in ambulatory children with Gross Motor Function Classification System (GMFCS) level I and II spastic cerebral palsy. In this cross-sectional, observational study, three-dimensional gait analysis data were obtained during gait trials to evaluate the GDI in 35 children (age 5-16 years) with spastic palsy. Motor function was evaluated using FTSST and gait speed. Gross motor function was evaluated using GMFCS. Children with GMFCS level II spastic cerebral palsy demonstrated lower GDI (p < 0.001) and poorer FTSST (p = 0.031) than those with GMFCS level I spastic cerebral palsy. Correlation analysis showed that FTSST results were significantly correlated with GDI (r = -0.624; p < 0.001). Motor function may be important for the maintenance of gait quality in patients with GMFCS level I and II spastic cerebral palsy and should not be ignored. In conclusion, reduction in gait impairment may affect the values of FTSST and GDI in patients with spastic cerebral palsy who can ambulate without an assistive device.

DOI： 10.3390/jcm9010028

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BACKGROUND AND PURPOSE: Despite the development of neuroimaging, identification of focal cortical dysplasia remains challenging. The purpose of this study was to show the longitudinal changes of MR imaging and FDG-PET in patients with West syndrome and subtle focal cortical dysplasia. MATERIALS AND METHODS: Among 52 consecutive patients with West syndrome, 4 were diagnosed with subtle focal cortical dysplasia on 3T MR imaging. MR imaging and PET findings were evaluated longitudinally at onset and at 12 and 24 months of age. RESULTS: At the onset of West syndrome, MR imaging demonstrated focal signal abnormalities of the subcortical white matter in 2 patients. In the other 2 patients, focal subcortical high-intensity signals became visible on follow-up T2WI as myelination progressed. PET at onset showed focal cortical hypometabolism in 3 patients, with 1 of these patients also having focal hypermetabolism and 1 having normal findings. On PET at 24 months, hypometabolism persisted in 2 patients and disappeared in 1, and hypermetabolism disappeared in 1. In 1 patient with normal MR imaging and PET findings at onset, focal hyperintensity and hypometabolism first appeared at 24 months of age. The findings on MR imaging and PET in these patients evolved differently with brain maturation and the clinical course. CONCLUSIONS: Subtle focal cortical dysplasia can be undetectable on MR imaging at the onset of West syndrome and is not always accompanied by hypometabolism or hypermetabolism on PET. Longitudinal MR imaging and PET studies may be useful for detecting such lesions. Even in West syndrome with a congenital structural abnormality, PET findings evolve differently with brain maturation and the clinical condition.

DOI： 10.3174/ajnr.A5772

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[Purpose] This study aimed to identify a simple and useful muscle parameter for use with the Gait Deviation Index in assessment of ambulatory children with unilateral and bilateral spastic cerebral palsy. [Participants and Methods] Twenty-eight patients (aged 6 to 18 years; 16 females and 12 males) participated in this cross-sectional study. Outcome measurements included the Gait Deviation Index, grip strength, 5-repetition chair stand test, upper limb skeletal muscle mass index, and lower limb skeletal muscle mass index. [Results] By multiple regression analysis, significant independent correlations were observed between the Gait Deviation Index and 5-repetition chair stand test and the Gait Deviation Index and lower limb skeletal muscle mass index, but not between the Gait Deviation Index and grip strength or upper limb skeletal muscle mass index. [Conclusion] The Gait Deviation Index was correlated with lower limb muscle mass in children with spastic cerebral palsy. Determination of lower limb muscle mass may be useful gait evaluation.

DOI： 10.1589/jpts.30.1176

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OBJECTIVE: To clarify longitudinal changes in white matter microstructures from the onset of disease in patients with West syndrome (WS) of unknown etiology. METHODS: Diffusion tensor imaging (DTI) was prospectively performed at onset and at 12 and 24 months old in 17 children with WS of unknown etiology. DTI was analyzed using tract-based spatial statistics (TBSS) and tract-specific analysis (TSA) of 13 fiber tracts, and fractional anisotropy (FA) and mean diffusivity (MD) were compared with those of 42 age-matched controls. Correlations of FA and MD with developmental quotient (DQ) at age 24 months were analyzed. Multiple comparisons were adjusted for using the false discovery rate (q-value). RESULTS: TBSS analysis at onset showed higher FA and lower MD in the corpus callosum and brainstem in patients. TSA showed lower MD in bilateral uncinate fasciculi (UF) (right: q < 0.001; left: q = 0.03) at onset in patients. TBSS showed a negative correlation between FA at onset and DQ in the right frontal lobe, whereas FA at 24 months old exhibited a positive correlation with DQ in the diffuse white matter. MD for bilateral UF at 24 months old on TSA correlated positively with DQ (q = 0.04, both). SIGNIFICANCE: These findings may indicate the existence of cytotoxic edema in the immature white matter and dorsal brainstem at onset, and subsequent alterations in the diffuse white matter in WS of unknown etiology. Microstructural development in the UF might play important roles in cognitive development in WS.

DOI： 10.1111/epi.13988

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Language：English  

OBJECTIVE: Nonspecific manifestations and a varied distribution of brain lesions can delay the diagnosis of herpes simplex encephalitis (HSE) in neonates. The aim of this study was to report predominant brain lesions in neonatal HSE, and then to investigate the association between pattern of predominant brain lesions, clinical variables and neurodevelopmental outcome. STUDY DESIGN: A multicenter retrospective study was performed in neonates diagnosed with HSE between 2009 and 2014. Magnetic resonance (MR) images, including diffusion-weighted images, were obtained in the acute and chronic phase. RESULTS: Three predominant areas of brain injury could be defined based on characteristic MRI findings in 10 of the 13 infants (77%). The inferior frontal/temporal pole area was involved in five (38%) patients. The watershed distribution was present in six (46%) patients. Four (31%) infants involved the corticospinal tract area. No significant association was found between any predominant distribution of brain lesion pattern and sex, country, viral type or viral load. However, the corticospinal tract involvement was significantly associated with motor impairment (P=0.045). CONCLUSION: Three predominant areas of brain lesion could be recognized in neonatal HSE. Recognition of those areas can improve prediction of neurodevelopmental outcome.

DOI： 10.1038/jp.2017.114

PubMed

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Language：Japanese   Publisher：(公社)日本小児科学会  

1995年から16年間に出生し、豊田市こども発達センターを受診した豊田市在住の重症心身障害児39例について、診療録と療育記録から親の離婚の有無、患者の医療的背景、親・家族の社会的背景、離婚時の家庭状況を後方視的に調査した。親の離婚あり群は6例(15%)で一般の離婚率と比べて高くなかったが、身体的虐待・家族間暴力のある家庭で離婚リスクが高く、加害者はすべて父親であった。また、親の離婚に子どもの基礎疾患や生活行為としての医療ケアの内容、重症度の影響はみられなかったが、親の離婚あり群では睡眠障害のある子どもが多かった。重症心身障害児の家庭の破綻を事前に防ぐためには、療育施設と医療機関の連携を深め、情報を共有するシステムの構築が必要であり、父親の役割に注目した支援のあり方や子どもの睡眠障害への積極的な介入も重要と考えられた。

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Authorship：Lead author,　Corresponding author   Language：Japanese   Publisher：(公社)日本小児科学会  

重度肢体不自由児(重肢児)における急性期医療の現状を把握する目的で、豊田市に在住する2〜14歳の全小児を対象に、トヨタ記念病院および豊田厚生病院での診療状況を2009〜2013年度までの5年間にわたり調査し、大島分類で重肢児に該当する群(重肢群)と非該当群に分けて比較検討した。結果、各群の総数に占める救急外来受診者数の割合は全年度において重肢群のほうが有意に高く(リスク比=1.8〜3.3)、各群の総数に占める入院者数の割合も全年度で重肢群が有意に高かった(リスク比=12.2〜19.3)。経年的変化として、非該当群では気管支炎・肺炎の減少に伴い入院者の総数も減少しているのに対し、基礎疾患に伴う合併症での入院が多い重肢群では変化がみられなかった。これに伴い、延べ入院日数に占める非該当群の割合が減少して重肢群の割合が増加し、2013年度には重肢群の割合が7.0%に達していた。

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

PURPOSE: Although it has been reported that some antiepileptic drugs have inducing or inhibiting effects on lamotrigine (LTG) clearance, whether they have the same effects in Asian epilepsy patients as in those in other countries has not been clarified, especially in children. The aim of this study was to determine the effects of co-medications on LTG clearance in Japanese children with epilepsy. METHODS: A total of 342 routine serum concentration measurements of LTG in 102 Japanese epilepsy patients under 20years of age were reviewed. The dose-corrected concentration (DCC) of LTG was calculated as [concentration]/[dose/(body weight)], and the DCC of LTG was compared by co-medication. The difference in the DCC of LTG was compared between patients with and without valproic acid (VPA) and between those with and without drugs inducing glucuronic acid conjugation (phenytoin (PHT), carbamazepine (CBZ), and phenobarbital (PB)). RESULTS: The DCC of LTG was significantly higher in patients on VPA and significantly lower in patients on drugs inducing glucuronic acid conjugation than in patients on LTG monotherapy. The DCC of LTG was significantly higher in patients on CBZ than in patients on PHT or PB. There was no correlation between the DCC of LTG and the concentration of VPA or metabolic inducers within the therapeutic range. Other antiepileptic drugs including clobazam, clonazepam, zonisamide, and levetiracetam had little effect on LTG concentration. CONCLUSION: LTG concentration changes dramatically with concomitant antiepileptic drugs in Japanese children, as previously reported from other countries, and special attention is required. Although the dose of LTG should be adjusted when starting or discontinuing VPA or metabolic inducers, no adjustment is needed when changing the dose of VPA or metabolic inducers in the therapeutic range.

DOI： 10.1016/j.braindev.2016.03.004

PubMed

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Language：Japanese   Publisher：(株)東京医学社  

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Language：English  

BACKGROUND AND PURPOSE: West syndrome is an epileptic encephalopathy characterized by epileptic spasms, a specific pattern on electroencephalography of hypsarrhythmia, and developmental regression. Our aim was to assess white matter abnormalities in West syndrome of unknown etiology. We hypothesized that diffusion tensor imaging reveals white matter abnormalities, especially in patients with poor seizure and developmental outcomes. MATERIALS AND METHODS: We enrolled 23 patients with new-onset West syndrome of unknown etiology. DTI was performed at 12 and 24 months of age. Fractional anisotropy images were compared with those of controls by using tract-based spatial statistics. We compared axial, radial, and mean diffusivity between patients and controls in the fractional anisotropy skeleton. We determined correlations of these parameters with developmental quotient, electroencephalography, and seizure outcomes. We also compared DTI with hypometabolism on fluorodeoxyglucose positron-emission tomography. RESULTS: At 12 months of age, patients showed widespread fractional anisotropy reductions and higher radial diffusivity in the fractional anisotropy skeleton with a significant difference on tract-based spatial statistics. The developmental quotient at 12 months of age correlated positively with fractional anisotropy and negatively with radial and mean diffusivity. Patients with seizure and abnormal findings on electroencephalography after initial treatments had lower fractional anisotropy and higher radial diffusivity. At 24 months, although tract-based spatial statistics did not show significant differences between patients and controls, tract-based spatial statistics in the 10 patients with a developmental quotient of <70 had significant fractional anisotropy reduction. In patients with unilateral temporal lobe hypometabolism on PET, tract-based spatial statistics showed greater fractional anisotropy reduction in the temporal lobe ipsilateral to the side of PET hypometabolism. CONCLUSIONS: Diffuse abnormal findings on DTI at 12 months of age suggest delayed myelination as a key factor underlying abnormal findings on DTI. Conversely, asymmetric abnormal findings on DTI at 24 months may reflect underlying focal pathologies.

DOI： 10.3174/ajnr.A4589

Open Access

PubMed

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Language：Japanese   Publisher：(株)東京医学社  

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Other Link： https://search-tp.jamas.or.jp/index.php?module=Default&action=Link&pub_year=2014&ichushi_jid=J00621&link_issn=&doc_id=20140625130016&doc_link_id=%2Faq9syuse%2F2014%2F004406%2F016%2F0813-0818%26dl%3D0&url=https%3A%2F%2Fwww.medicalonline.jp%2Fjamas.php%3FGoodsID%3D%2Faq9syuse%2F2014%2F004406%2F016%2F0813-0818%26dl%3D0&type=MedicalOnline&icon=https%3A%2F%2Fjk04.jamas.or.jp%2Ficon%2F00004_2.gif

Authorship：Lead author,　Corresponding author   Language：Japanese   Publisher：(公社)日本小児科学会  

肺炎球菌結合型ワクチン1回接種後に発症した肺炎球菌性髄膜炎の症例(12歳男児)について報告した。本例は生後9ヵ月に同ワクチンの1回目の接種を受けたが、感冒のため2回目の接種を延期していたところ、生後12ヵ月で肺炎球菌性髄膜炎を発症した。入院時の血液・髄液培養から血清型の6B肺炎球菌が分離された。治療への反応は良好であったが、両側難聴の後遺症が残った。入院時および退院時の血液中の、血清型オプソニン活性が不十分で、このことが発症に関与した可能性が推測されたため、退院後に2回目及び回目の接種を行ったが、血中オプソニン活性は不十分なままであった。本例は肺炎球菌結合型ワクチンを複数回接種しても、抗体のオプソニン活性が不十分な症例が存在することが示唆された症例であった。

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Language：Japanese   Publisher：金原出版(株)  

症例1:3歳9ヵ月女児。母親が切迫早産のため入院した後、「食事拒否」「不自然な話し方」「表情の硬さ」が出現した。心理士による面接を行い、本児はこだわりが強く、発達検査で言語理解と対人関係の遅れがみられ、原因として突然母と会えなくなったという環境の変化に戸惑い、理解不十分のため不安になっていることが考えられた。母との面会のパターンを作ってもらい、食事は無理強いせず、簡単な言葉での状況説明を心がけたところ、1週間ほどで症状は消失した。症例2:3歳1ヵ月女児。担当の保育士が退職したのをきっかけに、保育園のトイレでおしっこができなくなり、母や祖母が出向く必要があった。心理士による面接では、本児はこだわりが強く、コミュニケーションが一方的になりがちであった。母親には「焦らないことが一番」と伝え、新しい担当保育士には共感的な声掛けを提案した。徐々に担当保育士との関係が築かれ、ふたりでトイレに入れるようになり、その後は手にタッチするというパターンでおしっこに行けるようになった。また、保育士の移動に際しては、カレンダーを見せてお別れの日を予告することで乗り越えることができた。

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Language：Japanese   Publisher：(株)日本小児医事出版社  

2007年から2009年の3年間について当院における救急車来院小児科患者につき解析した。各年の当院救急外来を受診する全小児科患者の約4%(年間のべ約500人)が救急車で来院していた。その61%の主訴がけいれんで、うち79%が熱性けいれんと診断された。脳炎、髄膜炎の患者は6例(0.6%)であった。熱性けいれんと診断された児の61%が初発患者であり、年齢分能は1歳がピークであった。年間の熱性けいれん患者数は地域流行感染の発生状況に左右された。熱性けいれんの持続時間が5分未満の群が70%を占め、15分以上の群は4%であった。発熱からけいれんまでの時間は、69%が12時間以内と早期であった。熱性けいれん診断後入院したのは17%で、その主な理由の60%は複雑型けいれんであった。入院例の血液検査において、15分以上けいれんが持続した群で血糖値が有意差を持って高かった。(著者抄録)

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Other Link： https://search.jamas.or.jp/index.php?module=Default&action=Link&pub_year=2011&ichushi_jid=J00643&link_issn=&doc_id=20110106250011&doc_link_id=%2Fag1snrsd%2F2011%2F006401%2F013%2F0078-0084%26dl%3D0&url=https%3A%2F%2Fwww.medicalonline.jp%2Fjamas.php%3FGoodsID%3D%2Fag1snrsd%2F2011%2F006401%2F013%2F0078-0084%26dl%3D0&type=MedicalOnline&icon=https%3A%2F%2Fjk04.jamas.or.jp%2Ficon%2F00004_2.gif

Language：Japanese   Publisher：(株)日本小児医事出版社  

当院小児科で約6年間に加療した川崎病患者148例につき後方視的に検討した。単一施設においてもその季節性、年齢分布、心後遺症について川崎病全国調査と同様の疫学調査結果が出た。また、治療開始時より免疫グロブリン療法(IVIG)を施行した83例につき既存の2つのリスクスコア(群馬スコア、久留米スコア)と、診断に至るまでのすべてのデータを対象とした「トヨタ版久留米スコア」につき、その有用性について検討した。IVIG不応予測カットオフ点を、群馬スコアで6点、久留米スコアとトヨタ版久留米スコアで4点とすると、それぞれの感度/特異度が73%/82%、60%/74%、67%/77%となった。本論文では久留米スコアの「改訂版」を提示し、より現実的な結果を得た。今後スコアリングによる川崎病初期治療層別化の有用性がより一層検証され、新たな治療戦略が確立し、より多くの川崎病罹患児に還元されることを望む。(著者抄録)

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Other Link： https://search.jamas.or.jp/index.php?module=Default&action=Link&pub_year=2010&ichushi_jid=J00643&link_issn=&doc_id=20100924040013&doc_link_id=%2Fag1snrsd%2F2010%2F006310%2F013%2F2135-2141%26dl%3D0&url=https%3A%2F%2Fwww.medicalonline.jp%2Fjamas.php%3FGoodsID%3D%2Fag1snrsd%2F2010%2F006310%2F013%2F2135-2141%26dl%3D0&type=MedicalOnline&icon=https%3A%2F%2Fjk04.jamas.or.jp%2Ficon%2F00004_2.gif

Language：Japanese   Publisher：トヨタ自動車(株)トヨタ記念病院  

症例1(2歳女児)。血便、嘔吐を主訴とした。症例2(1歳男児)。発熱、血便を主訴とした。症例3(3歳女児)。血便を主訴とした。いずれの症例も出血性大腸炎の診断で入院となり、経過中に溶血性尿毒症症候群を発症したが、抗菌薬投与とともに輸液管理が行なわれ、透析を施行することなく軽快が得られた。

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Event date： 2024.3

Presentation type：Oral presentation (invited, special)  

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Presentation type：Oral presentation (invited, special)  

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Language：Japanese   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

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Presentation type：Public lecture, seminar, tutorial, course, or other speech  

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Type：Academic society, research group, etc. 

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