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BACKGROUND: Non-occlusive mesenteric ischemia (NOMI) is a rare and severe pathological condition that can cause intestinal necrosis without mechanical obstruction of the mesenteric artery. NOMI often develops during the treatment of severe disease in elderly patients and mostly occurs in the intestine supplied by the superior mesenteric artery (SMA). We experienced a 12-year-old patient with NOMI that was segmentally localized in the ascending colon and rectum during encephalitis treatment. CASE PRESENTATION: A 12-year-old boy was hospitalized with limbic encephalitis. On day 41 after admission, he abruptly developed hypotension following diarrhea and fever, and presented abdominal distension. A computed tomography scan revealed pneumatosis intestinalis localized in the ascending colon and rectum coexisting with portal venous gas. The presence of peritoneal signs required an emergency laparotomy. Intraoperatively, skip ischemic lesions were found in the ascending colon and the rectum without bowel perforation. SMA and superior rectal arterial pulsation were present, and the patient was diagnosed with NOMI. The remaining colon, from the transverse to the sigmoid colon, appeared intact. We performed a distal ileostomy without bowel resection. Postoperative colonoscopies were carried out and revealed rectal and ascending colon stenosis with ulceration but demonstrated the patency of the two lesions. We confirmed the improvement of the transient bowel strictures; therefore, the ileal stoma was closed 14 months after the previous laparotomy. CONCLUSION: NOMI can be present in childhood during encephalitis treatment and can be segmentally localized in the ascending colon and the rectum. Although NOMI is most often seen in elderly patients, we should also consider the possibility of NOMI when pediatric patients with severe illness manifest abdominal symptoms.

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OBJECTIVE: Attention-deficit/hyperactivity disorder (AD/HD) is a common developmental disorder. Many reports have suggested that symptoms of AD/HD are related to frontal lobe dysfunctions, particularly disinhibition. However, measuring neurological findings with biomarkers during frontal functional tasks has sometimes been difficult in children with AD/HD. This study aimed to investigate frontal inhibitory function objectively in children with AD/HD during "rock, paper, scissors" (RPS) tasks, as a familiar game for Japanese children, using near-infrared spectroscopy (NIRS). SUBJECTS AND METHODS: Eighteen children with AD/HD were compared with 27 typically developing children (TDC). Children from each group were divided into two age groups: younger, 6-10years; and older, 11-16years. Changes in oxygenated hemoglobin [oxy-Hb] were measured in the prefrontal region using NIRS during a 'to lose' RPS task, in which subjects were asked to present the RPS signal that would lose in response to one of the three signals displayed randomly on a computer screen every 2.0s. RESULTS: The rate of correct performance with both TDC and AD/HD increased with age. Only in the older group, the rate of correct performance was significantly higher with TDC than with AD/HD. However, children with AD/HD in both age groups showed significantly lower [oxy-Hb] activity in the prefrontal region during the 'to lose' RPS task, particularly in the dorsolateral area. CONCLUSIONS: Our results suggest that prefrontal region activation during the 'to lose' RPS task could offer a biomarker for diagnosing AD/HD, and may help in the early treatment of AD/HD.

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We report the case of a 5-year-old boy with acute encephalopathy presenting with transient executive dysfunction such as functional disability in various new tasks and hypoperfusion of the right frontal and temporal lobes on single photon emission tomography (SPECT). He presented with a 2-day history of disturbed consciousness, and electroencephalography in an awaked state showed diffuse high-voltage slow waves. Although MRI did not show any abnormality 3 days after initial onset of illness, SPECT showed hypoperfusion of the right frontal and temporal lobes at the same time. At 20 days after onset, the Kaufman assessment battery for children (K-ABC) test showed that sequential processing scale scores were significantly lower than simultaneous processing scale and achievement scale scores. He showed transient executive dysfunction such as functional disability in various new tasks at the same time. Abnormal brain perfusion on SPECT was improved at 8 months after onset and the sequential processing scale of K-ABC was likewise improved at 12 months after onset. These findings suggest that SPECT is helpful for diagnosing pathophysiological mechanisms with acute encephalopathy, and the combination of neuropsychological examination and SPECT study is useful for evaluating higher brain dysfunctions such as executive dysfunction.

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OBJECT: Improving quality of life (QOL) is one of the most important therapeutic goals for children with attention-deficit hyperactiv- ity disorder (AD/HD). The aim of this study was to measure QOL in AD/HD children without comorbidity and to examine associations between QOL and clinical symptoms of AD/HD for targeting early intervention. METHODS: Twenty-two enrolled patients and their parents completed the Questionnaire for Measuring Health-Related Quality of Life in Children (KINDL-R). Patients and teachers completed AD/HD rating scale-IV. Associations between QOL and clinical symptoms were assessed using t tests and correlations. RESULTS: Mean total score of the self-reported KINDL-R was 70.8. No difference in total QOL score was seen between AD/HD children and controls; however, the self-esteem subscale rated by AD/HD children was significantly higher than that of controls (p < 0.001). Total KINDL-R score correlated negatively with AD/HD rating scale-IV rated by teachers (p < 0.05). A difference was observed between AD/HD children in a lower QOL group and their parents in a subscale regarding QOL at school. CONCLUSIONS: These findings suggest that evaluation of QOL in AD/HD children without comorbidity is useful for identifying AD/HD children who might benefit from early intervention.

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OBJECTIVES: The purpose of the present study was to evaluate the efficacy and safety of topiramate (TPM) on inter-ictal headache in children with epilepsy. METHODS: Patients were interviewed regarding whether they suffered from headaches. Data obtained from each patient included seizure frequency. Inter-ictal headache was defined as a headache beginning outside an hour before or after the seizure. The study group included 85 outpatients (42 valproate-treated, 34 carbamazepine-treated, 6 combination therapy, 3 other) between 5 and 15 years old. For children with headache, TPM was administered twice daily at a total initial dose of 0.5 mg/kg/day, up to 3.0 mg/kg/day in accordance with symptoms. RESULTS: Of 85 patients, 18 (21.2%) patients (8 valproate-treated, 6 carbamazepine-treated, 3 combination therapy, and 1 other) complained of inter-ictal headache. Seizure frequency was significantly higher in children with headache (2.6 times/year) than in children without headache (0.9 times/year; p < 0.0001). The responder rate (rate of patients with a > 50% reduction in headache frequency or degree) was 13/18 (72%). Six children (33.3%) achieved complete cessation for the entire 6 months. Mean dose of TPM was significantly lower in responders (1.1 mg/kg/day) than in non-responders (2.7 mg/kg/day; p < 0.001). CONCLUSIONS: Headache is encountered more frequently in patients with frequent seizures. In addition, TPM represents a useful addition to the treatments available for headache in children with epilepsy. The effective dose of TPM for headache may be lower than that for seizure.

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BACKGROUND: Improvement of the quality of life (QOL) for children with epilepsy is one of the most important therapeutic goals. It is widely acknowledged that in adults with epilepsy one of the best QOL predictors is psychiatric comorbidity. In children with epilepsy, however, it is not clear whether psychiatric comorbidity impairs QOL. AIMS: The aim of this study was to evaluate QOL in children with epilepsy and to identify the strongest predictors of the same. METHODS: A total of 28 enrolled patients completed the Questionnaire for Measuring Health-Related Quality of Life in Children (KINDL-R) and 3 assessments of clinical status: the Depression Self-Rating Scale for Children (DSRS-C), the Children Manifest Anxiety Scale (CMAS), and the Side Effects and Life Satisfaction (SEALS). Various demographic and clinical factors were analyzed as possible predictors of KINDL-R scores. RESULTS: The strongest predictor of QOL was the total DSRS-C score (r = -0.69, p < 0.01), which also predicted physical (r = -0.58, p < 0.01) and emotional wellbeing (r = -0.53, p < 0.05) subscale scores. CONCLUSIONS: Symptoms of depression were more predictive of QOL than were seizure type, seizure duration, number and adverse effects of AEDs, or anxiety. Number of AEDs did have an effect, just not as prominent as symptoms of depression.

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OBJECTIVE: Near-infrared spectroscopy (NIRS) is commonly used to investigate continuous changes of brain activation and has excellent time resolution. Verbal fluency task (VFT) is widely used as a neuropsychological test of frontal lobe function. The aim of this study was to investigate normal developmental change in frontal lobe function during VFT performance using multi-channel NIRS, specifically focusing on oxygenation hemoglobin (oxyHb) changes. METHODS: The subjects were 9 adults and 37 childrens who were all healthy right-handed volunteers. Children were divided into four age groups (group A, 6-8 years; group B, 9-11 years; group C, 12-14 years; group D, 15-18 years). The [oxyHb] changes were measured with 22 channels of NIRS during VFT. We defined the frontopolar region as the region of interest for analysis, and calculated the Z-score to compare the data between groups. RESULTS: The task performance changed with age. There were significant differences between group A and other groups. The Z-score of [oxyHb] also significantly increased with age, when comparing adults to groups A and B. The task performances decreased with time in all groups. In contrast, [oxyHb] only continued to increase in the adult group. CONCLUSION: The verbal retrieval functions begin to mature in early adolescence and continue to grow up to adulthood.

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We investigated the relationship between neuropsychological disturbance, assessed using the global assessment of functioning (GAF) and the ADHD-rating scale (ADHD-RS), paroxysmal EEG abnormalities, and treatment with valproate sodium (VPA) in children with both attention deficit hyperactivity disorder (ADHD) and paroxysmal abnormality (PA). Participants with ADHD but without obvious epilepsy were recruited between April 1, 2003 and March 31, 2008. Paroxysmal abnormality was scored by measuring the spike frequency. Of 46 children, 16 showed PA; 3 of the 16 were excluded because no follow-up EEG was available. The EEG improved with VPA treatment in 5 of 8 patients with frontal PA and 3 of 5 patients with rolandic PA. While 83.3% of the patients with improvements in both assessments had frontal PA, only 16.7% had rolandic PA. The patients with frontal PA showed a significantly higher correlation between PA frequency and improvement in ADHD-RS compared with those with rolandic PA. In this study of children with ADHD, EEG improvement with antiepileptic drug treatment showed a high correlation with behavioral improvements as shown by ADHD-RS and GAF scores. However, this was not a population-based study, and the relative importance of detecting and treating PA in ADHD has yet to be determined.

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BACKGROUND: Little is known about what parents think and how they act when their child experiences febrile seizure (FS). This study therefore surveyed parents of 78 children who had experienced a first FS regarding their thoughts and actions. METHODS: The questionnaire was divided into three parts: details of the child and their family; medical management of the child before reaching hospital; and parental thoughts and actions when the child experienced convulsions. RESULTS: Parents without prior knowledge of FS showed a higher rate of thinking that FS were harmful than parents with prior knowledge (P < 0.03). Parents with prior knowledge were aware that their child was having an FS at a higher rate than parents without prior knowledge (P < 0.001). Moreover, parents without prior knowledge managed the convulsions less appropriately than parents with prior knowledge (P < 0.03). CONCLUSIONS: Parental fears that the death of their child was imminent and the misperception of FS as a serious, life-threatening condition indicate a lack of knowledge regarding FS. Organizing parental support groups and effective educational intervention programs for parents should be given priority in the care of children with FS.

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PURPOSE: The purpose of the present study was to evaluate the efficacy and safety of levetiracetam (LEV) in refractory epileptic children. METHODS: The study group included 61 outpatients (7 generalized, 48 localization-related, 3 undetermined, 3 unclassified) aged between 16 months and 18 years. LEV was given twice daily at a total dose of 10 mg/kg/day. The final mean dose was 50.7 mg/kg/day. The mean number of prior anti-epileptic drugs was 5.2. The entire treatment period was more than 6 months after LEV administration. RESULTS: Fifteen children (24.6%) became seizure-free for 6 months after starting LEV, and 18 (29.5%) had a seizure reduction of more than 50% for the entire 6 months. The response rate was 33/61 (54.1%). Responders included 2/3 of patients (66.7%) with epilepsy with continuous spikes and waves during slow sleep and 13/19 (68.4%) with frontal lobe epilepsy. The effective dosage of LEV in the responders demonstrated a wide range (mean, 46.1 mg/kg/day; range, 19.4-59.1 mg/kg/day), and showed bimodal distribution. Adverse events occurred in only two patients who did not require LEV discontinuation. CONCLUSION: LEV represents an important addition to the treatments available for refractory epileptic children.

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BACKGROUND: The high occurrence of epilepsy in children with autism spectrum disorders (ASD) is a clear indication that ASD has a neurobiological basis. The current understanding of the association between epilepsy and ASD is still limited, but from a clinical point of view, this association should not be overlooked. AIMS: We investigated the electroencephalogram (EEG) paroxysmal abnormality in children with ASD and the incidence of later development of epilepsy. METHODS: Participants were recruited from University of Yamanashi hospital and 5 satellite hospitals between April 1, 2001 and March 31, 2005. EEG recordings and clinical evaluations were performed every 6 months for at least 6 years, focusing on paroxysmal abnormality. We scored the occurrence and the location of spikes and evaluated the relation with later development of epilepsy. RESULTS: The prospective study included 21 patients with ASD (12 males and 9 females) between the ages of 3 and 6 years. EEG paroxysmal abnormalities were present in 11/21 patients (52.4%). In addition, six of 21 patients (28.6%) had epilepsy at some point in their lives. The presence of frontal paroxysms was significantly associated with later development of epilepsy compared with centrotemporal paroxysmus (p < 0.003). The type of seizure diagnosed was mainly partial; in particular, partial with secondary generalization in 4/6 (66.7%). CONCLUSION: The presence of frontal paroxysms may indicate a higher risk of epilepsy in ASD.

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BACKGROUND: The possible consequences of seizures in the immature brain have been the subject of much conjecture. We prospectively measured frontal and prefrontal lobe volumes using three-dimensional (3D) magnetic resonance imaging (MRI)-based volumetry in patients with frontal lobe epilepsy (FLE) presenting with the same seizure semiology. The pathogenesis of repeated seizure-induced brain damage is discussed herein. METHODS: Serial changes in regional cerebral volumes were measured in two patients with FLE presenting with intractable clinical courses and cognitive impairments/behavioral problems (FLE(+)) and four FLE patients without cognitive impairments/behavioral problems (FLE(-)). Eleven normal subjects (4-13 years old) served as controls. Volumes of the frontal and prefrontal lobes were determined using a workstation, and the prefrontal-to-frontal lobe volume ratio was calculated. RESULTS: Frontal and prefrontal lobe volumes revealed growth disturbance in FLE(+) compared with those of FLE(-) and control subjects. In addition, prefrontal-to-frontal lobe volume ratio increased serially in FLE(-) similarly to controls, but was stagnant or decreased in FLE(+). Prefrontal growth also revealed more rapid recovery in a FLE(+) patient with shorter active seizure period. CONCLUSION: These findings suggest that repeated seizures may lead to prefrontal growth disturbance. The occurrence of frequent seizures in patients with FLE may be associated with prefrontal lobe growth retardation, which relates to neuropsychological problems and ultimate neuropsychological outcome.

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Event-related potentials (ERPs) were measured in eight healthy right-handed adults during three different visual Go/NoGo tasks, which consisted of color, figure, and Chinese character (kanji) paradigms. Subjects responded to a Go stimulus after a cue stimulus from the monitor during the execution of a continuous performance test (CPT). We obtained ERPs of the Go and NoGo conditions from 13-channel EEG recordings and measured peak latency, amplitude, and topographic distribution of each component. The N2 components of the figure and kanji NoGo tasks were located from Fz to Cz;o n the other hand, the N2 distribution of the color task was significant from Cz to Pz. The P3 component of both the Go and NoGo tasks were mostly seen from Cz to Pz. The NoGo-P3 components were distributed in more anterior regions than Go-P3 topography. In the color task, the amplitude of NoGo P3 was highest and the latency was shortest of the three tasks. These results suggest that ERP components in visual Go/NoGo tasks are most distinguishable in the color task.,and that NoGo potentials in a color task might be a useful tool for cognitive testing in children.

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In order to investigate the developmental changes of response inhibition in frontal function, event related potentials (ERPs) during a Go/NoGo task were collected. Twelve developmentally normal subjects, ranging in age from 7 to 15, and 9 normal adults performed a visual Go/NoGo task, which is a continuous performance test (CPT) using 5 color samples. We obtained ERPs of the Go and NoGo conditions from 13-channel EEG recordings and measured peak latency, amplitude, and topographic distribution of the NoGo N2 and NoGo P3 components. The NoGo N2 components were located at Fz in childhood, but changed from Cz to Pz in adulthood. The NoGo N2 amplitude diminished linearly with age. The NoGo P3 components were significant at Cz at all ages and the amplitude increased with age. The pattern of NoGo N2 and NoGo P3 components in 7-to 15-year-old children were similar, but differed in adults. These results suggest that the maturation of response inhibition in frontal lobe is achieved beyond 15 years of age, and that NoGo 3 amplitude particularly reflects the maturation of response inhibition.

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