ADHD and ADHD-related neural networks in benign epilepsy with centrotemporal spikes: A systematic review.

BACKGROUND: Attention-deficit/hyperactivity disorder (ADHD) and benign epilepsy with centrotemporal spikes (BECTS or rolandic epilepsy) present with a very high level of comorbidity. We aimed to review the existing literature focusing on two aspects: the possible role of epileptic activity in the damage of ADHD-related neural networks and the clinical approach to patients presenting with both conditions. MATERIAL AND METHODS: A systematic review was performed using Sapienza Library System and PubMed. The following search terms have been considered: attention networks, ADHD, attention systems, rolandic epilepsy, benign epilepsy with centrotemporal spikes, centrotemporal spikes epilepsy, and focal epilepsy in children. The target population consisted of patients under 18 years of age diagnosed with either BECTS and ADHD or healthy controls. RESULTS: Nine case-control and cohort studies have been selected. The reported prevalence of ADHD in patients with BECTS was around 60%. No clinical correlation was found between the medical records and the presence of ADHD in patients with BECTS, if not due to febrile convulsion (FC). One study showed higher levels of bilateral discharges in patients with severe ADHD. The negative influence of the age at onset of seizures was demonstrated on attention but not on intelligence quotient (IQ). Moreover, the frequency of seizures and the occurrence of discharges during nonrapid eye movement (NREM) sleep were correlated to attention impairment. From a neurobiological point of view, functional connectivity in patients with BECTS and ADHD appears to be disrupted. Two studies reported a specific impairment in selective visual attention, while one study underlined a decreased activation of the dorsal attention network (DAN). Two different studies found that patients with BECTS and comorbid ADHD presented with altered thickness in their magnetic resonance imaging (MRI) scans in the cortical and subcortical regions (including the frontal lobes, lingual-fusiform cortex, cuneus and precuneus, limbic area and pericalcarine cortex among others). This might explain the cognitive and behavioral symptoms such as poor selective visual attention, speech disturbance, and impulsivity. CONCLUSIONS: Despite BECTS being considered to have a relative benign course, many studies have documented cognitive and/or behavioral problems in patients diagnosed with this type of epilepsy. In particular, children affected by rolandic epilepsy should receive a complete neuropsychological evaluation at seizure onset considering the high rate of comorbidity with ADHD. A further investigation of the common pathogenic substrate is desirable to better orientate the clinical and therapeutic interventions applied.

Alexithymia, Metacognition, and Theory of Mind in Children and Preadolescents With Migraine Without Aura (MWoA): A Case-Control Study.

Background: Some studies have demonstrated the high impact of headache and migraine in several areas of children and adolescents' life. In recent years, there has been an increase in scientific interest in the relationship between migraine and emotional regulation, investigating the possible consequences of emotional dysregulation on physical and mental health. While some studies have been carried out on the relationship between alexithymia and headache or migraine (especially in adults), no data exist on relationship between Theory of Mind (TOM), metacognition, and alexithymia in children and adolescents with migraine. Methods: Children with diagnosis of migraine without aura (MWoA) (36 males and 34 females) were compared to a healthy control group (31 males and 39 females). The age range was from 8 to 13 years in both groups. All children completed the Alexithymia Questionnaire for Children (AQC) for the assessment of alexithymia levels and the Domain of Social Perception included in the NEPSY-II to evaluate levels of TOM. Metacognitive development was evaluated with Io e La Mia Mente for children aged between 8 and 10 years and with Metacognitions Questionnaire for Children (MCQ-C) for children from 11 to 13. Results: There were no differences between children with MWoA and the control group in metacognitive abilities; only in the subscale "Negative Meta Worrying" of MCQ-C girls scored higher than boys, regardless of the group they were part of. Also, in the NEPSY-II subscale there were no statistically significant differences between the two groups. Children with MWoA scored higher in the AQC subscales "Difficulty Identifying Feelings" and "Difficulty Describing Feelings" than controls. Moreover, children between 8 and 10 years statistically differed from older children in "Difficulty Identifying Feelings" and in Total Score. Conclusion: Our data suggest that children with MWoA have no metacognitive and TOM problems compared to a healthy group. The experimental group showed higher traits of alexithymia, confirming what suggested by other studies in the literature. Future research will have to focus on migraine with aura and tension-type headache to evaluate any differences with children with MWoA.

Behavioral therapies in headache: focus on mindfulness and cognitive behavioral therapy in children and adolescents.

Introduction: A wide proportion of children and adolescents with headache tends not to respond to various pharmacological treatments in use. The failure to respond to symptomatic treatment and prophylaxis is often due to the presence of a comorbid psychopathology undiagnosed or not properly treated. For these reasons and for the negative impact of headache on the quality of life of the patients and on the costs for the public health system, the adoption of an integrated multi-disciplinary perspective in the diagnosis and treatment of headache is needed.Areas covered: Several researchers have shown that behavioral treatment is effective as pharmacological treatment, not only for headache management but also to maintain a lifetime response to the headache treatment. In particular, Cognitive Behavioral Therapy (CBT) and Mindfulness have proven to be very resolutive both in the management of pain and in the management of stressful situations that can trigger the headache in children and adolescents with headache.Expert opinion: Although studies on behavioral treatments on children and adolescents with primary headaches are still few but a combined approach seems to be very useful in improving the quality of life of these subjects, especially in those who have internalizing disorders in comorbidities.

Broadening phenotype of adenylosuccinate lyase deficiency: A novel clinical pattern resembling neuronal ceroid lipofuscinosis.

We describe a 7-year-old boy presenting with a developmental encephalopathy, severe epilepsy, retinopathy with salt and pepper fundus, and ultrastructural skin alterations resembling a neuronal ceroid lipofuscinosis. Whole exome-sequencing detected biallelic variants in the ADSL gene (c.65C > T [p.(Ala22Val)] and c.340 T > C [p.(Tyr114His)]). The increase of SAICAR and S-Ado in blood and urine was consistent with the pattern of adenylosuccinate lyase deficiency (OMIM 103050). An unusual increase of AICAR, that was due to a residual ADSL enzyme activity of about 28%, was also detected. Neither salt and pepper retinopathy nor ultrastructural skin alterations had been reported in ADSL deficiency before. Impaired purinergic signaling inside the retina is probably involved in visual failure. Ultrastructural alterations in fibroblasts suggest a possible damage of autophagic processes, whose role in the pathogenesis of neurological dysfunction deserves further study.

Controlled-release melatonin, singly and combined with cognitive behavioural therapy, for persistent insomnia in children with autism spectrum disorders: a randomized placebo-controlled trial.

Although melatonin and cognitive-behavioural therapy have shown efficacy in treating sleep disorders in children with autism spectrum disorders, little is known about their relative or combined efficacy. One hundred and sixty children with autism spectrum disorders, aged 4-10 years, suffering from sleep onset insomnia and impaired sleep maintenance, were assigned randomly to either (1) combination of controlled-release melatonin and cognitive-behavioural therapy; (2) controlled-release melatonin; (3) four sessions of cognitive-behavioural therapy; or (4) placebo drug treatment condition for 12 weeks in a 1 : 1 : 1 : 1 ratio. Children were studied at baseline and after 12 weeks of treatment. Treatment response was assessed with 1-week actigraphic monitoring, sleep diary and sleep questionnaire. Main outcome measures, derived actigraphically, were sleep latency, total sleep time, wake after sleep onset and number of awakenings. The active treatment groups all resulted in improvements across all outcome measures, with moderate-to-large effect sizes from baseline to a 12-week assessment. Melatonin treatment was mainly effective in reducing insomnia symptoms, while cognitive-behavioural therapy had a light positive impact mainly on sleep latency, suggesting that some behavioural aspects might play a role in determining initial insomnia. The combination treatment group showed a trend to outperform other active treatment groups, with fewer dropouts and a greater proportion of treatment responders achieving clinically significant changes (63.38% normative sleep efficiency criterion of >85% and 84.62%, sleep onset latency <30 min). This study demonstrates that adding behavioural intervention to melatonin treatment seems to result in a better treatment response, at least in the short term.

Sleep in children with autism with and without autistic regression.

The purpose of the present investigation was to characterize and compare traditional sleep architecture and non-rapid eye movement (NREM) sleep microstructure in a well-defined cohort of children with regressive and non-regressive autism, and in typically developing children (TD). We hypothesized that children with regressive autism would demonstrate a greater degree of sleep disruption either at a macrostructural or microstructural level and a more problematic sleep as reported by parents. Twenty-two children with non-regressive autism, 18 with regressive autism without comorbid pathologies and 12 with TD, aged 5-10years, underwent standard overnight multi-channel polysomnographic evaluation. Parents completed a structured questionnaire (Childrens' Sleep Habits Questionnaire-CSHQ). The initial hypothesis, that regressed children have more disrupted sleep, was supported by our findings that they scored significantly higher on CSHQ, particularly on bedtime resistance, sleep onset delay, sleep duration and night wakings CSHQ subdomains than non-regressed peers, and both scored more than typically developing controls. Regressive subjects had significantly less efficient sleep, less total sleep time, prolonged sleep latency, prolonged REM latency and more time awake after sleep onset than non-regressive children and the TD group. Regressive children showed lower cyclic alternating pattern (CAP) rates and A1 index in light sleep than non-regressive and TD children. Our findings suggest that, even though no particular differences in sleep architecture were found between the two groups of children with autism, those who experienced regression showed more sleep disorders and a disruption of sleep either from a macro- or from a microstructural viewpoint.

Sleep in children with autistic spectrum disorder.

Children and adolescents with autistic spectrum disorders (ASD) suffer from sleep problems, particularly insomnia, at a higher rate than typically developing children, ranging from 40% to 80%. Sleep problems in ASD might occur as a result of complex interactions between biological, psychological, social/environmental, and family factors, including child rearing practices that are not conducive to good sleep. Interestingly, children with a history of developmental regression have a more disturbed sleep pattern than children without regression. Even though regulation of sleep in children with ASD is still poorly understood, circadian abnormalities in autism might be the result of genetic abnormalities related to melatonin synthesis and melatonin's role in modulating synaptic transmission. Recently a bifurcation of the sleep/wake cycle with increased sensitivity to external noise and short sleep duration causing irregular sleep onset and wake up times has been suggested. Identifying and treating sleep disorders may result not only in improved sleep, but also impact favorably on daytime behavior and family functioning. Several studies have also demonstrated effectiveness of behavioral interventions for sleep onset and maintenance problems in these populations. When behavioral interventions are not effective or lead only to a partial response, pharmacological treatment options should be considered. Studies of melatonin use in children with ASD provide evidence for its effectiveness and safety in the long run. The clinician assessing a child with an ASD should screen carefully for sleep disorders and make referrals as indicated.

Family and cultural influences on sleep development.

Sleep is a biopsychosocial process that is influenced by the complex interaction of biologic rhythms, including sleep homeostatic mechanisms, chronobiologic factors, parenting, and temperament. However, family structure, culturally specific values, and beliefs are all elements that impact the ecology of a child's sleep. Dramatic demographic changes in many countries have been witnessed in recent years, which have resulted in health professionals and pediatricians being confronted with families of widely differing cultural origins. Attitudes and beliefs on infant sleeping strongly influence whether or not infant sleep behavior is perceived as problematic. Thus, it is advantageous to explore a child's sleep behavior in a multicultural setting. Cultural comparisons are of intrinsic value because they allow not only a better understanding of sleep in different contexts but also an evaluation of the eventual benefits and consequences of different cultural sleep practices. This article provides an overview of some specific sleep-related behaviors and practices highly influenced by different cultural contexts.

Sleep patterns in autism spectrum disorders.

Like children with other developmental disabilities, children with autism spectrum disorders suffer from sleep problems at a greater rate than typically developing children. It is increasingly recognized that addressing these sleep problems may improve daytime functioning and decrease family stress. The sleep problems experienced by children with autism spectrum disorders are presented in this article.

An investigation of sleep characteristics, EEG abnormalities and epilepsy in developmentally regressed and non-regressed children with autism.

This study investigated sleep of children with autism and developmental regression and the possible relationship with epilepsy and epileptiform abnormalities. Participants were 104 children with autism (70 non-regressed, 34 regressed) and 162 typically developing children (TD). Results suggested that the regressed group had higher incidence of circadian rhythm disorders than non-regressed children. The regressed group showed higher Children's Sleep Habits Questionnaire Bedtime Resistance, Sleep Onset Delay, Sleep Duration and Night-Wakings scores. Epilepsy and frequent epileptiform EEG abnormalities were more frequent in regressed children. Past sleep disorders and a history of developmental regression were significantly associated with sleep disorders. This study is an initial step in better understanding sleep problems in regressed children with autism, further studies are necessary to better investigate these aspects.

Cosleeping versus solitary sleeping in children with bedtime problems: child emotional problems and parental distress.

This study investigated sleep, behavioral and emotional problems, and parental relationships and psychological distress in a group of school-aged children with bedtime problems and persistent cosleeping, compared to solitary sleepers and controls. Participants were 148 school-aged children with bedtime problems (44 cosleepers, 104 solitary sleepers) and 228 healthy peers. Results suggested that cosleepers have a significantly later bedtime, shorter nighttime sleep duration, higher Children's Sleep Habits Questionnaire (CSHQ) bedtime resistance and sleep anxiety scores, and more behavioral and emotional problems compared to other groups. Parents of cosleepers have a significantly higher level of psychological and couple distress. A past history of sleep problems, couple and maternal distress, CSHQ bedtime resistance, sleep anxiety, and night wakings subscale scores, and nighttime fears were significantly predictive of cosleeping. Thus, when cosleeping is present, the child's emotional adjustment, family relationships, and parental psychological problems should be investigated.

The relationship between reported sleep quality and sleep hygiene in Italian and American adolescents.

OBJECTIVE: The purpose of the study was to examine the relationship between self-reported sleep quality and sleep hygiene in Italian and American adolescents and to assess whether sleep-hygiene practices mediate the relationship between culture and sleep quality. METHODS: Two nonprobability samples were collected from public schools in Rome, Italy, and Hattiesburg, Mississippi. Students completed the following self-report measures: Adolescent Sleep-Wake Scale, Adolescent Sleep Hygiene Scale, Pubertal Developmental Scale, and Morningness/Eveningness Scale. RESULTS: The final sample included 776 Italian and 572 American adolescents 12 to 17 years old. Italian adolescents reported much better sleep hygiene and substantially better sleep quality than American adolescents. A moderate-to-strong linear relationship was found between sleep hygiene and sleep quality in both samples. Separate hierarchical multiple regression analyses were performed on both samples. Demographic and individual characteristics explained a significant proportion of the variance in sleep quality (Italians: 18%; Americans: 25%), and the addition of sleep-hygiene domains explained significantly more variance in sleep quality (Italians: 17%; Americans: 16%). A final hierarchical multiple regression analysis with both samples combined showed that culture (Italy versus United States) only explained 0.8% of the variance in sleep quality after controlling for sleep hygiene and all other variables. CONCLUSIONS: Cross-cultural differences in sleep quality, for the most part, were due to differences in sleep-hygiene practices. Sleep hygiene is an important predictor of sleep quality in Italian and American adolescents, thus supporting the implementation and evaluation of educational programs on good sleep-hygiene practices.

Sleep hygiene and sleep quality in Italian and American adolescents.

This study investigated cross-cultural differences in adolescent sleep hygiene and sleep quality. Participants were 1348 students (655 males; 693 females) aged 12-17 years from public school systems in Rome, Italy (n = 776) and Southern Mississippi (n = 572). Participants completed the Adolescent Sleep-Wake Scale and the Adolescent Sleep Hygiene Scale. Reported sleep hygiene and sleep quality were significantly better for Italian than American adolescents. A moderate linear relationship was observed between sleep hygiene and sleep quality in both samples (Italians: R =.40; Americans: R =.46). Separate hierarchical multiple regression analyses showed that sleep hygiene accounted for significant variance in sleep quality, even after controlling for demographic and health variables (Italians: R(2) =.38; Americans: R(2) =.44). The results of this study suggest that there are cultural differences in sleep quality and sleep hygiene practices, and that sleep hygiene practices are importantly related to adolescent sleep quality.

Knowledge of sleep in Italian high school students: pilot-test of a school-based sleep educational program.

PURPOSE: To evaluate knowledge about sleep and the effect of a sleep educational program on a group of Italian adolescents. METHODS: The program consisted of a 2-hour interactive sleep educational course. To assess students' sleep patterns and habits, the School Sleep Habits Survey was completed by 540 students, aged 17 to 19 years, who were attending three secondary public schools. Students were randomly assigned to the control and intervention group. Only the latter participated in the educational program. A pre-test evaluated baseline knowledge about sleep, a post-test measured the gain in knowledge after the course, and a 3-month follow-up test evaluated long-term retention of information. Improvement in knowledge was measured by increase in correct answers and comparisons of means between baseline and follow-up mean scores. Control group completed only baseline and 3-month follow-up tests. Data were analyzed using repeated measures analysis of variance, Cochran Q, and McNemar tests. RESULTS: Paired data were available for 425 students. Results showed unhealthy sleeping habits in 34% of students associated with high level of subjective sleepiness, increased vulnerability to injuries, and poor daytime functioning. Pre-test score showed a value of 4.2, post-test of 8.6 and 3-month follow-up of 6.7. Low baseline knowledge about sleep, an increase in knowledge, with an average of 50% gain in the percentage of correct answers immediately after the course, with a good long-term retention of information were found for the students who received the intervention as compared with control group. CONCLUSION: Sleep educational programs for secondary students are recommended to improve information about sleep.

Cosleeping and sleep behavior in Italian school-aged children.

The prevalence and predictors of cosleeping were investigated in 901 healthy school-aged children. Parent reports on the Children's Sleep Habits Questionnaire and Child Behavior Checklist were used to assess children's sleep and behavioral problems. Regular, long-lasting cosleeping was present in 5% of our sample. Cosleepers rated higher on the Children's Sleep Habits Questionnaire total score and Bedtime Resistance, Sleep Anxiety, Nightwakings, and Parasomnias subscales than solitary sleepers. No significant behavioral problems were found in cosleepers. Regression results showed that low socioeconomic status, one parent who is a shiftworker, one-parent families, one parent who coslept as a child, prolonged breastfeeding, and previous and current sleep problems significantly predicted cosleeping. The high incidence of parents reporting having coslept as a child also suggested a lifestyle choice. Thus, cosleeping seems to reflect a parent's way to cope with sleep problems, and the long persistence of this practice may be related to the lifestyle of families.

Circadian preference, sleep and daytime behaviour in adolescence.

The aim of this study was to determine the relationship between circadian preferences, regularity of sleep patterns, sleep problems, daytime sleepiness and daytime behaviour. As a part of an epidemiological survey on sleep in a representative sample of Italian high-school students, a total of 6631 adolescents, aged 14.1-18.6 years, completed the School Sleep Habits Survey, a comprehensive questionnaire including items regarding sleep, sleepiness, substance use, anxiety and depressed mood, use of sleeping pills, school attendance and a morningness/eveningness scale. The sample consisted of 742 evening-types (315 males and 427 females; mean age 17.1 years) and 1005 morning-types (451 males and 554 females; mean age 16.8 years). No significant sex differences were found for morningness/eveningness score. Eveningness was associated with later bedtime and wake-up time, especially on weekends, shorter time in bed during the week, longer weekend time in bed, irregular sleep-wake schedule, subjective poor sleep. Moreover, evening types used to nap more frequently during school days, complained of daytime sleepiness, referred more attention problems, poor school achievement, more injuries and were more emotionally upset than the other chronotype. They referred also greater caffeine-containing beverages and substances to promote sleep consumption. Our results suggest that circadian preference might be related not only to sleep pattern, but also to other adolescent behaviours.