Randomised trial showed no difference in behavioural symptoms between surgical methods treating paediatric obstructive sleep apnoea.

AIM: Our previous randomised controlled trial of children with obstructive sleep apnoea (OSA) showed no significant differences between adenotonsillectomy (ATE) and adenotonsillotomy (ATT) in improving nocturnal respiration and quality of life after 1 year. The aim of this report was to evaluate the effects on behavioural symptoms using the Strengths and Difficulties Questionnaire (SDQ). METHODS: Children between 2 and 6 years with OSA were randomised to ATT or ATE. Parents, blinded to method, answered the SDQ while their child underwent polysomnography before and 1 year after surgery. Differences between the total SDQ scores were analysed between the treatment groups. RESULTS: The SDQ was filled out in 87% of the cases preoperatively, and in 86% postoperatively. At follow-up, the mean total SDQ score was 9.6 SD ± 5.1 in the ATE group (n = 31), and 8.2 ± 6.7 in the ATT group (n = 37), P = .09. The mean total SDQ score for all was preoperatively 10.6 ± 5.0, and postoperatively 8.8 ± 6.0, P = .0002. CONCLUSION: There were no significant differences in SDQ scores between the groups at follow-up, indicating that the more conservative ATT is a treatment option in paediatric OSA. The whole group of patients showed a significant improvement after surgery.

Change in risk for narcolepsy over time and impact of definition of onset date following vaccination with AS03 adjuvanted pandemic A/H1N1 influenza vaccine (Pandemrix) during the 2009 H1N1 influenza pandemic.

PURPOSE: To estimate risk for narcolepsy in defined time windows following exposure to adjuvanted A(H1N1) pandemic vaccine (Pandemrix) and impact of different definitions of index date for the narcolepsy diagnosis. METHODS: Vaccine exposure in approximately 30% of the Swedish population in 2009 was linked to information on narcolepsy diagnosis retrieved from the national patient registry. Cases were verified by a systematic chart review. Poisson regression was used to compare incidence in defined time windows following vaccination. RESULTS: Of 266 cases of narcolepsy identified, 25% (66/266) were prevalent cases with symptom onset documented before vaccine exposure. Incident cases had a median time interval between first symptom and the date recorded in the patient registry of 64 weeks (IQR 39-107) when vaccinated (N = 182) and 65 weeks (IQR 51-72) when unvaccinated (N = 16). With first symptom defining index date, the adjusted risk for narcolepsy in younger patients was increased 14 times during the first year after vaccination, three times elevated the second year, but with no detectable increased risk more than 2 years after vaccination exposure. Using the index date from the patient registry, the adjusted increase in risk was about seven times elevated for all three time intervals. CONCLUSIONS: The magnitude of the estimated increased risk for narcolepsy following exposure to the A(H1N1) pandemic vaccine is highly dependent on the method used to determine the index date for disease onset. The sometimes very long and potentially variable interval from first symptom to a health care registry diagnosis complicates estimations of risk.

Pandemrix-induced narcolepsy is associated with genes related to immunity and neuronal survival.

BACKGROUND: The incidence of narcolepsy rose sharply after the swine influenza A (H1N1) vaccination campaign with Pandemrix. Narcolepsy is an immune-related disorder with excessive daytime sleepiness. The most frequent form is strongly associated with HLA-DQB1*06:02, but only a minority of carriers develop narcolepsy. We aimed to identify genetic markers that predispose to Pandemrix-induced narcolepsy. METHODS: We tested for genome-wide and candidate gene associations in 42 narcolepsy cases and 4981 controls. Genotyping was performed on Illumina arrays, HLA alleles were imputed using SNP2HLA, and single nucleotide polymorphisms were imputed using the haplotype reference consortium panel. The genome-wide significance threshold was p < 5 × 10-8, and the nominal threshold was p < 0.05. Results were replicated in 32 cases and 7125 controls. Chromatin data was used for functional annotation. FINDINGS: Carrying HLA-DQB1*06:02 was significantly associated with narcolepsy, odds ratio (OR) 39.4 [95% confidence interval (CI) 11.3, 137], p = 7.9 × 10-9. After adjustment for HLA, GDNF-AS1 (rs62360233) was significantly associated, OR = 8.7 [95% CI 4.2, 17.5], p = 2.6 × 10-9, and this was replicated, OR = 3.4 [95% CI 1.2-9.6], p = 0.022. Functional analysis revealed variants in high LD with rs62360233 that might explain the detected association. The candidate immune-gene locus TRAJ (rs1154155) was nominally associated in both the discovery and replication cohorts, meta-analysis OR = 2.0 [95% CI 1.4, 2.8], p = 0.0002. INTERPRETATION: We found a novel association between Pandemrix-induced narcolepsy and the non-coding RNA gene GDNF-AS1, which has been shown to regulate expression of the essential neurotrophic factor GDNF. Changes in regulation of GDNF have been associated with neurodegenerative diseases. This finding may increase the understanding of disease mechanisms underlying narcolepsy. Associations between Pandemrix-induced narcolepsy and immune-related genes were replicated.

Sleep patterns in school-age children with Asperger syndrome or high-functioning autism: a follow-up study.

The course of sleep patterns over 2-3 years was compared between 16 school-age children with Asperger syndrome (AS) or high-functioning autism (HFA) and 16 age- and gender-matched typically developing children, using 1-week actigraphy at baseline and follow-up. At baseline (mean age 11.1 years), children with AS/HFA had longer sleep latency and lower sleep efficiency during school days, but earlier sleep start and sleep end during weekends. At follow-up (mean age 13.7 years), children with AS/HFA had longer night wakings and lower sleep efficiency during weekends than the controls. The overall change of sleep patterns, however, is similar in children with AS/HFA and typically developing controls over a 2 to 3-year period.

Health-related quality of life in parents of school-age children with Asperger Syndrome or High-Functioning Autism.

BACKGROUND: The estimated prevalence rate of Pervasive Developmental Disorders (PDD) in children is 6 per 1.000. Parenting children who are intellectually impaired and have PDDs is known to be linked to the impaired well-being of the parents themselves. However, there is still little available data on health-related quality of life (HRQL) in parents of children with Asperger Syndrome (AS) and High-Functioning Autism (HFA), or other PDD diagnoses in children of normal intelligence. The present study aimed to evaluate aspects of HRQL in parents of school-age children with AS/HFA and the correlates with child behaviour characteristics. METHODS: The sample consisted of 31 mothers and 30 fathers of 32 children with AS/HFA and 30 mothers and 29 fathers of 32 age and gender matched children with typical development. Parental HRQL was surveyed by the use of the 12 Item Short Form Health Survey (SF-12) which measures physical and mental well-being. The child behaviour characteristics were assessed using the structured questionnaires: The High-Functioning Autism Spectrum Screening Questionnaire (ASSQ) and The Strengths and Difficulties Questionnaire (SDQ). RESULTS: The mothers of children with AS/HFA had lower SF-12 scores than the controls, indicating poorer physical health. The mothers of children with AS/HFA also had lower physical SF-12 scores compared to the fathers. In the AS/HFA group, maternal health was related to behaviour problems such as hyperactivity and conduct problems in the child. CONCLUSION: Mothers but not fathers of children with AS/HFA reported impaired HRQL, and there was a relationship between maternal well-being and child behaviour characteristics.

Sleep patterns of school-age children with Asperger syndrome or high-functioning autism.

Sleep patterns of 32 school-age children with Asperger syndrome (AS) and high-functioning autism (HFA) were compared to those of 32 typically developing age- and gender-matched children, using parent survey and one week of diary and actigraphic monitoring. Parents of children with AS/HFA more commonly reported that their children had difficulty falling asleep. One week of sleep recording with diary and actigraphy confirmed that children in the AS/HFA group spent a longer time awake in bed before falling asleep than children in the control group, possibly because the children in the AS/HFA group had earlier bedtimes. Other essential aspects of sleep patterns coincided between the groups. The sleep patterns of children with AS and HFA did not differ.

Insomnia in school-age children with Asperger syndrome or high-functioning autism.

BACKGROUND: Asperger syndrome (AS) and high-functioning autism (HFA) are pervasive developmental disorders (PDD) in individuals of normal intelligence. Childhood AS/HFA is considered to be often associated with disturbed sleep, in particular with difficulties initiating and/or maintaining sleep (insomnia). However, studies about the topic are still scarce. The present study investigated childhood AS/HFA regarding a wide range of parent reported sleep-wake behaviour, with a particular focus on insomnia. METHODS: Thirty-two 8-12 yr old children with AS/HFA were compared with 32 age and gender matched typically developing children regarding sleep and associated behavioural characteristics. Several aspects of sleep-wake behaviour including insomnia were surveyed using a structured paediatric sleep questionnaire in which parents reported their children's sleep patterns for the previous six months. Recent sleep patterns were monitored by use of a one-week sleep diary and actigraphy. Behavioural characteristics were surveyed by use of information gleaned from parent and teacher-ratings in the High-Functioning Autism Spectrum Screening Questionnaire, and in the Strengths and Difficulties Questionnaire. RESULTS: Parent-reported difficulties initiating sleep and daytime sleepiness were more common in children with AS/HFA than in controls, and 10/32 children with AS/HFA (31.2%) but none of the controls fulfilled our definition of paediatric insomnia. The parent-reported insomnia corresponded to the findings obtained by actigraphy. Children with insomnia had also more parent-reported autistic and emotional symptoms, and more teacher-reported emotional and hyperactivity symptoms than those children without insomnia. CONCLUSION: Parental reports indicate that in childhood AS/HFA insomnia is a common and distressing symptom which is frequently associated with coexistent behaviour problems. Identification and treatment of sleep problems need to be a routine part of the treatment plan for children with AS/HFA.

The Minimal Insomnia Symptom Scale (MISS): a brief measure of sleeping difficulties.

To evaluate basic psychometric properties and obtain normative values for a novel 3-item scale, the Minimal Insomnia Symptom Scale (MISS), a sleep questionnaire was sent out to a randomly selected sample of the general population, aged 20-64 years. Responses were obtained from 1075 subjects corresponding to a response rate of 78%. Results showed that MISS possessed satisfactory reliability and validity. Women scored significantly higher than men while there was no age relationship. A receiver operating characteristic curve analysis revealed that MISS was able to distinguish subjects with a clinical insomnia according to ICD-10 research criteria. The main advantage of MISS over other insomnia instruments is its brevity and ease of use. Evidence was provided for the utility of MISS in epidemiological settings. MISS also showed promise as a convenient ultra-short screening measure of insomnia in health care settings.

Validity of the Swedish version of the Strengths and Difficulties Questionnaire (SDQ-Swe).

The Strengths and Difficulties Questionnaire (SDQ) is an internationally widely used, brief screening instrument for mental health problems in children and teenagers. The SDQ probes behaviours and psychological attributes reflecting the child's difficulties as well as strengths, and targets hyperactivity/inattention, emotional symptoms, conduct problems, peer problems and prosocial behaviour. Also, the instrument taps the impact aspect, i.e. whether the child is judged to suffer from emotional or behavioural problems severe enough to cause distress or social impairment. Studies of the original English SDQ, as well as of translations into several other languages, attest to a compelling usefulness and validity of the instrument. In this investigation, the adequacy of the Swedish adaptation of the SDQ (SDQ-Swe) was tested in comparisons between parent reports on 5-15-year-old children drawn from a community sample (n=263) and from a child psychiatric sample (n=230). Results showed that the instrument differentiated well between the community and the psychiatric samples, the latter displaying more symptoms, fewer strengths and more social impairment. Moreover, ROC analyses showed satisfactory sensitivity and specificity of the principal scales of the SDQ-Swe at proposed cut-offs. Hence, results showed adequate validity of the SDQ-Swe, suggesting that this new instrument, an instrument in tune with the ideas of contemporary child psychiatry and psychology, is a useful tool for mental health screening in children and adolescents.

The Strengths and Difficulties Questionnaire in the Nordic countries.

BACKGROUND: The Strengths and Difficulties Questionnaire (SDQ) has been translated into the different Nordic languages between 1996 and 2003. During the past few years, SDQs have been completed for nearly 100,000 children and adolescents in population-based studies as well as in clinical samples. The largest studies have been performed in Norway and Denmark, and in these countries the diagnostic interview DAWBA has also been used in conjunction with the SDQ. AIMS: In addition to a brief overview of past and ongoing SDQ work in Sweden, Finland, Norway, Denmark, and Iceland, we present scale means and standard deviations from selected community studies with comparable age groups, including parental reports for 7, 9 and 11 year-old children and self-reports of 13 and 15 year-olds. CONCLUSIONS: The descriptive statistics suggest that the distributions of SDQ scores are very similar across the Nordic countries. Further collaborative efforts in establishing norms and evaluating the validity of the SDQ as a screening instrument are encouraged.