Cognitive and academic outcome following cranial irradiation and chemotherapy in children: a longitudinal study

Cranial irradiation therapy (CRT) and chemotherapy are associated with neurobehavioural deficits. Many studies have investigated late effects of these treatments, but few have evaluated changes in abilities over time. This study employed a longitudinal design to map abilities following these treatments. Three groups of children were studied: Group 1 (n = 35): children treated with CRT (18 Gy) + chemotherapy, aged 5 years or less at time of diagnosis; Group 2 (n = 19): children treated with chemotherapy alone, aged 5 years or less at time of diagnosis; Group 3 (n = 35): healthy children. All children were aged 7–13 years at time of initial assessment, with no pre-diagnosis history of neurologic, developmental, or psychiatric disorder. Intellectual and educational abilities were evaluated twice: T1, not less than 2 years post-treatment, and T2, 3 years later. Group 1 achieved poorest results at T1, with comparison groups performing similarly. At T2 group differences were maintained. For verbal skills differences remained stable. Group 1 exhibited deterioration on non-verbal and processing tasks, while comparison groups showed improved abilities. Group 1 exhibited increases in literacy skills, with educational intervention predicting progress. Results suggest cumulative deficits in non-verbal and information processing skills for children treated with CRT + chemotherapy, with other deficits remaining relatively stable over time. Improved literacy skills suggest that gains can occur with remediation. © 2000 Cancer Research Campaig

Pharmacological and non-pharmacological interventions for non-respiratory sleep disturbance in children with neurodisabilities : a systematic review

BACKGROUND: There is uncertainty about the most appropriate ways to manage non-respiratory sleep disturbances in children with neurodisabilities (NDs). OBJECTIVE: To assess the clinical effectiveness and safety of NHS-relevant pharmacological and non-pharmacological interventions to manage sleep disturbance in children and young people with NDs, who have non-respiratory sleep disturbance. DATA SOURCES: Sixteen databases, including The Cochrane Central Register of Controlled Trials, EMBASE and MEDLINE, were searched up to February 2017, and grey literature searches and hand-searches were conducted. REVIEW METHODS: For pharmacological interventions, only randomised controlled trials (RCTs) were included. For non-pharmacological interventions, RCTs, non-randomised controlled studies and before-and-after studies were included. Data were extracted and quality assessed by two researchers. Meta-analysis and narrative synthesis were undertaken. Data on parents' and children's experiences of receiving a sleep disturbance intervention were collated into themes and reported narratively. RESULTS: Thirty-nine studies were included. Sample sizes ranged from 5 to 244 participants. Thirteen RCTs evaluated oral melatonin. Twenty-six studies (12 RCTs and 14 before-and-after studies) evaluated non-pharmacological interventions, including comprehensive parent-directed tailored (n = 9) and non-tailored (n = 8) interventions, non-comprehensive parent-directed interventions (n = 2) and other non-pharmacological interventions (n = 7). All but one study were reported as having a high or unclear risk of bias, and studies were generally poorly reported. There was a statistically significant increase in diary-reported total sleep time (TST), which was the most commonly reported outcome for melatonin compared with placebo [pooled mean difference 29.6 minutes, 95% confidence interval (CI) 6.9 to 52.4 minutes; p = 0.01]; however, statistical heterogeneity was extremely high (97%). For the single melatonin study that was rated as having a low risk of bias, the mean increase in TST was 13.2 minutes and the lower CI included the possibility of reduced sleep time (95% CI -13.3 to 39.7 minutes). There was mixed evidence about the clinical effectiveness of the non-pharmacological interventions. Sixteen studies included interventions that investigated the feasibility, acceptability and/or parent or clinician views of sleep disturbance interventions. The majority of these studies reported the 'family experience' of non-pharmacological interventions. LIMITATIONS: Planned subgroup analysis was possible in only a small number of melatonin trials. CONCLUSIONS: There is some evidence of benefit for melatonin compared with placebo, but the degree of benefit is uncertain. There are various types of non-pharmacological interventions for managing sleep disturbance; however, clinical and methodological heterogeneity, few RCTs, a lack of standardised outcome measures and risk of bias means that it is not possible to draw conclusions with regard to their effectiveness. Future work should include the development of a core outcome, further evaluation of the clinical effectiveness and cost-effectiveness of pharmacological and non-pharmacological interventions and research exploring the prevention of, and methods for identifying, sleep disturbance. Research mapping current practices and exploring families' understanding of sleep disturbance and their experiences of obtaining help may facilitate service provision development. STUDY REGISTRATION: This study is registered as PROSPERO CRD42016034067. FUNDING: The National Institute for Health Research Health Technology Assessment programme

Behavioural treatment to reduce sleep problems in children with autism or fragile X syndrome

There are few well-controlled, published evaluations of sleep interventions for children with developmental disabilities. This paper evaluates a parent training programme using behavioural principles to reduce sleep problems in children with autism or fragile X syndrome (FXS). Training included bedtime routines, reinforcement, effective instructions, partner support, and extinction (removing reinforcement to reduce a behaviour). Programme efficacy was demonstrated by using a multiple baseline across-participant design. Social validity was also assessed. Five children with autism and one with Asperger syndrome (four males, two females; mean age 5y 6mo; age range 3y 5mo to 7y 4mo) and their parents; and seven children with FXS (six males, one female; mean age 4y 9mo; age range 1y 11mo to 9y 1mo) and their parents participated. Ten families completed intervention within the multiple baseline design. Most parents' goals were achieved and visual analysis of the sleep data indicated improvement. Settling problems, night waking, and co-sleeping were effectively reduced. The programme had high social validity and outcomes were clinically significant and maintained at follow-up

Cognitive and academic outcome following cranial irradiation and chemotherapy in children: a longitudinal study.

Cranial irradiation therapy (CRT) and chemotherapy are associated with neurobehavioural deficits. Many studies have investigated late effects of these treatments, but few have evaluated changes in abilities over time. This study employed a longitudinal design to map abilities following these treatments. Three groups of children were studied: Group 1 (n = 35): children treated with CRT (18 Gy) + chemotherapy, aged 5 years or less at time of diagnosis; Group 2 (n = 19): children treated with chemotherapy alone, aged 5 years or less at time of diagnosis; Group 3 (n = 35): healthy children. All children were aged 7-13 years at time of initial assessment, with no pre-diagnosis history of neurologic, developmental, or psychiatric disorder. Intellectual and educational abilities were evaluated twice: T1, not less than 2 years post-treatment, and T2, 3 years later. Group 1 achieved poorest results at T1, with comparison groups performing similarly. At T2 group differences were maintained. For verbal skills differences remained stable. Group 1 exhibited deterioration on non-verbal and processing tasks, while comparison groups showed improved abilities. Group 1 exhibited increases in literacy skills, with educational intervention predicting progress. Results suggest cumulative deficits in non-verbal and information processing skills for children treated with CRT + chemotherapy, with other deficits remaining relatively stable over time. Improved literacy skills suggest that gains can occur with remediation