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Three Part Question

In [children with Downs Syndrome] can [behavioural programmes] work to tackle [sleep difficulties where there is no clear physical cause for the sleep problems]?

Clinical Scenario

A mother brings her 7-year-old son with Down syndrome to clinic complaining of sleep difficulties. He won’t go to sleep alone, frequently wakes in the night and will not be settled unless transferred to his parents’ bed. His parents are exhausted, and his mother believes his lack of sleep is also disrupting his daytime behaviour. He has always been difficult to settle and seldom slept through the night without waking. The child is overweight, but not obese, and upon enquiry his mother tells you that he does not usually snore, or suffer from nocturnal enuresis (bedwetting), which makes obstructive sleep apnoea an unlikely cause. His mother tells you, “I’m sure he’s just waking up out of stubbornness and not because anything’s wrong, but we’re all worn out. I don’t know what to do.” You wonder if a behavioural treatment programme might be able to help in this situation.

Search Strategy

Cochrane Libray, Medline and Psychinfo
“sleep*” and “child*” (Cochrane). “sleep*” AND “developmental disabilities” limit to child and review articles (Medline and Psychinfo)

Search Outcome

Cochrane 2 relevant. Medline 1 relevant

Relevant Paper(s)

Author, date and country	Patient group	Study type (level of evidence)	Outcomes	Key results	Study Weaknesses
Ramchandani et al 2000	9 controlled trials, 132 children randomised to drug treatments, 235 children randomised to psychological treatments. All children aged 5 years and under with a sleeping problem	Systematic review, narrative synthesis (level 1a-)	Parent report (number of night awakenings, time to settle, number of nights disturbed)	Effect sizes not reported. Drug trials showed short term benefits, but no effects were seen at two months follow up. Behavioural treatments produced both short and long term (6-12 weeks), but the loss of control groups at follow up was noted	Conclusions are undermined my poor quality of trials. Drug trials were assessed as of better quality than non-drug trials.
Mindell 1999	39 studies, 1,697 children aged 5 years and under with bedtime refusal or night waking problems	Review, narrative synthesis (level 4)	Parent report (sleep diaries, questionnaires)	Effects sizes not reported. Interventions rated according to the number and quality of trials that showed effects.Extinction was considered ‘well-established’ Graduated extinction & Scheduled awakenings ‘probably efficacious’	No studies were excluded due to poor study methodology.
Lancioni et al 1999	21 studies, 258 young people aged between 4 and 23 years with a range of developmental disabilities with sleeping problems	Review, narrative synthesis (level 4)	Findings dichotomised into improvement or no improvement in sleep pattern	100% of those treated using bedtime fading with or without response cost improved. 82% of those treated using bedtime routine plus gradual distancing of parents improved.	Degree of sleep improvement not assessed.No assessment of study quality was used, nor were any studies excluded on the basis of their methodology.

Comment(s)

Three reviews of evidence on this topic were located. Only one of these reviews searched the grey literature or included non-English language publications 1. The other reviews provided insufficient information on search procedure to be certain that they were systematic 2;3. Reviews of prevalence suggest that occurrence of sleep disorders among children with developmental disabilities is high 4;5. The evidence available would suggest that behavioural interventions are successful for young children without developmental disabilities. Caution should be used when considering the potential impact of intervention on sleep problems due to the heterogeneity of the presenting problems, the subjects used, the changes measured and the programmes implemented. These problems may be exacerbated when using children or infants as subjects. The definition and treatment strategies for a sleep disorder in a 6-month-old baby cannot be assumed to be equivalent to definition and treatment strategies in a 6 year old. In particular, it may be inappropriate to assume that findings from samples of younger, non-disabled children are necessarily applicable in this instance. The review of behavioural interventions for sleep difficulties with young adults and children with learning difficulties would suggest that behavioural interventions are also efficacious in this group. However, due to the shortage of studies in this area, single-case studies and non-controlled studies were included in the review and size of effect could not reliably be assessed. These factors are likely to over-estimate intervention effects. Particular problems with conducting research within this area were highlighted by this review. Treatment ‘overlap’ seemed to be common, for instance studies using Melatonin was used in conjunction with behavioural interventions, making it difficult to attribute cause to the observed effects on sleep. (But see Archimedes from September 2002 for more on this – Ed.) Families prefer behavioural interventions in the first instance. However, the success of such interventions will also depend on the parents’ ability and motivation to implement them. Studies do not appear to have evaluated adherence to behavioural programmes, which may be problematic for interventions that advocate not responding to crying or disruptive behaviour. There is little evidence to favour one behavioural intervention over another 3. Alternative strategies are: Sleep scheduling/scheduled awakening: Altering sleep pattern by instituting fixed bedtime and waking time. Sleep outside of scheduled times is avoided, although fixed naps during the daytime may also scheduled Combination of bedtime routine and gradual distancing from parents: Establishing positive bedtime routines. Parental involvement is gradually reduced, for example sleeping in the same bed is gradually reduced by the parent moving from bed to sitting next to the bed until the child is asleep, then replaced with doll. Bedtime fading with or without response cost: The retraining of bedtime habits by beginning bedtime at a relatively late time when child will fall asleep quickly, then gradually bringing bed time forward until desired bedtime is achieved. Response cost involves the child being taken out of bed for a certain period if he or she doesn’t fall asleep within a specified time. Extinction: bedtime fading and gradual withdrawal of parental involvement. The parents re-enter the room encouraging the child to sleep at intervals of 3-5 minutes, and these intervals are increasingly extended. No evidence was found which would suggest a behavioural intervention would do harm. In comparison, treatment of sleep breathing disorders, in the absence of infection, by physical intervention are invasive treatments with poor evidence for efficacy 6;7 and drug treatments will cause side-effects. Nonetheless, the impact on the child and family of extinctions strategies should be considered.

Clinical Bottom Line

Families prefer behavioural to drug, or surgical interventions for sleep problems in children Behavioural interventions improve children’s sleep patterns, but evidence is insufficient to predict impact Children with developmental delays or disabilities also appear to benefit from behavioural programmes

References

1. Ramchandani P, Wiggs L, Webb V, Stores G. A systematic review of treatments for settling problems and night waking in young children. British Medical Journal 2000;320(7229):209-13.
2. Mindell JA. Empirically supported treatments in pediatric psychology: bedtime refusal and night wakings in young children. Journal of Pediatric Psychology 1999;24(6):465-81.
3. Lancioni GE, O'Reilly MF, Basili G. Review of strategies for treating sleep problems in persons with severe of profound mental retardation or multiple handicaps. American Journal of Mental Retardation 1999;104(2):170-86.
4. Stores G. Children's sleep disorders: modern approaches, developmental effects, and children at special risk. Developmental Medicine and Child Neurology 1999;41(8):568-73.
5. Wiggs, L. Sleep problems in children with developmental disorders. Journal of the Royal Society of Medicine 2001;94(4):177-9.
6. Lim J, McKean M. Adenotonsillectomy for obstructive sleep apnoea in children (Protocol for a Cochrane Review). In: The Cochrane Library, Issue 4, 2002. Oxford: Update Software.
7. Bridgman SA, Dunn KM, Ducharme F. Surgery for obstructive sleep apnoea (Cochrane Review). In: The Cochrane Library, Issue 4, 2002. Oxford: Update Software.