Sleep problems in autism: prevalence, cause, and intervention

Amanda L Richdale* PhD, Lecturer in Psychology, Department of Psychology and Intellectual Disability Studies, RMIT, Bundoora, Victoria, Australia, 3083. *Correspondence to author at above address.

Autism, Aspergers disorder (AD), and pervasive developmental disorder not otherwise specied (PDDNOS) are commonly referred to as autistic spectrum disorders (ASD)1 and are classied as pervasive developmental disorders (PDD) in the fourth edition of the Diagnostic and Statistical Manual on Mental Disorders (DSM-IV)2. Deviant and delayed development in social and communication skills and the presence of restricted routines and interests, and stereotypic behaviours are variously present in ASD2,3. The majority of children with autism have an intellectual disability (ID) while those with AD do not2. Sleep problems are among a number of secondary behavioural difculties which may occur in children with autism2, but little is known about their occurrence in AD or PDDNOS. Seigel4 comments that autism is not uncommonly related to disturbances of sleep (p 292). Sleep problems are reported to differentiate young children with autism from those with other disabilities, particularly retrospectively5,6. Nevertheless, Rapin and Katzman7 comment that the Study of the prevalent and troublesome sleep disorders of autism has barely started (p 8). Additionally, studies of sleep difculties in children with an ID indicate that such problems can be a source of stress for families8, particularly when children have autism9. This paper reviews sleep problems in children with autism and addresses the issues of aetiology and intervention. Reference to AD and PDDNOS is also made. To set the nature of these sleep difculties in context, brief reviews of sleep problems in typically developing children and children with an ID are provided. Sleep problems in typically developing children The establishment of a mature sleepwake rhythm is a developmental phenomenon. The infant moves from the polyphasic pattern of the newborn infant to a longer night sleep with two daytime naps around 3 months of age, gradually reducing to one nap per day during the latter part of the rst year. By 3 to 4 years the daytime nap is abandoned. Factors which entrain the sleepwake rhythm include the lightdark cycle, and regular events in the childs environment related to feeding and social activity. Sleep problems are common in childhood and may be dened as a sleep behaviour that is

disturbing in some way to the child, the childs family, or both; and is distinct from a sleep disorder which implies an underlying abnormal physiological function10. There are a range of sleep problems which occur in children: settling difculties and night waking are common in infancy and the preschool years, with around 30% or more of children in this age group reported to have problems11,12. During this period, nightmares, confusional arousals, and night terrors also begin13. During middle childhood, sleep improves considerably, with only a small percentage of children still experiencing or developing sleeping difculties14,15. Studies suggest that difcult temperament, fearfulness, and anxiety may also be associated with sleep problems14,16,17. Usually, behavioural interventions13,18, or progressive rescheduling of the sleepwake cycle19 can be successfully used to treat sleep problems in typically developing children such as settling difculties and night waking. However, there is little research on treatment of the parasomnias13. There are a number of recent reviews10,20,21 of sleep problems in children and their treatment. Sleep problems in children with an ID There is still little research concerning sleep difculties in children with developmental disabilities. However, while the available literature is considerably less than for typically developing children, there are a number of recent studies which document the frequency and type of problems found8,2326. In her review, Johnson22 reported that 34% to 80% of children with an ID have a sleep problem, suggesting that such children are likely to have unique factors (p 674) which contribute to their sleep problems. As for typically developing children, problems are more common at younger ages8,23,24. The majority of sleep studies in children with an ID consider mixed-disorder groups and the cause of the disability is known for some children, but not others. Typically, studies investigate children with more severe levels of ID. The frequency with which sleep-onset and sleep-maintenance problems occur is considerably higher in children with an ID than in typically developing children, and sleep problems appear to be associated with more difcult and problematic behaviours8,23,24,26 and communication difculties8,24. The latter is

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likely to be important for the comprehension of social cues and daily routines which inuence the establishment of an appropriate sleepwake rhythm. Little is known about the parasomnias in these children, but rates appear to be low9,26. Stores27 suggests that sleep problems in children with an ID may vary with aetiology. Studies of sleep problems in specic disorder groups support this notion. For example, those with PraderWilli syndrome suffer from excessive daytime sleepiness, excessive night sleep, and REM-sleep abnormalities28; girls with Rett syndrome have problems with night waking, early waking, short night sleep, and increased daytime napping with problems worsening with age29, and children with Down syndrome suffer from sleep apnea30, with about two-thirds of children exhibiting settling difculties, and around a fth, more severe night-waking problems31. Other groups with severe sleep difculties associated with sleep onset and maintenance include those with tuberous sclerosis32 and Sanlippo syndrome33. There are recent reviews of the literature in relation to sleep difculties associated with specic disorders27,34. The causes of sleep difculties in children with an ID are likely to be related to specic aetiological factors which result in impairment in either or both the control and maintenance of sleep processes. Additionally, behavioural difculties, particularly severe communication problems, are likely to affect the development and maintenance of appropriate sleepwake routines. Thus, as previously dened, sleep disorders and/or sleep problems10 may be present in this group. Sleep problems in children with autism Studies of children with an ID have suggested that sleep problems are associated with autistic behaviours8,23 and that children with autism may suffer from sleep problems more frequently than other groups of children with an ID26,35. Rates of 56%23, 65%36, and 68%26 for sleep difculties in children with autism have been reported, with more variable sleep patterns when compared with typically developing children36. Reported rates of occurrence for sleep problems in other specic disability groups have ranged from 40 to 58%26 to 44 to 71%8. In two separate studies, groups containing children with mixed diagnoses exhibited sleep problems at rates of 31%26 and 83%8. However, while there were no children with autism in the former group, it is not known whether any children with autism were in the latter. Patzold et al.37and Richdale and Prior38 found that between 44% and 83% of children with autism had a sleep problem. Rates differed according to whether the problem was reported to be current or past. Altogether, 89% of a group of higher-functioning children had a current sleep problem and/or had one in the past38. Comparing rates of sleep problems within disability groups, sleep problems occur in autism as much as or more than those reported for other groups, particularly children with Down syndrome. The high rate of reported sleep problems in children with autism appears to occur at all IQ levels, including those who do not have an ID3638 and those with AD who appear to have sleep problems which are qualitatively similar to those in children with autism37. Also, given that AD has only recently been recognized in the DSM-IV2 it is likely that some of the higher-functioning children with autism included in earlier studies would now be given a diagnosis of AD. Studies which

include children with PDDNOS have not been reported, although it was found that sleep problems in a group of children with PDD, but no diagnosis of autism, did not differ from those of children with autism35. In other disability groups, children with sleep problems are typically those with more severe levels of ID, although the preliminary results of Richdale et al.9 suggested that, in general, sleep problems in children with an ID were not signicantly associated with level of ID. The question as to whether the lack of a relation between sleep problems and intellectual ability in autism is due to factor(s) specic to the disorder cannot be answered at present as there appears to be little reported concerning sleep problems in those children with mild to moderate levels of ID and other disabilities. Patzold et al.37 reported no substantial relations between sleep and IQ in either their autism or control groups, but analysis of their data by IQ group alone showed that children with a low IQ were more likely to exhibit night waking. Studies of sleep in children with autism have generally reported severe problems associated with sleep onset and maintenance. Irregular sleepwake patterns, problems with sleep onset, poor sleep, early waking, and poor sleep routines have been found at all developmental levels, with increasing severity at lower developmental levels36. Additionally, shortened night sleep, alterations in sleep onset and wake times, night waking39,40, and irregular sleep patterns (with the presence of a free-running rhythm in one case40 ) have been reported. In a recent sleep diary study, children with autism particularly under 8 years of age were likely to exhibit severe sleep problems, including long sleep latencies, night waking, early-morning waking, and shortened night sleep. These difculties improved with age, but older children still slept less at night and tended to have long sleep latencies. Those with higher IQ had more severe current sleep problems38. In a follow-up study37 similar difculties were found, with the exception of early-morning waking, though the children with autism were more likely to wake spontaneously in the morning. Sleep difculties were not related to IQ, and while there were some improvements with age, older children still exhibited sleep problems. Similarly, using a sleep questionnaire, Schreck35 found a greater frequency of dyssomnias in children with autism, as compared with children with an ID, children with specic developmental problems, and typical children. However, reduced sleep length was not found. This conict in ndings may be due to differences in methodology between the latter and two diary studies. Unusual sleep routines may also be disruptive in that sleep problems are likely to occur when the conditions for the routine are not met. Unusual routines for settling to sleep have been noted in children with autism41. A more specic investigation showed that the children with autism had unusual and problematic sleep routines compared with a control group of children, though the frequency of sleep routines was similar for both groups37. There is little data concerning the parasomnias in autism. Inclusion of questions relating to sleepwalking, sleeptalking, and nightmares in two diary studies indicated that the incidence of these problems was low, and did not differ from those of children in comparison groups37,41. Conversely, Schreck35 included questions specic to the parasomnias and found that children with autism were more likely to

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exhibit parasomnias than were other children in her study. In particular nightmare behaviours, sleepwalking, and bruxism were reported to be increased. As for children with an ID, more difcult daytime behaviours have been reported to be associated with sleep problems in children with autism36. Richdale and Prior38 reported that while there were no correlations between difcult behaviour and sleep problems, a current sleep difculty was associated with more energetic and excited behaviour during the day. However, Patzold et al.3 found signicant associations between more problematic daytime behaviour as measured on the Developmental Behaviour Checklist (DBC)42 and sleep problems, while in their comparison group, a higher score on the autistic relating scale of the DBC was associated with a shorter night-sleep length. Schreck35 reported that both abnormalities of communication and social interaction were related to the presence of parasomnias while, more specically, apnea and bruxism were related to communication problems. It appears that children with autism have a specic constellation of sleep problems, particularly in relation to onset and maintenance. Problems occur at a high frequency and are more severe than those found in typically developing children and many children with other developmental disabilities. There are also associations between problematic behaviours and sleep problems, and more energetic daytime behaviour and sleep problems. Sleep difculties occur at all levels of intellectual functioning. These ndings suggest that sleep difculties are related to some particular decits found in children with autism, rather than to an impairment in intellectual functioning per se. Aetiology of sleep problems in autism The cause(s) of these sleep problems is not known. Associations between sleep disturbances and: (1) difcult daytime behaviour; and (2) communication difculties, suggest that these may be fruitful areas of inquiry. However, these two factors are probably interrelated. Sleep studies also refer to overactive behaviour, waking spontaneously in the morning, and being energetic, together with evidence of less night sleep in children with autism. These behaviour descriptions do not appear to be consistent with the usual effects of sleep loss, which may be expected to result in lowered energy levels and irritable behaviours. However, there may be some relation between poor routines, including sleep, and difcult behaviours. A relation between social and communication difculties and sleep problems is possible35,38,41. The sleepwake cycle is a circadian rhythm and there is evidence to suggest that, as well as the lightdark cycle, humans use social cues to entrain circadian rhythms43,44. Routine and social cues are thought to help young infants develop stable sleepwake patterns with the longest sleep occurring during the night hours. Children with a primary social-communication decit may therefore nd it difcult to use such cues to entrain their rhythms, resulting in problems with their sleepwake schedule. A similar argument is also put forward by Johnson22. For a subgroup of children with autism, the underlying cause of the sleep difculties may be related to production of melatonin. Melatonins major physiological role relates to the synchronization of bodily rhythms to photoperiodic information45. It is thought to assist in phase-setting the cir-

cadian pacemaker46, and is also important in the regulation of sleep45,47. The association between the sleep and melatonin rhythms, and the relation between alterations in the synchronization of the melatonin rhythm and the presence of sleep problems have been noted47. There are recent reviews regarding melatonin and its relation to sleep and psychiatric disorders45,47. Synthesis of melatonin begins with the essential amino acid tryptophan, reported by some to be either elevated48 or reduced49 in children with autism. The neurotransmitter serotonin is generally reported to be elevated in about a quarter7 to a third or more50 of children with autism and is further along this synthetic pathway. However, the relation between altered blood levels of these compounds and brain synthesis of serotonin or melatonin respectively has yet to be clearly established. While Chamberlain and Herman52 rst suggested that melatonin regulation may be abnormal in autism, there have been few studies of melatonin levels. Daytime elevation53, decreased amplitude54, and lack of night-time elevation55 have been reported. Children in the latter study were also reported to have sleep difculties. Alterations in the melatonin rhythm may be responsible for sleep-onset and maintenance difculties in autism, with the speculation that those with sleep-onset problems may have a melatonin rhythm which peaks later in the night, while reduced rhythm amplitude may be related to night waking and early-morning waking41. Others have also suggested that abnormalities in the melatonin rhythm may be related to the sleep problems found in children with autism7,37. The issue of melatonin regulation and its possible relation to sleep disturbance in children with autism certainly warrants further investigation. While melatonin is also postulated to assist in synchronizing other rhythms to the lightdark cycle, there is no evidence to support a general problem with the synchronization of circadian rhythms in children with autism41,51. However, sleep problems in autism may be related to a greater sensitivity to changing photoperiod, with sleep problems and sleep length changing in relation to the seasons. Thus it would be hypothesized that sleep length would be longer in the winter months37,41. Such an hypothesis may also explain anecdotally reported periodicity in behaviours, including sleep difculties, in children with autism. This has yet to be tested. Given that many children with autism wake spontaneously and sleep less compared with other children, and given the associations between daytime activity and problematic sleep, a further hypothesis regarding sleep difculties may be a relation with arousal factors. Early literature on children with autism suggested that they may be either hyper- or hypoaroused5658 and stereotypical behaviours are hypothesized to serve an arousal function. Problems with sleep onset and maintenance may therefore relate to arousal factors38, with a need for less sleep related to heightened arousal41. A relation between disturbed night sleep and the need to alter stimulation through stereotypical behaviour has also been suggested22. Anxiety is a prominent feature for many children with autism and autobiographical reports include feelings of fear and anxiety59,60. Anxiety may cause insomnia in both children and adults10 and adversely affect sleep61. Children with autism have been found to have signicantly higher DBC anxiety scores than comparison children37 and a relation

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between psychosocial factors and sleep problems in children with an ID has been suggested27. Therefore, fears and anxiety may also signicantly contribute to sleep problems in children with autism, particularly in more-able children who may be more likely to think deeply about any fears. A further area of interest with regard to sleep difculties in children with autism is the sleep EEG, but studies are limited. Ornitz et al.62 reported that there were no EEG abnormalities, but later studies suggested an immaturity in the EEG pattern of REM-sleep episodes and eye movements63,64, with immaturity in sleep spindles in non-REM sleep63. More recently Elia et al.65 reported a higher REM density with fewer REM clusters in a group of adolescents with autism. These ndings suggest that there may be developmental differences in sleep EEG in children with autism. Considered together with the known sleep problems, further EEG studies appear warranted. Sleep EEG measurements in conjunction with melatonin administration would be of particular interest. Melatonin has been reported to increase stage-2 sleep, decrease stage-3 and -4 sleep, and to alter REM-sleep latency in adults without autism66. A nal area of interest is the relation between brain pathology and sleep problems in children with autism. The site of any brain pathology in autism has not been denitively identied. Reviews regarding brain pathology can be found in Bauman and Kemper67. Some recent studies have suggested that abnormalities may occur in the cerebellar vermis68, the limbic system69,70, and in the Purkinje cells of the cerebellum69,70. Any relation between these putative sites and sleep problems appears to be unexplored, and speculation is beyond the scope of this review. Intervention for sleep problems While we may still only speculate about the cause, there is now sufcient evidence to show that children with autism are highly likely to suffer from severe sleep disturbances. Sleep disturbances are also stressful for families8 and should not be viewed as an inevitable consequence of autism. Effective intervention strategies are required. Thus the presence of sleep problems must be perceived by both clinicians and researchers as an important and potentially stressful behaviour which requires intervention. Wiggs and Stores71 reported that less than half of parents of children with a disability and a sleep disorder received help. Medication was the most prevalent form of help, though parents viewed behavioural interventions as more helpful. Just over half of the parents wanted help; the remainder had declined for reasons unspecied. Interventions may be based upon techniques which are known to be effective in other groups of children, but should also be informed by an analysis of the sleep behaviour and the postulated cause(s) of the problem. Interventions which should be explored include: (1) behavioural interventions; and (2) interventions related to the circadian regulation of the sleepwake cycle, including melatonin, light therapy, and chronotherapy. No sleep intervention should begin without appropriate and thorough history-taking. One requires both parental description of the problem and baseline data in the form of a diary kept for a minimum of 1 week. As parents typically remember the worst aspect of the childs sleep, a history alone is unlikely to determine sufciently the nature and extent of the problem10. Our data have shown that while chil-

dren with autism have, both by description and diary report, severe sleep problems, there is considerable inter- and intraindividual variation37,38. Some parents also anecdotally describe what appear to be cyclical changes in sleep. Thus, before appropriate interventions can be formulated one may need to collect data both at the time of presentation, and at a later period, to determine what factors may precipitate cycles of poor sleep. Behavioural interventions have been shown to be effective in treating daytime behavioural difculties in children with autism72,73 but reports of the effectiveness of such interventions for sleep problems are rare and large-scale studies do not appear to exist. Some authors have suggested that behavioural interventions are effective for children with a disability and sleep problems10,19, including those with autism22. Families of children with an ID and sleep problems nd behavioural interventions preferable to medication71, and effective and acceptable74. Strict and appropriate bedtime routines also appear to be important in the establishment of less problematic sleep patterns in children with autism37,38. In some cases, medication may be required in conjunction with behavioural intervention18. Behavioural programmes for sleep problems in children are typically based upon parents developing appropriate bedtime routines for their children and ignoring their childs cries for attention in a systematic way. The choice and effectiveness of the various approaches will be subject to individual child and family variables18. There are two single case-study reports of successful behavioural interventions for sleep problems in autism75,76. More recently, a comparison of two behavioural interventions for sleep problems in a group of 14 children, most of whom had severe or profound levels of ID and other behavioural problems, included three children with autism77. One group of seven children, including two with autism, was treated using a faded bedtime (gradually changing bedtime) with response cost (removal from bed to prevent sleeping) and signicant improvements were found in ve children. One child with autism showed the most improvement in sleep, while the other child showed little improvement. The second group of seven children included one child with autism. They were treated using bedtime scheduling, but improvement in sleep was generally small to minimal. The child with autism was described as improving slightly after treatment (p 417). Thus faded bedtime with response cost was more successful in reducing sleep problems in children with an ID than was a bedtime-scheduling procedure. The authors hypothesized that the former method was superior because it used both classical and operant conditioning, as well as gradual adjustment of the sleepwake rhythm to appropriate hours of the day. A parent-training programme for parents of young children with autism and sleep problems has been investigated78. Four families began the programme, with one family remaining at follow-up. Over a 6-week period, the programme taught parents how to monitor behaviour and evaluate their childs progress, give effective instructions to their child, reinforce appropriate sleep behaviours, decrease inappropriate behaviours; and provide partner support. Progress was monitored throughout. Improvements in childrens sleep were reported during the programme. For the family and their 3-year-old child completing the programme

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and evaluation, reduction in parent stress, signicant improvements in bedtime routine, a reduction of night-time disturbances, and an increase of about 1 hour in night sleep was found. Signs of increases in night-sleep length were also noted in two other children (aged 3 and 4 years). However, the children still slept less than the average 3- to 4-year-old. Daytime compliance was also investigated for three of the four children in the programme and signicant increases were found79. It was hypothesized that this may be related to improvements in night-time sleep. Two of three mothers also transferred their behaviour management skills to other situations at home and this may also have accounted for improvements in compliance. These results illustrate the potential usefulness of a behavioural programme which may alleviate not only sleep problems but may also transfer across settings, resulting in general improvements in both behaviour and parental management skills. Although restricted, the data suggest that behavioural interventions can be successful in treating sleep problems in children with autism. No single behavioural approach is likely to be the most appropriate for these children, rather the intervention needs to be tailored to the individual child. Other factors which need to be taken into account are the age and developmental level of the child. No child with autism referred to in these behavioural studies was older than 8 years and the lowest functioning child had a profound level of disability. Age and intellectual ability may impact on the efcacy of particular behavioural interventions but this possibility has not been investigated. Additionally, the suggested improvements in parenting skills and daytime compliance79 also require investigation. Melatonin administration prior to bedtime has been shown to be helpful in specic circadian-rhythm disturbances, and has been widely used to alleviate sleep problems due to shift work and jet lag, and for adult sleep disorders45,47. It has been shown to induce sleep and alter sleep architecture in adults without autism66 and to entrain the sleepwake rhythm of blind subjects with free-running sleepwake cycles80. Additionally, administration of melatonin to children with severe sleep disorders, along with other severe disabilities (including autism), has been reported to help resolve sleep problems81,82. Typically melatonin is given orally just prior to desired sleep onset. Further studies regarding the efcacy and safety of melatonin for sleep problems in children with autism or other disabilities are required. Two additional treatments for sleep disorders which involve adjustment of the circadian sleepwake cycle, are light therapy and chronotherapy. Light therapy may be used to treat a variety of rhythm problems, including sleep problems21,83: bright light suppresses the secretion of melatonin.. Additionally, it has been shown that periods of bright light treatment in the morning will advance the melatonin and sleepwake rhythms, while bright light treatment in the evening has a delaying effect45,47,84. Thus its mechanism of action in relation to sleep problems is most likely via alterations to the melatonin rhythm and sleep onset. Morning and midday phototherapy, together with strict daytime and bedtime routines, was successfully used to treat ve of 14 young children with severe brain damage and intractable sleep problems, and at long-term follow-up, several years later, sleep had not deteriorated85. It was concluded that this treatment was a useful option when others had failed.

Chronotherapy is a procedure where the sleepwake cycle is successively phase delayed until the desired bedtime is reached. Typically it is used to treat individuals, including children and adolescents, with delayed sleep-phase syndrome21. Recently chronotherapy was reported to be successful in the treatment of severe sleep problems in an 8-year-old girl with severe ID and autism86. Average night sleep increased and the girl was still sleeping appropriately at a 4-month follow-up. Both light therapy and chronotherapy are non-invasive and are promising interventions in the treatment of severe and intractable sleep problems in children with autism. They deserve further consideration, particularly in severe cases where more traditional interventions have failed. Conclusion Children with autism frequently suffer from severe sleep difculties of unknown origin which should not be dismissed as an inevitable consequence of the disorder. Sleep problems may be related to deviant and delayed social and communication skills, adherence to routines, fear and anxiety, or to an abnormality in the secretion of melatonin. The issue of brain pathology and sleep disturbances remains unexplored and EEG studies are few. Nevertheless, careful history-taking and baseline measures of sleep should enable the clinician to formulate appropriate interventions. Behavioural interventions can be very helpful and should be the rst choice. In intractable cases, medication may be necessary as an additional treatment, or administration of melatonin, or light therapy, or chronotherapy in conjunction with behavioural measures may be required. Clearly more research regarding both cause and intervention is required concerning sleep problems in autism. The issue of sleep problems in the other autism spectrum disorders, AD, and PDDNOS has yet to be addressed. However, indications are that children with AD have sleep problems which do not differ qualitatively from those of children with autism37.
Accepted for publication 28th May 1998. Acknowledgement An earlier version of this paper was presented as a keynote paper at Encuentro Mundial de Educacin Especial, Cancn, Mexico, May, 1997.

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Annotation 65

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