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

Transcription

1 Behavioural treatment to reduce sleep problems in children with autism or fragile X syndrome Sophie Weiskop DPsych; Amanda Richdale* PhD; Jan Matthews MEd, Psychology and Disability Studies, RMIT University, Bundoora, Australia. *Correspondence to second author at Psychology and Disability Studies, School of Health Sciences, RMIT University, PO Box, Bundoora, Victoria 8, Australia. amanda.richdale@rmit.edu.au 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 y mo; age range y mo to y mo) and their parents; and seven children with FXS (six males, one female; mean age y 9mo; age range y mo to 9y mo) 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. Parent-reported sleep problems commonly occur in children with an intellectual or developmental disability (Didden et al. ). Some researchers also argue that variability in sleep difficulties is related to the disorder underlying the child s disability (Wiggs and Stores 99, Richdale 999). Children with fragile X syndrome (FXS) and those with autism suffer from sleep onset and maintenance difficulties (Patzold et al. 998, Richdale ); these problems are particularly common in children with autism, about two-thirds of whom are likely to experience some type of sleep disturbance during childhood (Richdale ). Sleep problems in these children and others with an intellectual or developmental disability are stressful for parents and are associated with child behaviour problems (Richdale et al., Didden et al. ). One issue associated with identifying sleep problems in these children is the lack of a common definition in this field. It is agreed, however, that sleep problems generally fall into one of four categories: dyssomnias, parasomnias, circadian sleep disturbances, or excessive daytime sleepiness (Stores ). It is also agreed that these problems may be extrinsically or intrinsically maintained. Parents most commonly report dyssomnias (i.e. problems associated with sleep onset or maintenance; Quine, Didden et al. ). As it is parents who are likely to bring the child for a professional consultation about sleep difficulties, another way of viewing sleep problems is in relation to parental perception of the child s sleep (Ferber 99). That is, if the child s parents perceive that there is a sleep problem, a problem exists, though it may be related to parents expectations or a behavioural issue surrounding sleep, rather than sleep physiology. Despite the common occurrence of sleep problems in children with a developmental disability, systematic and controlled investigations of treatment options and adequate clinical advice for families are both needed (Wiggs and Stores 99, Robinson and Richdale ). Several authors provide clinical guidance (Durand 998, Stores ), and Lancioni et al. (999) and Wiggs and France () provide reviews of recent literature. However, these serve to underscore the lack of controlled intervention research and long-term on the efficacy, effectiveness, and acceptability of sleep interventions for these children. In general, evidence suggests that parents do not like to use drugs to treat their children s sleep problems and that drugs may have undesired effects (Bramble 99). Although there are several reports on the efficacy of behavioural interventions for young, typically-developing children, particularly for settling and night waking (Mindell 999), little has been published on their usefulness and applicability to children with developmental disabilities. There are several approaches to behavioural intervention, the most common being extinction (removing reinforcement to reduce a behaviour) and various forms of graduated extinction. Extinction meets criteria for efficacious treatment of settling and night waking problems in healthy children (Mindell 999). Lancioni et al. (999) suggested that graduated extinction approaches were potentially useful for children with an intellectual disability, but that extinction may be too stressful for many children and their families, whereas Wiggs and France () noted that extinction may not be suitable for children with a physical illness or self-injurious behaviours. There are now single-case studies (Didden et al. 998, Curfs et al. 999, Weiskop et al. ) and group studies (Bramble 9 Developmental Medicine & Child Neurology, : 9

2 99, Thackeray and Richdale ) showing that extinction is both rapid and effective for treating sleep onset and maintenance difficulties in children with a range of disabilities. Also, parents find extinction to be an effective and acceptable approach to intervention (Bramble 99, Weiskop et al., Thackeray and Richdale ) with treatment gains being maintained for at least and up to 8 months postintervention (Bramble 99, Weiskop et al., Thackeray and Richdale ). Thus, there is a lack of controlled studies about the effectiveness of extinction for the treatment of sleep problems in children with an intellectual or developmental disability, and a lack of attention to the potential impact of the disability itself on treatment effectiveness or long-term of treatment gains. Furthermore, although there are claims that extinction may be both stressful and unacceptable to parents, few researchers have tested this. Therefore, the aim of the current study was to assess the effectiveness of extinction for treating parent-referred sleep onset and maintenance difficulties in young children with an autism spectrum disorder (ASD) or FXS, by using a concurrent, multiple baseline design. In addition, both the maintenance of any treatment gains at and months postintervention and the social validity of the intervention, were examined. Method PARTICIPANTS Ten males and three females (mean age y mo; age range y mo to 9y mo) from families participated across two studies: six families (participants to ) with a child with an ASD (five with autism, one with Asperger syndrome) participated in study ; seven families (participants to ) with a child with FXS (five full mutation, two pre-mutation; none comorbid for autism) participated in study. The children s mean adaptive behaviour score (Vineland Adaptive Behavior Scale Survey Form; Sparrow et al. 98) was (SD.; range to 99) and their mean language age (Pre-School Language Scale; Zimmerman et al. 99) was years months (SD 8 years months; range year months to years month). All children were receiving specialist educational services, except the child with Asperger syndrome who was in a regular school. None of the children with autism was taking medication; five of the seven children with FXS were taking medication for either behaviour management (participants, 8, 9, ) or for behaviour management and the sleep problem (participant ). Medications included fluvoxamine, clonidine hydrochloride, dexamphetamine, methylphenidate, and risperidone. All the children except one with FXS lived in two-parent families and were from metropolitan Melbourne, Australia, or its near environs. Apart from four fathers from the FXS group, all parents participated in the parent-training programme. Mothers ages ranged from to years (mean ), whereas the fathers ranged from 8 to years (mean 8). Four mothers worked outside the home (one full-time) and all the fathers except one were in full-time employment. Two children from each study had no siblings, and one family from each study withdrew before completing the intervention. Participants were recruited through an advertisement in a disability newsletter or by referral from their medical practitioner. Criteria for inclusion were that: () the parents perceived their child to be experiencing difficulty settling, bed refusal, night waking, co-sleeping (i.e. a child sleeping in the same bed as one or both parents during the night), or early morning waking; () the child was previously diagnosed with an ASD or FXS; and () the child was not diagnosed with epilepsy. Also, children in study were aged between years months and years, and were not taking medication for either sleep problems or daytime behaviour management. Because of difficulty recruiting children with FXS, the age range was broadened and the medication criteria were waived for study. Age was chosen taking into account that sleep problems are more common in younger children, and the suitability of the intervention for this age range. MATERIALS An interview schedule, modified from Murphy et al. (98), was used to collect detailed information about the child s sleep problem (its history and possible initiating and maintaining variables), as well as additional information about family background, and medical, developmental, and educational history. A sleep diary, developed by the researchers and based on considerations from the literature (e.g. Ferber 98, Patzold et al. 998), was employed to keep a daily record of each child s bedtime and sleep behaviour. Variables including behaviour, lights out, sleep onset, night waking, co-sleeping, and morning wake time were recorded by parents each day during baseline, intervention, and periods. The Goal Achievement Scale (GAS; Hudson et al. 99) was used to assess the clinical significance of any changes in each child s sleep behaviour and to provide a quantifiable measure of programme success. The GAS was based on parent-stated goals: a separate GAS was developed for each sleep behaviour identified by parents as a goal for behaviour change. For each behaviour parents wanted to change, % success (i.e. no improvement) was set as the baseline rate of that behaviour. Before intervention a decision was made as to what constituted % (total) success for each goal. Parents made this decision with the guidance of the therapist (SW). Total success did not have to mean total elimination of the problematic behaviour, but represented the level of improvement parents thought would make a difference to their lives, and which was developmentally appropriate. At the end of intervention, the change in each problematic behaviour was expressed as a percentage of success over baseline. Further evidence for the social validity of the intervention was ascertained by using a modified version of Griffin and Hudson s (98) Program Evaluation Questionnaire. This consisted of three open-ended questions: what the parents liked best and least about the programme and what they would change. The fourth question asked if their child currently had a sleep problem and to rate the severity. There were then five items rated on a five-point Likert scale: parental approval of the techniques; improvements seen in the child s sleep and general behaviour; reduction in parental stress levels; and how strongly the parents would recommend the programme to a friend. Scores on the three items related to sleep and the intervention programme (approval of techniques, willingness to recommend to a friend, and perceived improvement in sleep) were combined to give an overall measure of participant satisfaction (maximum score ). EXPERIMENTAL DESIGN AND PHASES A concurrent multiple baseline design across participants was used. Baseline length varied and lasted for three weeks (cell ), Behavioural Treatment to Reduce Sleep Problems Sophie Weiskop et al. 9

3 four weeks (cell ), or five weeks (cell ). Each child was paired with another child from the same study, and the pair was assigned to one of the three cells. In study, where there were seven participants, one cell contained three participants. Participants were paired to prevent families having to wait too long before treatment began and so that if one family dropped out, there was still a child in that cell of the multiple baseline. Intervention was introduced for cell after weeks, whereas baseline conditions remained in effect for the other two cells. Intervention began after four weeks for cell, and after five weeks for cell. Following baseline, every participant received the same intervention for a minimum of seven weeks. Data collection extended beyond seven weeks in cases where there was interruption to intervention such as illness. Three months after intervention ended, a two week was conducted for both studies. Twelve months after intervention ended, another two week was conducted for study. Because of time constraints a month was not conducted for study. PROCEDURE Assessment before intervention Session was an interview conducted at the RMIT University Psychology Clinic with both parents and the target child present if possible. Using the interview schedule, detailed information was collected about the child and his/her family. Parents were also taught how to complete the sleep diary. However, they were given no instruction as to how to manage their child s sleep problem and were asked to continue responding to the child as they had done before becoming involved with the study. During baseline, the therapist telephoned the parents once a week to check and collect the sleep diary data. Functional assessment When designing the intervention programme, general hypotheses were developed about maintenance of sleep problems in young children. Then, after the interview and at least two weeks of baseline data, a functional assessment was conducted to formulate hypotheses about the determinants of each child s specific problematic sleep behaviours. It also assisted in explaining to parents the rationale for certain intervention techniques and in providing training exemplars. The data suggested that for all the participants where night waking was a problem, the child went to sleep under inappropriate stimulus control in that they required certain conditions (e.g. bottle of milk, mother s presence, or being on the couch) to fall asleep. In some cases, the stimuli events routinely occurred. These children learned to associate falling asleep with a specific stimulus and thus when they woke during the night, the conditions needed to be re-established for them to resume sleep. In the other cases where disruptive settling behaviour was an issue, the stimulus conditions did not occur all the time and these children engaged in disruptive behaviour at bedtime because of the lack of cues that bedtime was approaching. In one case, it seemed that bedtime was set too early in relation to the amount of sleep required and thus a natural stimulus for falling asleep (i.e. tiredness) was absent. The functional assessment suggested that in all instances, aversive child behaviours were positively reinforced by parent responses and in each case the parent s behaviour (e.g. attending to or lying with the child, or allowing the child to co-sleep, or stay up later and fall asleep on the couch) was negatively reinforced by the termination or prevention of the disruptive child behaviour. The only aversive child behaviour that seemed not to be positively reinforced by a parent response was rocking (participant ). The functional assessment suggested that this behaviour was either a self-stimulatory behaviour that was intrinsically reinforced, or that the child was in fact asleep while rocking and that the rocking was a parasomnia. Given the constraints of the study, the cause of this behaviour was never established and the parents were informed that the rocking might not improve with the planned intervention programme as parasomnias were not a target of intervention. Intervention After the baseline period, the parents attended three weekly, individual parent-training sessions (sessions two to four), without their child, if possible. The first training session (session ) was conducted in the families homes and began with goal setting. Parents stated between one and five goals and they were then assisted to make a decision as to what would constitute % (total) success for each goal. Next, the parents were taught basic principles of learning theory; that is, how antecedents and consequences affect child behaviour. Then a bedtime was set for the child, based on his/her average sleep requirements (collected from sleep diary data) and consideration of the child s average sleep requirements and the time that he/she was expected to rise. A bedtime routine was then established. In consultation with the therapist, parents decided on bedtime routine components or activities that were to occur every night at the same time and in the same order. The therapist advised parents to choose quiet activities before bed, to draw a distinction between the purpose of the bedroom for sleep and not for play, and to conduct these activities in the living-room rather than the bedroom. The parents were instructed to teach the routine to the child using modelling with a doll. The intervention incorporated elements to assist with implementation of the bedtime routine. The therapist instructed parents how to use reinforcement procedures such as labelled praise and a reward every time the child complied with a bedtime routine component, and suggested that they could use reinforcement with other child behaviours as well. Parents were also instructed to construct a colourful visual representation of the bedtime routine and place a sticker on the chart every time a component was completed. At the next training session (session ), in the university clinic, parents were taught how to give effective instructions and how to use an abbreviated version of the partner support strategies developed by Sanders and Dadds (99). Partner support strategies facilitate consistency in parenting, and teach communication and problem-solving skills that help partners assist and encourage one another with their parenting tasks. Extinction techniques were taught during the final training session (session ) conducted in the families homes. First, standard extinction was described, then parents were informed about gradual ignoring, and ignoring with parental presence. They were given the choice of these three techniques, but all chose standard extinction. This was also the therapist s preference, and this was communicated to parents with its advantages and disadvantages. 9 Developmental Medicine & Child Neurology, : 9

4 For standard extinction, parents explain the rules to the child, and when the child is in bed, leave the room and ignore all crying or calling out. If the child comes out of his/her room, the parent takes the child back to bed immediately without making eye contact, cuddling, talking to, or yelling at the child. Parents were informed that they may need to do this many times. The same rules applied if the child woke during the night. It was explained that partial awakenings are a normal phenomenon, but many children awake fully because the conditions they associate with falling asleep (e.g. parental presence or a bottle of milk) are no longer present and they need the parent to restore these conditions to resume sleep. The therapist suggested that the child needed to learn to use his/her own resources to fall asleep (i.e. without a bottle in bed or a parent present) both when first put to bed for the night and during the night. If the child complied with the extinction procedure, parents were to reinforce the child in the morning. Regardless of what happened during the night, parents were encouraged to give the child plenty of positive attention during the day. Finally, parents were informed about the possibility of an extinction burst (temporary escalation of targeted behaviour). They were also advised to cease using extinction if the child was in danger or became ill, and to inform neighbours of the extinction programme before using it. In all training sessions, every skill or technique was taught using modelling and role-play, supplemented with written information and parent checklists. Parents were instructed to implement the techniques immediately after the session in which they were taught. At the beginning of each session, the sleep diary and homework (practice strategies taught and complete checklists) were collected and any difficulties or ambiguous entries were discussed. A review session (session ) was conducted at the university clinic five weeks after session. Goals were re-evaluated and the parents were taught how to gradually phase out reinforcers using a variable interval schedule by gradually increasing the number of nights before a reward was provided. Later they phased out the stickers using the same procedure. Parents were also told to stop using the sleep diary, but to continue using the techniques taught. Finally, all mothers completed the Program Evaluation Questionnaire. Throughout intervention the therapist maintained weekly phone contact with the parents and daily contact during the first few nights of implementation of the extinction technique. The phone contact served several purposes: to check progress, obtain data, answer questions, assist with problems parents were experiencing, prompt appropriate behaviour, and to praise parents success. The therapist also encouraged parents to contact her if they had any problems or questions. After session phone contact was gradually reduced. Table I: Parent-stated programme goals for each participant Participants Age, y:m Parent-stated programme goals % success :. Will not disrupt her parents during the night disruptions per week. Will fall asleep without a bottle (at bedtime and during the night) bottles per week :.Will fall asleep on his own, in his own bed nights per week. Will stay in his own bed all night (i.e. no co-sleeping) nights per week :. Will stay in his own bed all night (i.e. no co-sleeping) nights per week. Will stay in his own room until :am mornings per week. Will fall asleep in his own bed nights per week :. Will be in bed by 9:pm nights per week. When instructed to go to bed, will do so without a tantrum nights per week. Will not rock during the night a nights per week :9. Will fall asleep in his own room nights per week. Will be in bed by 9:pm Sunday to Thursday nights per week :. Will engage in fewer pre-sleep disturbances disruptions per week. His sleep latency will decrease Average of min :. Sleep latency will decrease to min nights per week. Will engage in fewer pre-sleep disturbances disruption per week 8 :9. Will engage in fewer pre-sleep disturbances disruptions per week. Will fall asleep alone, in his own bed nights per week. Will stay in own bed all night (i.e. no co-sleeping) nights per week 9 9:. Will engage in fewer pre-sleep disturbances disruptions per week. Sleep latency will decrease to min nights per week :. Will fall asleep alone, in his own bed nights per week. Will not disturb his parents during the night disruptions per week. Will sleep alone all night (i.e. no co-sleeping) nights per week :. Will sleep alone all night (i.e. no co-sleeping) nights per week. Will not disturb his parents during the night disruptions per week :. Will sleep alone all night (i.e. no co-sleeping) nights per week. Will sleep until :am days per week. Will nap less during the day nap per week :. Will engage in fewer pre-sleep disturbances disruptions per week. Will sleep at his own house nights per week a This parent-stated goal was not a target of intervention. Children had autism; child had Asperger syndrome; children had fragile X syndrome. Behavioural Treatment to Reduce Sleep Problems Sophie Weiskop et al. 9

5 All participants received the intervention programme as described. However, the parents goals for the intervention varied (Table I) and there were minor variations to intervention for some participants in sessions,, and. After session, the older brother of participant with whom he shared a bedroom was reinforced for ignoring his sibling s disruptive night-time behaviour using a reward programme, whereas the mother of participant 8 was advised to cease waking him for toileting, and to only take him to the toilet when he woke and requested it. The parents of participant established a new rule that their child stay in his room until am. The parents initially reinforced the child if he did not co-sleep, regardless of what time he left his bedroom in the morning. Then, once he had stopped co-sleeping, the conditions changed so that he was reinforced only if he stayed in his room until am. Goals for participant included reducing both early morning waking and daytime naps. Thus his mother delayed his bedtime as much as possible, ensured his daytime naps occurred in his bedroom, and gradually shortened the naps. She also installed a baby sound monitor because the child s room was far from the parents and the child had severe asthma. For participant 9, bedtime in baseline (approximately :pm) was too early given his average sleep requirements and his morning rise time. A bedtime closer to 9pm was recommended. Participant 9 shared a bedroom with his younger brother who required more sleep, and given that the children were soon to have separate bedrooms as a result of home renovations, the therapist recommended that the younger child fall asleep in another room. Participant also shared a bedroom. At session (when extinction was taught) it was recommended to these parents that they either use a reward programme with the brother (like participant ), or move the older brother out of the room temporarily; they chose the latter. For the four children whose fathers did not participate in training (participants 8,,, ), changes were made to sessions and. In session, partner support strategies were explained, but not role-played or modelled. At session, these mothers were told that if the father assisted with programme implementation, he must do so in the same manner as the mother. If this was not possible, mothers were told it was best the father not be involved with the child s bedtime. The mothers were also instructed to inform the father of the extinction burst before starting the extinction technique. Follow-up Three months after session the sleep diary was mailed to the families and they completed it for weeks. This was repeated at months for study. DATA CODING Sleep diary Sleep diary data were graphed regularly during all phases to monitor progress. Data graphed were: () number of presleep disturbances per week, where pre-sleep disturbance was defined as any disruption occurring between the time that the child was put to bed and the time of sleep onset (e.g. calling out, crying, or leaving room); () number of nights per week that the child fell asleep alone in his/her own bed; () average sleep latency (min) per week, where sleep latency was defined as the number of minutes between first being settled to bed and sleep onset; () number of night wakings per week that parents were aware of; () number of nights per week that the child co-slept. Co-sleeping was not coded when the parents only lay with the child until the child fell asleep at bedtime; and () average duration (min) of night-time sleep per week. When the duration of sleep per night was calculated, the length of night wakings was subtracted if this information Table II: Distribution of ratings when baseline is compared with other phases Phase comparison Number of Rating (%) comparisons Substantial Moderate No change Moderate Substantial deterioration deterioration improvement improvement Baseline vs intervention Baseline vs three month Baseline vs month Table III: Level of success achieved (%) on goals at end of intervention, -, and -month for each participant Participant Goal Goal Goal Intervention mo mo Intervention mo mo Intervention mo mo No data. No data 9..9 No data a b. b a No data for this goal as sleep diary not completed properly. b Data for this goal (reducing daytime naps) is based on retrospective parental report. 98 Developmental Medicine & Child Neurology, : 9

6 was available. These six variables were analyzed for all participants. In some cases, additional child-specific variables were analyzed (e.g. morning wake time and adherence to bedtime). DATA ANALYSIS Given the applied nature of this research, when evaluating the data, emphasis was placed on clinical significance. This was achieved through the GAS as well as considering subjective evaluations; however, the experimental criterion was also considered. Analysis of change in individual participant s sleep data was performed by using visual analysis of graphs. In the interest of brevity the results of the two studies were combined and are presented together here. Visual analysis of graphs For every sleep variable graphed, visual analysis was used to make the following comparisons: baseline versus the last four weeks of the intervention phase (i.e. after the extinction technique was implemented), baseline versus the three month, and baseline versus the month. To determine the reliability of the visual analysis, every graph was rated independently by two clinicians (one of whom was not associated with the study), on a five-point rating scale modified from Hudson et al. (99). Ratings were: substantial improvement, moderate improvement, no change, moderate deterioration, or substantial deterioration. Definitions were provided for each descriptor. Graphs were not labelled, thus the raters did not know which sleep variables they pertained to. However, given that some of the behaviours were desirable and others undesirable, the raters were given an indication of the desired direction of change to determine whether an increase related to an improvement or deterioration. Where there was disagreement, the raters conferred and agreed on a rating. Results Three children were excluded from the analysis. Only baseline data were available for participant (Asperger syndrome) as his mother did not bring the sleep diary to the intervention sessions and in the week that extinction was to be implemented, both the mother and child became ill: as a result the family withdrew from the study. The mother of participant did not complete the intervention or supply data past baseline as she had family issues to attend to. Participant completed the intervention, but illness caused numerous interruptions to intervention and prevented his inclusion within the group analysis. Thus the results described here pertain to participants. For participant, no sleep data were collected in weeks and as the child was on holiday. The parents were also unavailable to collect month s data. Because a b Baseline Intervention month month Baseline Intervention month Participant 8 9 Participant * Number of pre-sleep disturbances per week 8 8 Participant Participant Participant Total pre-sleep disturbances per week Participant 8 Participant 9 Participant 8 9 Participant Participant Figure : Number of pre-sleep disturbances for each child with (a) autism and (b) with FXS during baseline, intervention, and. * No up data available for this participant on this variable because sleep diary was not completed. Behavioural Treatment to Reduce Sleep Problems Sophie Weiskop et al. 99

7 this child slept at his father s house on some weekends (and data were not collected), the data for all sleep variables were pro-rated to facilitate comparisons across weeks and across participants. Data for all sleep variables were also pro-rated for participants 8 and 9 because sleep data were only collected five or six nights per week. In addition, the parents of participant 9 failed to collect any data in the final week of the intervention phase and thus visual analysis was based on the last three weeks that data were collected. Although data were collected in each week for participant, week was disregarded from all analyses as hospitalization and recovery from an adenotonsillectomy affected his sleep during this time. Although sleep data were collected every night for the other participants, there were occasional nights where diaries were not completed fully: where this was the case, data were prorated. The only variables affected by missing data in these cases were sleep latency and sleep duration. ASSESSMENT OF CHANGES IN CHILDREN S SLEEP BEHAVIOURS Overall change in sleep behaviours based on visual analysis A total of graphs were rated and comparisons between phases were made. The two raters agreed on (8%) of the comparisons. When baseline was compared with the end of intervention, of the phase comparisons were given the rating of substantial improvement, 9 were given the rating of moderate improvement, three were given a rating of moderate deterioration, and none was rated as substantial deterioration. Although phase comparisons were given a rating of no change, in seven situations no change was expected because the child never engaged in the behaviour before intervention. Table II shows that when baseline was compared with each phase, the distributions of ratings were very similar to when baseline was compared with the end of intervention. Rate of change of each variable based on visual analysis Seven participants experienced an extinction burst (Fig. a,b) in the week that extinction was implemented (i.e. three weeks after intervention began for each child). For two participants, the frequency of pre-sleep disturbances clearly decreased as soon as intervention began. For all cases, at the end of intervention pre-sleep disturbances were rated as having improved from baseline levels, and in all cases where data were available, the improvement was maintained. a b Baseline Intervention month month Participant Baseline Intervention month Participant Number of nights per week child fell asleep alone and in own bed Participant Participant Participant Number of nights per week child fell asleep alone and in own bed Participant 8 Participant 9 Participant 8 9 Participant Participant 8 9 Figure : (a) Number of nights per week that each child with autism in fell asleep alone and in his/her own bed during baseline, intervention, and. (b) Number of nights per week that each child with fragile X syndrome in fell asleep alone and in his/her own bed during baseline, intervention, and. Developmental Medicine & Child Neurology, : 9

8 The rate at which eight participants fell asleep alone and in their own beds (Fig. a,b) at the end of intervention was judged to have improved. No change was expected for the other two children because of high baseline levels of appropriate behaviour. For one child rated as improved there was an increasing trend in baseline, but in the other children the increase occurred when intervention began or when the extinction technique was implemented. Improvement was maintained at in all but one child. At the end of intervention, sleep latency was rated as improved for six children (although one of these had a decreasing trend in baseline), deteriorated for two children, and not changed for two. All children rated as improved maintained this at, except for one who was now rated as deteriorated. Of the two participants rated as deteriorated at the end of intervention, only one improved at the three month but did not maintain this improvement at months. At the end of intervention and both s, night waking (Fig. a,b) was rated as improved for seven children (although one had a decreasing trend in baseline). No change was noted in the other three participants (participants,, 9); however, no change was expected for participants and 9, as they did not wake at night in baseline. For participant it was unclear whether she was awake or rocking during sleep. Figure a,b shows that before intervention co-sleeping was a problem for six participants. At the end of intervention the frequency of co-sleeping was rated as improved for these six and the change occurred when the extinction technique was implemented. They all maintained their change at three months, but at months one participant (participant ) was rated as deteriorated. Nevertheless, she co-slept very infrequently in baseline (i.e. co-sleeping was not a goal for behaviour change for this child). Average duration of night-time sleep for each participant was variable. In addition, there was little consistency among participants in the rate of change across phases. According to mothers responses to the programme evaluation question Do you consider that your child currently has a sleep problem?, five participants (,,,, ) still had a sleep problem after intervention although in all of these cases except one (participant ) the severity had decreased. SOCIAL VALIDITY Programme evaluation Responses to the open-ended questions on the Program Evaluation Questionnaire indicated that the best aspects of the a 8 Baseline Intervention month month Participant b 8 Baseline Intervention Follow-up Participant Number of night wakings per week Participant Participant Participant Number of night wakings per week Participant 8 Participant 9 Participant Participant 8 Child sick following surgery 8 9 Participant Figure : Number of night wakings per week for (a) each child with autism and (b) each child with fragile X syndrome during baseline, intervention, and. Behavioural Treatment to Reduce Sleep Problems Sophie Weiskop et al.

9 programme were the outcome, the support provided, the phone calls, and the method of instruction. Seven mothers identified record keeping as the least liked, although one acknowledged that it was essential. Two mothers also reported that they did not like sticking to a bedtime routine. One mother said that sessions were too long. Three mothers referred to the overall time consuming nature of the programme. When the three Likert scale items related to sleep and the intervention programme were combined to give a consumer satisfaction score out of, the mothers mean responses ranged from to (mean.8). They all approved of the techniques and noticed at least some improvement in their child s sleep. When asked if they would recommend the programme to a friend, all said yes and seven gave the maximum approval rating. Goal achievement scale Table III presents the level of success achieved for each child on each parent-stated goal at each phase. By the end of the intervention phase, out of goals were achieved with % success and the mean GAS was.%. These results improved further by the three month with a mean GAS of 8.8, but study accounted for this increase. In study, at the level of achievement increased for four goals, but decreased for four also. At the month (study only), the level of achievement either remained constant or increased for all goals except one; the mean GAS was 89%. Discussion The results support the hypothesis that sleep problems of children with autism or FXS would reduce after the behavioural intervention. Of the six common sleep variables, four changed: pre-sleep disturbances, falling asleep alone, night waking, and co-sleeping. For each of these variables, visual analysis showed that for each child who experienced the problem in baseline, there was improvement after intervention. In study, improvements were maintained at the three month and month. In study, most improvements were maintained at the a b Number of nights per week child co-sleeps Baseline Intervention month month Participant Participant Participant Participant b Number of nights per week child co-sleeps Baseline Intervention month Participant Participant 8 Participant 9 Participant 8 9 Participant 8 9 Participant Figure : Number of nights per week that (a) each child with autism and (b) each child with fragile X syndrome co-slept during baseline, intervention, and. Developmental Medicine & Child Neurology, : 9

10 three month. Thus, for these presenting sleep difficulties, the intervention was effective. There was insufficient evidence to support a change in sleep latency or duration. These were the only sleep variables where some individuals outcomes were rated as deteriorations on visual analysis, and where there was a lack of consistent change among participants across phases. Although sleep latency and/or duration did improve for some children, often the improvements were not clinically meaningful because the participant was either already sleeping adequately before intervention, or did not change after intervention. Sleep latency and duration were not goals for most participants; the improvement in the other four sleep variables together with the high degree of success for most goals as measured by the GAS, suggest that sleep problems were reduced. In fact, the GAS may be a better indicator of outcome because it considered the variables that are important for the individual (i.e. it addressed the clinical criterion for success). GAS scores clearly indicated that at least some aspects of each child s sleep problem were considerably improved and that the improvement was maintained. In addition, most parents perceived an improvement in their child s sleep. This is important because it means that changes in sleep were noticed by parents and made a difference to their lives. The high consumer satisfaction ratings also support the conclusion that the children s sleep problems improved. However, little or no improvement occurred for early morning waking or night rocking. Thus extinction was not appropriate for these particular behaviours, probably because they were not positively reinforced by parental responses before intervention. As well, there are two potential levels of treatment impact: behavioural and physiological/biological (Richdale ). Extinction addresses the behavioural components of a sleep problem rather than impacting on sleep physiology. This is consistent with Wiggs and Stores (998) recommendation to distinguish between interventions which lead to objective changes in a child s sleep versus those that do not objectively change sleep but which result in the child no longer disrupting their parents. Although the recorded night wakings decreased, consistent with the findings of Durand et al. (99), night wakings were not totally eliminated in any case. However, this is a normal behaviour experienced by most children (Ferber 98). In most cases, the positive changes in sleep did not occur until extinction was implemented. The sleep behaviour of participant 9 improved with a more appropriate bedtime and bedtime routine and thus extinction was not used. The sleep of participant also improved with the bedtime routine, but data were not correctly recorded for presleep disturbances, and the improvement in sleep latency was not maintained. The 9:pm bedtime for participant also improved with the introduction of the bedtime routine. Otherwise, changes did not occur until extinction was implemented and it caused a temporary increase in pre-sleep disturbances in seven children, two of which were extreme, confirming that extinction has the potential to be a difficult and stressful procedure for parents. Nevertheless, like earlier reports (Bramble 99, Didden et al. 998, Curfs et al. 999, Thackeray and Richdale ), the results demonstrate that an extinction can successfully reduce sleep problems of children with a developmental disability. This study is the first to demonstrate the effectiveness of extinction for sleep problems in children with autism or FXS using a concurrent multiple baseline design. However, the limited number of intervention studies, including extinction, across a range of developmental disabilities still leaves open the broader question of treatment efficacy. Thus research still needs to determine the best approaches to the various sleep problems in autism, FXS, and other developmental disabilities. Extinction took effect quickly, with the difficult behaviour only lasting a few days. By comparison, gradual approaches can take weeks or even months before any change is noticed (Howlin 98, Rodlier and Houton 98). Although it has been suggested that extinction may not be well accepted because it is stressful for the parent and child (Lancioni et al. 999), our parents approved of the techniques used, suggesting that educating parents about the extinction burst and providing support during implementation may prevent parental non-compliance and improve treatment acceptability. Social validity has rarely been considered in previous research examining behavioural interventions for sleep problems in children with a disability, and usually only one aspect, the social importance of the effects (i.e. clinical significance), is addressed. Like previous studies (Bramble 99, Thackeray and Richdale ), every parent approved of the technique used and most said they would recommend it to a friend. In addition, like Thackeray and Richdale (), the current study used an objective measure of clinical significance (i.e. the GAS), whereas others have not directly addressed this issue (Bramble 99, Didden et al. 998). Several methodological issues need to be considered when making conclusions about the effectiveness of this intervention. First, although the use of a multiple baseline counters threats to internal validity, the degree to which the findings can be extended to the wider population is limited. Other conditions need to be tested. For example, similar success may not be met with older children, children with epilepsy, children with Asperger syndrome, or families from non-english-speaking backgrounds. Nevertheless, the intervention was effective for two disability groups with diverse child characteristics (e.g. diagnosis, age, medication, developmental level, and sib-ship size) as well as parent characteristics (e.g. maternal status and level of employment). A further methodological issue relates to the two participants who dropped-out and for whom no sleep or programme evaluation data were obtained. Although reasons were given for withdrawal in both cases, other factors common to these participants, but not present for the others, could explain their early termination. Based on clinical impression, these two children were more non-compliant than the other participants. They were also the oldest (excluding participant 9). Perhaps extinction is too difficult or stressful to implement with extreme non-compliance or older children. The final methodological limitation is the reliance on parental report for the sleep diary. Practical considerations meant that it was not possible to provide reliability data on parents use of the sleep diary, but several past studies have shown good interrater reliability for parent-completed sleep diaries (Adams and Rickert 989, Didden et al. 999). Also, regular telephone support was provided to check on parent s progress, including diary completion. The use of more objective means of data collection (e.g. video or activity monitoring) may enhance future work. This research has important implications for clinicians and service providers as well as future Behavioural Treatment to Reduce Sleep Problems Sophie Weiskop et al.

11 applied research. Standard extinction (with bedtime routine) was effective for the sleep problems of children with autism or FXS, had no reported side effects, and was acceptable to parents. Thus, parents need not accept that sleep problems are an additional burden to be endured. Although modifications of extinction or other behavioural approaches may be more suitable for some clients, clinicians should not simply assume that parents will disapprove of standard extinction. Health professionals should inform these parents that common sleep problems can be ameliorated and then present treatment options, including extinction. As extinction has rapid results, it should be a treatment option if the sleep problem is maintained by parental attention or reinforcement. However, particular care should be taken when there are medical complications such as epilepsy or asthma, and consultation with a medical practitioner is recommended. The acceptance and accurate implementation of extinction by parents may be dependent on their understanding of the extinction burst. Thus clinicians should include education about the extinction burst in their programmes. The current intervention was intensive: administration was individual and involved much therapist support including home visits and phone calls. Given the other significant stressors on families affected by disability, it is likely that this level of support is often required. This entails obvious costs which, given limited community resources, may restrict the provision of this intervention. Further research needs to determine which components of the current intervention package are essential and what level of family support is required. In addition, further research could examine whether the intervention can be delivered to a group with an overall aim of reducing cost without diminishing the impact of intervention. DOI:./S8 Accepted for publication th May. Acknowledgements These two studies formed part of Dr Sophie Weiskop s Doctor of Psychology thesis. A case from study has been previously published (Weiskop et al. ), and preliminary data from study were reported at the Biennial National Autism Conference, Hobart, Australia, th August, 999 and published in that conference s proceedings. References Adams L, Rickert VI. (989) Reducing bedtime tantrums: comparison between positive routines and graduated extinction. Pediatrics 8:. Bramble D. (99) Consumer opinion concerning the treatment of a common sleep problem. Child Care Health Dev :. Bramble D. (99) Rapid-acting treatment for a common sleep problem. Dev Med Child Neurol 9:. Curfs LMG, Didden R, Sikkema SPE, Die-Smulders CEM. (999) Management of sleeping problems in Wolf-Hirschorn syndrome: a case study. Genet Couns :. Didden R, Curfs LMG, Sikkema SPE, de Moor J. (998) Functional assessment and treatment of sleeping problems with developmentally disabled children: six case studies. J Behav Ther Exp Psychiatry 9: 8 9. Didden R, de Moor J, Kruit IW. (999) The effects of extinction in the treatment of sleep problems with a child with a physical disability. Int J Disabil Dev Educ :. Didden R, Korzillius H, van Aperlo B, van Overloop C, de Vries M. () Sleep problems and daytime behaviour problems in children with intellectual disability. J Intellect Disabil Res :. Durand VM. (998) Sleep Better! A Guide to Improving Sleep for Children with Special Needs. Baltimore: Paul Brookes Publishing. Durand VM, Gernert-Dott P, Mapstone E. (99) Treatment of sleep disorders in children with developmental disabilities. Journal of the Association of Persons with Severe Handicap :. Ferber R. (98) Solve Your Child s Sleep Problems: The Complete Practical Guide for Parents. New York: Penguin Books. Ferber R. (99) Childhood sleep disorders. Neurol Clin : 9. Griffin M, Hudson A. (98) Parents as Therapists: The Behavioural Approach. Melbourne: PIT Press. Howlin P. (98) A brief report on the elimination of long-term sleeping problems in a -year-old boy with autism. Behav Psychother :. Hudson A, Wilken P, Jauernig R, Raddler G. (99) Regionally based teams for the treatment of challenging behaviour: a three year outcome study. Behav Change : 9. Lancioni GE, O Reilly MF, Basili G. (999) Review of strategies for treating sleep problems in persons with severe and profound mental retardation or multiple handicaps. Am J Ment Retard : 8. Mindell J. (999) Empirically supported treatments in pediatric psychology: bedtime refusal and night wakings in young children. J Pediatr Psychol : 8. Murphy GC, Hudson AM, King NJ, Remenyi A. (98) An initial interview schedule for use in the behavioural assessment of children s problems. Behav Change :. Patzold LM, Richdale AL, Tonge BJ. (998) An investigation into sleep characteristics of children with autism and Asperger s disorder. J Paediatr Child Health : 8. Quine L. () Sleep problems in primary school children: comparison between mainstream and special school children. Child Care Health Dev :. Richdale AL. (999) Sleep problems in autism: prevalence, cause and intervention. Dev Med Child Neurol :. Richdale AL. () Sleep in autism and Asperger s syndrome. In: Stores G, Wiggs L, editors. Sleep Disturbance in Children and Adolescents with Disorders of Development: Its Significance and Management. London: Mac Keith Press. p 8 9. Richdale AL. () A descriptive analysis of sleep behaviour in children with fragile X. J Intellect Dev Disabil 8:. Richdale A, Gavidia-Payne S, Francis A, Cotton S. () Stress, behaviour, and sleep problems in children with an intellectual disability. J Intellect Dev Disabil :. Robinson AM, Richdale AL. () Sleep problems in children with an intellectual disability: parental perceptions of sleep problems, and views of treatment effectiveness. Child Care Health Dev : 9. Rodlier A, Houton R. (98) Training parents to use extinction to eliminate nighttime crying by gradually increasing criteria for ignoring crying. Educ Treat Children : 9. Sanders MR, Dadds MR. (99) Behavioral Family Intervention. Boston: Allyn & Bacon. Sparrow S, Balla D, Cicchetti D. (98) Vineland Adaptive Behavior Scale Interview Edition. Minnesota: American Guidance Service. Stores G. () A Clinical Guide to Sleep Disorders in Children and Adolescents. Cambridge: Cambridge University Press. Thackeray E, Richdale A. () The behavioural treatment of sleep difficulties in children with an intellectual disability. Behav Intervent :. Weiskop S, Matthews J, Richdale A. () Treatment of sleep problems in a -year-old boy with autism using behavioural principles. Autism : 9. Wiggs L, France K. () Behavioural treatments for sleep problems in children and adolescents with physical illness, psychological problems or intellectual disabilities. Sleep Med Rev : 99. Wiggs L, Stores G. (99) Sleep problems in children with severe intellectual disabilities: What help is being provided? J Appl Res Intellect Disabil 9: 9. Wiggs L, Stores, G. (998) Factors affecting parental reports of the sleep patterns of children with severe learning disabilities. Br J Health Psychol : 9. Zimmerman IL, Steiner VG, Pond RE. (99) The Preschool Language Scale. San Antonio, Texas: Harcourt Brace Jovanovich. Developmental Medicine & Child Neurology, : 9