The Efficacy of a Brief Behavioral Sleep Intervention in School-Aged Children With ADHD and Comorbid Autism Spectrum Disorder

Abstract

Objective: Sleep problems are common in children with autism spectrum disorders (ASD) and ADHD and impact adversely on child and parent well-being. The study evaluated the efficacy of a brief behavioral sleep intervention in children with comorbid ADHD–ASD. Method: A subsample of children with ADHD–ASD (n = 61; 5-13 years; 89% male) participating in the Sleeping Sound With ADHD study were included in the current investigation. The subsample comprised of 28 children randomized to the sleep intervention group, while 33 were randomized to usual clinical care. The intervention consisted of two clinical consultations and a follow-up phone call covering sleep hygiene and standardized behavioral strategies. Results: Children with ADHD–ASD who received the intervention had large improvements in sleep problems and moderate improvements in child behavioral functioning 3 and 6 months post-randomization. Conclusion: These findings suggest that a brief behavioral sleep intervention can improve sleep problems in children with ADHD–ASD.

Keywords

ADHD autism spectrum disorders behavioral sleep intervention randomized controlled trial mental health

Autism spectrum disorder (ASD) and ADHD are common neurodevelopmental disorders. ASD, characterized by impaired social, communicative, and stereotypical behavioral patterns, affects approximately 1 in 88 individuals (Baio, 2012). ADHD affects approximately 5% of children and adolescents (Polanczyk, de Lima, Horta, Biederman, & Rohde, 2007) and is characterized by pervasive levels of impulsivity, inattention, and/or hyperactivity (American Psychiatric Association [APA], 2013). ADHD and ASD frequently co-occur; approximately 14% to 78% of children meet criteria for comorbid ASD–ADHD (Holtmann, Bolte, & Poustka, 2007; Keen & Ward, 2004; Lee & Ousley, 2006; Leyfer et al., 2006; Reiersen, Constantino, Volk, & Todd, 2007; Sinzig, Walter, & Doepfner, 2009; Yoshida & Uchiyama, 2004). Children and youth who experience comorbid autistic and ADHD symptoms experience greater impairment in psychopathology, interpersonal, school, family, and cognitive domains compared with typically developing controls (Gargaro et al., 2014; Kotte et al., 2013) increasing their risk of experiencing mental health difficulties over time.

Over the last decade, there has been an increasing focus on behavioral comorbidities in children diagnosed with neurodevelopmental disorders (Jeste, 2011). For example, estimates of parent-reported sleep disturbance in children diagnosed with ASD range from 73% to 86% (Liu, Hubbard, Fabes, & Adam, 2006; Polimeni, Richdale, & Francis, 2005) and up to 70% of children with ADHD have parent-reported sleep problems (Sung, Hiscock, Sciberras, & Efron, 2008). Children with ASD commonly have parent-reported difficulties with sleep onset and maintenance including long sleep onset latencies, short night sleep duration, and early morning waking (Krakowiak, Goodlin-Jones, Hertz-Picciotto, Croen, & Hansen, 2008; Richdale & Prior, 1995). Similar types of sleep problems have been reported in children with ADHD (Sung et al., 2008). The cause of sleep disturbance in neurodevelopmental disorders is unknown; however, neurobiological factors, such as abnormalities in melatonin secretion (Hollway & Aman, 2011), and behavioral factors, such as lack of family routines, have been associated with sleep problems (Richdale & Schreck, 2009). Internalizing and externalizing comorbidities have also been associated with sleep problems in children with ADHD (Lycett, Mensah, Hiscock, & Sciberras, 2014).

Sleep problems appear to be more persistent in children with neurodevelopmental disorders such as ASD and ADHD. In a recent parent report longitudinal study, 65% of the ASD group still experienced sleep disturbance 1 year later (May, Cornish, Conduit, Rajaratnam, & Rinehart, 2013), compared with 32% of controls. The picture appears very similar in children with ADHD. Seventy-two percent of sleep problems continued to be present 18 months later in a clinical sample of children with ADHD and/or anxiety (n = 76; Hansen, Skirbekk, Oerbeck, Wentzel-Larsen, & Kristensen, 2013). Furthermore, Lycett et al. (2014) recently demonstrated that comorbid internalizing and externalizing disorders were the strongest predictor of behavioral sleep problems in children with ADHD. In contrast, in a nationally representative longitudinal study, Quach, Hiscock, Canterford, and Wake (2009) found that only 2.9% of sleep problems persisted over a 2-year period.

Several studies have linked sleep problems with psychopathology in neurodevelopmental disorders. Sung et al. (2008; n = 239) found that children with ADHD and moderate or severe sleep problems were more likely to have poorer mental health than those without sleep problems. Goldman et al. (2009) demonstrated that children with ASD who were poor sleepers had more behavioral problems, for example, problems with attention span and social interactions compared with ASD children without parent-reported sleep difficulties. Using Diagnostic and Statistical Manual of Mental Disorders (4th ed.; DSM-IV; APA, 1994) criteria to establish a clinical diagnosis of insomnia in 32 children, Allik, Larsson, and Smedje (2006) reported that children with ASD and insomnia experienced more parent-reported autistic symptoms and emotional symptoms than ASD children who did not experience insomnia. Family stress has also been shown to be related to sleep problems in children with neurodevelopmental disorders (Doo & Wing, 2006); however, the only study to our knowledge that has investigated the impact of a behavioral sleep intervention on family stress in children with ASD, reported no significant improvements (Reed et al., 2009).

Despite the well-documented prevalence and impact of sleep problems in children with ASD and ADHD, little is known about their treatment. A recent systematic review of behavioral treatment of sleep problems in children with autism emphasized that existing studies consist of case reports or studies with small sample sizes (Vriend, Corkum, Moon, & Smith, 2011). In a non-randomized controlled study of children with ASD who are intellectually able (children who do not meet criteria for an intellectual disability), Reed et al. (2009) found that a parent sleep education workshop improved children’s daytime behavior (decreased hyperactivity, self-stimulatory behavior, and restricted behaviors), however, no significant differences in parental stress were observed. This study was limited by its small sample size (n = 20) and broad age range (3-10 years). A recent study conducted by Austin, Gordon, and O’Connell (2013) evaluated the effectiveness of a parent group program (sleepwise) for children with ASD and sleep disturbance, which included three workshop sessions as well as individualized treatment plans administered over a 15-week period. Although this program was effective in reducing parent-reported sleep disturbance and behavioral problems, this study was uncontrolled and had a small sample size (n = 7). Larger, more recent ASD sleep studies conducted by Gringras et al. (2011; n = 30) and Malow et al. (2014; n = 80) report that brief behavioral parent-based interventions are associated with improved sleep problems measured on the Children’s Sleep Habits Questionnaire. Despite this, Malow et al.’s (2014) study compared individual versus group treatment of sleep problems and did not have a treatment as usual control group and the large study conducted by Gringras et al. (2011) did not examine sleep disturbance in children with a specific diagnosis of ASD thus impacting the ability to generalize the findings from these studies.

To date, very few randomized controlled trials have been published evaluating the impact of behavioral sleep interventions in children with ADHD (Cortese et al., 2013). Keshavarzi et al. (2014) recently reported that a 12-week sleep-training program for 10-year old children with ADHD (n = 40) had beneficial effects for both sleep and psychosocial functioning compared with a control group of children with ADHD who did not receive the behavioral intervention (n = 20) and typically developing children (n = 20). We have also demonstrated that briefer behavioral sleep interventions are associated with considerable benefits for children with ADHD and sleep problems (Hiscock et al., 2015; Sciberras, Fulton, Efron, Oberklaid, & Hiscock, 2011). In comparison with children receiving usual clinical care, children receiving the two-session behavioral sleep intervention had improvements in sleep, ADHD symptom severity, quality of life, daily functioning, and home and classroom behavior (Hiscock et al., 2015). However, it is unclear whether this behavioral intervention also had positive benefits for those presenting with complex comorbidities, including ASD.

Until recently, the DSM precluded a comorbid diagnosis of ADHD and ASD; therefore, few studies have examined interventions in this group. There is clear evidence that children with a diagnosis of ASD or ADHD experience behavioral sleep problems (Liu et al., 2006; Polimeni et al., 2005; Sung et al., 2008), with a high proportion of children with ASD or ADHD meeting diagnostic criteria for a comorbid ASD–ADHD diagnosis (Holtmann et al., 2007; Keen & Ward, 2004; Lee & Ousley, 2006). Furthermore, research indicates that children with a comorbid ASD–ADHD diagnosis experience greater emotional and behavioral disturbance and poorer quality of life outcomes compared with children with a diagnosis of ASD or ADHD alone (Gargaro et al., 2014; Kotte et al., 2013). Given the high prevalence of sleep problems in ASD and ADHD and the relationship between behavioral sleep disturbance and mental health problems, there is a need to evaluate the efficacy of behavioral sleep interventions for children who present with a comorbid ASD–ADHD diagnosis. Despite this, it is unclear whether behavioral sleep interventions for children with ADHD are also effective for children who present with comorbid ASD.

The aim of this study is to evaluate the efficacy of a brief behavioral sleep intervention program previously demonstrated to be effective in treating sleep problems in children with ADHD, in children with comorbid ADHD and ASD. The “Sleeping Sound” study (Hiscock et al., 2015) did not exclude children with comorbid ASD, presenting a unique opportunity to examine whether the intervention was also effective in treating sleep problems in children whose parents reported that their child had been diagnosed by a health professional with ASD, in addition to ADHD. We aimed to determine whether the “Sleeping Sound” behavioral sleep intervention was effective in improving child sleep (primary outcome), behavior, quality of life, and parent mental health. We hypothesized that the intervention would improve sleep problems, child behavior, and quality of life. We explored whether the intervention would be associated with improved parent mental health.

Method

Study Design and Sample

We report on a subsample of participants from the Sleeping Sound With ADHD study who, in addition to ADHD, had a diagnosis of ASD. Diagnosis of ASD was ascertained by asking parents the following question: “In addition to ADHD, has your child been diagnosed with any of the following by a health professional?” The Sleeping Sound With ADHD study was a randomized controlled trial of a behavioral sleep intervention compared with usual clinical care for children with ADHD and sleep problems that has been described fully elsewhere (Sciberras et al., 2010). Briefly, pediatricians (n = 50) identified children aged 5 to 13 years whom they had diagnosed with ADHD and seen in the past 12 months. Families who did not opt out were contacted to assess eligibility and interest in participation. Inclusion criteria were (a) pediatrician diagnosis of ADHD, (b) parent-reported ADHD Rating Scale IV (rated off medication) symptom score of 6 or greater on either the inattention or the hyperactive/impulsive subscale, (c) age of onset prior to 7 years, (d) symptom duration of at least 6 months, and (e) cross-situational impairment (e.g., home and school). Inclusion criteria 3 to 5 were assessed using study-designed questions. In addition, children also needed to meet American Academy of Sleep Medicine (2001) criteria for at least one behavioral sleep disorder (i.e., sleep onset association disorder, limit setting disorder, delayed sleep phase, and/or idiopathic/psychophysiological insomnia) or be experiencing significant night time anxiety, assessed using a study-designed telephone interview. Any child receiving specialist sleep assistance, with a serious medical condition (e.g., cerebral palsy), an intellectual disability, or suspected obstructive sleep apnea was ineligible for the study.

Seventy-three percent (244 out of 336) of children identified as eligible consented to participate in the Sleeping Sound With ADHD study. There was no difference in child age, child gender, or neighborhood socio-economic disadvantage for participants versus non-participants. An independent statistician randomized consenting families using a computer-generated random number sequence to either the “intervention” group or the “usual care” group.

Sixty-one participants also had a diagnosis of ASD according to parent report and are reported in this article. The characteristics of this subsample are reported in Table 1. There were no significant differences between groups on any of the baseline sample characteristics (gender, age, socio-economic status, and comorbidities).

Table 1.

Baseline Sample Characteristics.

Characteristics	Intervention (n = 28)	Usual care (n = 33)
Child
Male, n (%)	24 (85.7)	30 (90.9)
Age in years, M (SD)	10.3 (1.7)	9.8 (2.0)
Medication use, n (%)	26 (92.9)	27 (81.8)
Ritalin/Ritalin LA (Long Acting)	11 (39.3)	13 (39.4)
Concerta	13 (46.4)	12 (36.4)
Atomoxetine	0 (0.0)	3 (9.1)
Clonidine	4 (14.3)	0 (0.0)
Parent-reported comorbidities
Learning difficulties, n (%)	3 (10.7)	9 (27.3)
Anxiety Disorders Interview Schedule for Children IV
Internalizing comorbidity, n (%)^a	18 (69.2)	26 (83.9)
Externalizing comorbidity, n (%)^b	17 (65.4)	22 (71.0)
Primary caregiver
Mother, n (%)	25 (92.6)	33 (100.0)
Age in years, M (SD)	40.1 (5.3)	38.9 (4.4)
Completed high school, n (%)	19 (67.9)	18 (54.6)
College/postgraduate degree, n (%)	7 (25.0)	6 (19.4)
Family
SEIFA, M (SD)	1,026.3 (58.9)	1,023.7 (39.5)

Note. SEIFA = socio-economic indexes for areas.

Children meeting criteria for two or more anxiety disorders (separation anxiety disorder, social phobia, specific phobia, generalized anxiety disorder, panic disorder, obsessive compulsive disorder, post-traumatic stress disorder) or one mood disorder (major depressive episode or dysthymia).

Children meeting criteria for oppositional defiant disorder or conduct disorder on the Anxiety Interview Schedule for Children IV.

Intervention

The intervention consisted of two face-to-face sleep consultations and a follow-up phone call with a trained clinician, each 2 weeks apart. Trained clinicians included five psychologists with 1 to 4 years of clinical experience or a trainee consultant pediatrician with 4 years of pediatric clinical experience. All clinicians received two 3-hr training sessions on pediatric sleep management, conducted by H.H. and E.S. Fidelity of clinicians was assessed using a standardized consultation form that required clinicians to record consultation duration and key program components (history, diagnoses, management). Clinicians also met fortnightly with H.H. and E.S. to address any clinical issues.

The first consultation involved assessment of the child’s sleep problem, discussion of parental goals, and psychoeducation about normal sleep, sleep cycles, and sleep hygiene strategies. During this consultation, a behavioral sleep management plan was formulated that was tailored to the child’s sleep problem. Examples of behavioral recommendations to families are included in Table 2. Parents were asked to maintain a sleep diary (e.g., time of sleep onset, time of awakening, night wakings, etc.) between the first and second session. The second consultation and follow-up phone call were used to review the sleep diary, reinforce strategies, and troubleshoot any problems encountered. Parents were also provided with information sheets addressing normal sleep, common sleep problems, and strategies for managing specific problems.

Table 2.

Examples of Behavioral Recommendations to Families for the Management of Sleep Disorders.

Sleep disorder	Definition	Examples of behavioral interventions
Sleep onset association disorder	Child associates falling asleep with a certain object (e.g., television) or person and is unable to fall asleep without it.	Parental presence at sleep time can be managed with adult fading (i.e., graduated extinction). This technique, called “camping out” requires gradual withdrawal of parental presence from the child’s bedroom over 7 to 10 days. Alternatively, parents can use a “checking method,” which involves going in to see the child at regular time intervals (2, 5, or 10 min depending on what the child can handle with intervals increasing over time) to check on them. This helps reassure children that parents are still there to assist them if need be.
Delayed sleep phase	Shift in the child’s sleep phase whereby the child falls asleep late and wakes late in the morning.	Bedtime fading is used whereby the child’s bedtime is temporarily set later and gradually brought forward. The child is then woken at a pre-set time in the morning. Early morning light exposure is encouraged.
Limit setting sleep disorder	Non-compliant behavior at bedtime where a parent fails to set appropriate bedtime limits.	Assisting parents to ignore child protests and reward compliance with bedtime routines. A “bedtime pass,” whereby the child can only leave the bedroom one time before sleep, can be used to promote compliant behavior. Bedtime fading can be used in conjunction with these strategies.
Primary insomnia	Child is unable to initiate or maintain sleep even if they go to bed at a later time.	Relaxation training, basic cognitive restructuring, and restricting time in bed (e.g., temporarily setting the bedtime later as per delayed sleep phase or getting out of bed and doing a relaxing activity if the child cannot fall asleep).
Night time anxiety	Child lies in bed worrying about things that have happened during the day or about specific night time fears (e.g., fear of the dark).	Discussing fears during the day rather than just before bedtime, encourage children to stay in bed, so that they can learn that their bedroom is a safe place, rewarding brave behavior, use of a security object, avoiding scary television shows, and relaxation training.

Usual Care

Participants randomized to usual care were able to continue assessing health care from their pediatrician, as they normally would, which does not routinely include addressing sleep issues (Sung et al., 2008). Pediatricians typically see children with ADHD every 6 months to check their height, weight, and blood pressure and to re-issue a prescription for medication and/or provide brief behavioral strategies, where necessary.

Measures

All measures were administered at baseline and then again at 3 and 6 months post-randomization. Participants in the larger study were administered a large battery of measures. To reduce the chance of Type 1 errors due to multiple comparisons given the smaller sample size reported in this article, we chose to limit the secondary outcome measures to those that we hypothesized would improve as a result of improved sleep in this population. A complete list of measures completed by families can be found in Sciberras et al. (2010).

Child Sleep Habits Questionnaire (CSHQ)

The primary outcome measure was parent-reported child sleep problems assessed using the CSHQ (Owens, Spirito, & McGuinn, 2000). The CSHQ is a validated parent-reported 33-item scale that measures child difficulties in initiating and maintaining sleep over the past week. The measure consists of a number of subscales measuring specific sleep problem domains including Bedtime Resistance, Sleep Onset Delay, Sleep Duration, Sleep Anxiety, Night Wakings, Parasomnias, and Daytime Sleepiness.

Secondary outcome measures

Behavior

The parent- and teacher-reported Strengths and Difficulties Questionnaire (SDQ; Goodman, 2001) was used to assess child behavior. The SDQ is a widely used and psychometrically robust 25-item scale that yields a total difficulties score and five subscale scores (Emotional Problems, Conduct Problems, Hyperactivity/Inattention, Peer Problems, and Prosocial Behavior). We report on the SDQ total score and the Emotional, Conduct, and Peer Problems scales in this study.

ADHD symptom severity

The parent- and teacher-reported ADHD Rating Scale IV (DuPaul, Power, Anastopoulos, & Reid, 1998) is a validated 18-item measure of inattention and hyperactivity/impulsivity experienced over the past month whereby higher scores reflect greater symptom severity.

Quality of life

Child psychosocial quality of life was assessed via the parent-reported Pediatric Quality of Life Inventory 4.0 (PedsQL 4.0; Varni, Limbers, & Burwinkle, 2007). The PedsQL 4.0 is a well-validated 23-item measure of child quality of life; 15 items sum to give a psychosocial health summary score, which can range from 0 to 100 with higher scores indicating a better quality of life.

Parent mental health

Parent mental health was assessed using the Depression Anxiety Stress Scale (DASS; Lovibond & Lovibond, 1995), a widely used, validated measure of mental health in adults. Parents rated their own symptoms over the past 4 weeks with higher scores indicating more mental health difficulties.

Statistical Analysis

For parent-reported outcomes, change scores denoting the difference in scores between 3 months and baseline, and 6 months and baseline were calculated for each outcome. Mean differences in change scores (baseline to 3 months; baseline to 6 months) between the intervention and usual care group and 95% confidence intervals (CIs) were estimated using linear regression. Given the pilot nature of this study, all presented analyses are unadjusted and do not account for potential confounding variables.

For teacher-reported outcomes, scores were compared between the intervention and control groups after adjusting for baseline scores. Baseline scores were adjusted given that most teachers who completed follow-up assessments differed from those who completed the baseline survey.

To calculate effect sizes (ESs), change scores were standardized to a mean of 0 and a SD of 1 and regression analyses were repeated. ESs are considered as small, ~0.20 SD; moderate, ~0.50 SD; and large, ~0.80 SD (Cohen, 1992). Analyses were completed using Stata 13.0.

Results

Primary Outcome: Child Sleep Problems

At 3 months post-randomization, data about sleep problems were collected from 21 families (75%) in the intervention group and 24 families (73%) in the usual care group. At 6 months post-randomization, 26 families (93%) in the intervention group and 26 families (79%) in the usual care group provided information about child sleep problems.

At 3 months post-randomization, compared with families receiving usual care, families in the intervention group reported a greater decrease in child sleep problems (ES = −0.7, p = .02), although this difference attenuated somewhat at 6 months post-randomization (ES = −0.5, p = .08, see Table 3). When examining the subscales of the CSHQ at 3 months, the improvement in total sleep problems appeared to be driven by intervention children having greater improvements in Sleep Onset Delay (ES = −0.9, p < .001) and Parasomnias (ES = −0.6, p = .04). There was some evidence that the intervention group may also have had improvements at 3 and 6 months in Sleep Duration (ES = −0.5, p = .1 and −0.8, p = .003, respectively), and Sleep Anxiety (ES = −0.5, p = .12) at 3 months post-randomization.

Table 3.

Mean Change in Sleep Problems at 3 and 6 Months Post-Randomization.

	Study group				Difference in change score
	Intervention		Control		(Intervention–control)
Outcome	n	M (SD)	n	M (SD)	M (95% CI)	Effect size	p
Child Sleep Habits Questionnaire
Total score
Baseline	28	56.7 (6.6)	33	57.7 (9.4)
3 months	21	49.8 (6.8)	24	54.2 (7.6)	−4.3 [−8.1, −0.6]	−0.7	.02
6 months	26	51.6 (7.0)	26	54.4 (8.4)	−2.8 [−5.9, 0.4]	−0.5	.08
Bedtime resistance
Baseline	28	8.6 (1.6)	33	10.3 (3.0)
3 months	21	7.4 (2.0)	24	9.2 (2.6)	−0.1 [−1.2, 1.0]	−0.07	.81
6 months	26	7.7 (1.6)	26	9.1 (2.8)	−0.05 [−1.0, 0.9]	−0.03	.92
Sleep onset delay
Baseline	28	2.8 (0.4)	33	2.5 (0.7)
3 months	21	1.8 (0.7)	24	2.3 (0.8)	−0.7 [−1.1, −0.3]	−0.9	<.001
6 months	27	2.2 (0.8)	26	2.3 (0.8)	−0.3 [−0.7, 0.1]	−0.4	.12
Sleep duration
Baseline	28	6.7 (1.5)	33	6.1 (1.5)
3 months	21	5.3 (1.6)	24	5.4 (1.5)	−0.7 [−1.6, 0.1]	−0.5	.10
6 months	27	5.3 (1.5)	26	5.6 (1.6)	−1.0 [−1.6, −0.3]	−0.8	.003
Sleep anxiety
Baseline	28	6.6 (1.4)	33	7.8 (2.1)
3 months	21	6.0 (1.8)	24	7.4 (2.0)	−0.6 [−1.3, 0.2]	−0.5	.12
6 months	26	5.7 (1.7)	26	7.2 (1.8)	−0.5 [−1.3, 0.3]	−0.3	.25
Night wakings
Baseline	28	4.8 (1.7)	33	4.9 (1.8)
3 months	21	4.7 (1.6)	24	5.0 (1.9)	−0.1 [−1.0, 0.8]	−0.08	.80
6 months	26	4.7 (1.6)	26	4.7 (1.8)	0.1 [−0.4, 0.6]	0.1	.70
Parasomnias
Baseline	28	10.7 (2.5)	33	10.7 (2.8)
3 months	21	9.7 (2.5)	24	10.6 (2.3)	−1.0 [−2.0, −0.03]	−0.6	.04
6 months	26	9.7 (2.6)	26	10.7 (2.8)	−1.3 [−2.2, −0.4]	−0.7	.006
Daytime sleepiness
Baseline	28	15.2 (4.0)	33	15.2 (4.6)
3 months	21	13.9 (3.6)	24	13.7 (3.5)	−1.0 [−3.0, 1.0]	−0.3	.33
6 months	26	15.0 (3.7)	26	14.2 (4.0)	0.4 [−1.4, 2.3]	0.1	.64

Note. CI = confidence interval.

Secondary Outcomes

Compared with children in the usual care group, children in the intervention group had greater improvement in psychosocial quality of life 6 months post-randomization (ES = 0.5, p = .05, Table 4). Due to the small sample size, we were unable to detect statistically significant differences in our other secondary outcomes. However, we interpret ESs of 0.3 and greater as indicating clinically significant differences in outcome. There was some evidence that the intervention group had greater improvements in teacher-reported child behavior at 6 months (SDQ total problems: ES = −0.4, p = .19) and parent-reported ADHD symptoms at 3 months (ES = −0.3, p = .25).

Table 4.

Mean Change in Secondary Outcomes at 3 and 6 Months Post-Randomization.

	Study group				Difference in change score
	Intervention		Control		(Intervention–control)^a
Outcome	n	M (SD)	n	M (SD)	M (95% CI)	Effect size	p
ADHD Rating Scale IV total score—Parent report
Baseline	28	36.3 (10.7)	33	37.2 (9.3)
3 months	23	32.4 (10.7)	25	33.6 (6.8)	−2.6 [−7.0, 1.9]	−0.3	.25
6 months	27	32.1 (11.5)	27	33.4 (8.2)	−1.6 [−6.0, 2.9]	−0.2	.48
ADHD Rating Scale IV total score—Teacher report
Baseline	23	28.2 (13.6)	27	29.2 (14.1)
3 months	21	24.5 (13.9)	24	28.8 (13.2)	−1.3 [−7.4, 4.7]	−0.1	.66
6 months	22	22.3 (13.1)	28	24.0 (13.1)	−1.5 [−8.1, 5.1]	−0.1	.64
PedsQL—Psychosocial quality of life score
Baseline	28	46.3 (13.4)	33	42.8 (11.0)
3 months	21	56.6 (16.8)	24	47.5 (14)	4.8 [−2.3, 11.9]	0.4	.18
6 months	27	55.4 (28.3)	25	46.4 (14.5)	6.5 [0.0, 12.9]	0.5	.05
DASS—Total score
Baseline	27	38.3 (24.4)	32	39.4 (29.9)
3 months	19	30.1 (21.7)	24	33.9 (23.6)	−3.8 [−17.9, 10.3]	−0.2	.59
6 months	26	34.7 (26.4)	24	41.9 (30.9)	−7.2 [−23.5, 9.1]	−0.3	.38
SDQ parent report—Total score
Baseline	28	22.5 (6.2)	33	24.0 (4.7)
3 months	21	20.9 (5.3)	24	23.0 (3.8)	−0.8 [−0.01, 1.5]	−0.2	.49
6 months	26	20.6 (7.2)	26	22.3 (5.3)	−0.3 [−3.1, 2.5]	−0.06	.83
SDQ teacher report—Total score
Baseline	23	16.6 (6.8)	27	19.2 (7.9)
3 months	21	14.3 (6.7)	23	19.5 (7.3)	−1.7 [−4.3, 0.9]	−0.2	.20
6 months	22	13.2 (6.6)	28	17.9 (7.5)	−2.7 [−6.9, 1.4]	−0.4	.19

Note. CI = confidence interval; DASS = Depression Anxiety Stress Scale; SDQ = Strengths and Difficulties Questionnaire.

For teacher-reported outcomes, mean score at 3 and 6 months adjusted for baseline scores.

We also examined outcomes for children on three SDQ subscales: Emotional, Conduct, and Peer Problems (not shown in tables). Intervention children had greater improvement in emotional functioning as measured by the SDQ by both parent (mean difference = −1.0, 95% CI = [−2.0, −0.1]; ES = −0.6, p = .04) and teacher report (mean difference = −1.2, 95% CI = [−2.3, −0.1]; ES = −0.5, p = .04) at 3 months; however, these differences dissipated at 6 months (ES = −0.03 and 0.01, respectively). Although not statistically significant, there was some evidence to suggest intervention children had greater improvement on the SDQ conduct problems subscale by teacher report (mean difference = −0.8, 95% CI = [−2.2, 0.5]; ES = −0.3, p = .23) at 6 months. Although according to parent report intervention children had less improvement in conduct problems at 3 (mean difference = 0.6, 95% CI = [−0.3, 1.5]; ES = 0.4, p = .19) and 6 months (mean difference = 0.7, 95% CI = [−0.2, 1.7]; ES = 0.4, p = .12) compared with the usual care group. Last, there was some evidence that intervention children had small improvements in peer relationships at 6 months (SDQ Peer Problems scale) by both parent (mean difference = −0.6, 95% CI = [−1.6, 0.4]; ES = −0.3, p = .22) and teacher report (mean difference = −1.0, 95% CI = [−2.3, 0.3]; ES = −0.4, p = .11).

Parent Mental Health

There was no difference between groups on parent mental health after 3 months, although there was a small non-significant effect at 6 months post-randomization, whereby parents of intervention children appeared to have greater improvement in mental health (ES = −0.3, p = .38) than parents of children in usual care.

Discussion

This study aimed to investigate the efficacy of a behavioral sleep intervention in children with comorbid ADHD and ASD. Similar to the larger Sleeping Sound With ADHD trial, we found that children with ADHD–ASD who received the intervention had moderate to large improvements in sleep problems at 3 and 6 months post-randomization, compared with the usual care group. Children in the intervention group also had small to moderate improvements in psychosocial quality of life, ADHD symptom severity and child behavior. The ESs obtained in this substudy were comparable with the larger Sleeping Sound trial, suggesting that this intervention is similarly efficacious for children presenting with both ADHD and ASD. Previous research has demonstrated that behavioral sleep interventions can be effective in improving sleep in typically developing children (Mindell, Kuhn, Lewin, Meltzer, & Sadeh, 2006). There is preliminary evidence that behavioral sleep interventions may also be effective in improving sleep in children with ADHD (Sciberras et al., 2010; Sciberras et al., 2011) and children with ASD (Gringras et al., 2011; Malow et al., 2014). Despite this, prior studies have been conducted with samples of children with ASD who also have an intellectual disability or are of preschool age (Vriend et al., 2011) and have not taken into account comorbid ADHD–ASD. This is the first randomized controlled trial, to our knowledge, to demonstrate, that a brief behavioral sleep intervention is effective in improving sleep problems in children with comorbid ADHD–ASD who are intellectually able.

As well as improving sleep problems, this intervention was associated with small to moderate improvements in a broad range of domains including child psychosocial quality of life, ADHD symptom severity, and child behavior. These findings are consistent with a randomized controlled trial (RCT) recently conducted by Malow et al. (2014), which found that a parent sleep education program improved child’s quality of life. Our study also compliments previous studies that report the effectiveness of behavioral sleep interventions for child and family health in ASD (Reed et al., 2009). Given the association between sleep disturbance and emotional problems (Malow et al., 2006), it is not surprising that we found that the Sleeping Sound intervention was associated with improved emotional symptoms. It has been demonstrated that sleep problems during childhood are predictive of behavioral and emotional problems in mid-adolescence (Gregory & O’Conner, 2002); thus, targeting sleep disturbances early may aid in preventing poorer mental health outcomes over time.

Prior research has demonstrated an association between parent mental health and child sleep problems in children with ASD (Doo & Wing, 2006) and ADHD (Sung et al., 2008); however, the direction of the relationship is unknown. Reed et al. (2009) did not find any improvement in parenting stress levels after the administration of their sleep workshop, which may suggest that family stress may contribute to a child’s sleep problem. Our current findings suggest there may be small improvements in parent mental health at 6 months post-randomization; however, larger powered studies are necessary to explore this further.

This study has a number of strengths. A rigorous methodology was employed, and this is the only sample to our knowledge that has considered comorbid ADHD–ASD in a randomized controlled behavioral sleep intervention trial. The sleep intervention administered is one that can be employed in everyday practice by both psychologists and pediatricians and was designed to be embedded into current treatment plans, rather than added onto the several intervention hours that children with ASD might access.

The study does, however, have some limitations. This study relied upon parent report of ASD diagnosis, and independent assessment for ASD was not possible. Although a semi-structured diagnostic interview was not used to confirm ADHD diagnosis, our approach was rigorous. All children needed to have a previous diagnosis of ADHD made by a pediatrician and, in addition, needed to meet current symptom criteria using the ADHD Rating Scale IV. All the participants in this study were intellectually able, so it is not known whether this intervention would be effective for children with ADHD–ASD and an intellectual disability. Parents were not blinded to the intervention, which may have led to response bias; however, blinded teacher-reports were included to overcome this limitation. It is also important to note that there was some missing data at the 3- and 6-month follow-up. Given the small sample size and pilot nature of this study, approaches such as multiple imputation were not used to replace missing values. Thus, the present results should be viewed as preliminary and need to be replicated using a well-powered RCT design. Last, the current sample comprised of children with a comorbid diagnosis of ADHD–ASD; therefore, our results may not directly generalize to children with ASD alone. However, the presence of comorbid ADHD in the current sample may have made the successful management of sleep problems more difficult to achieve given that many children with ADHD display oppositional behaviors and are also medicated. Thus, the effects of the current sleep intervention may be an underestimate of what could be achieved in a group of children with ASD alone.

In conclusion, sleep problems are common in children with ADHD and ASD. A brief behavioral sleep intervention can improve sleep in children with ADHD–ASD and has positive effects on children’s social, emotional, and behavioral functioning and parental mental health. Rigorous, controlled trials are now needed with a sample size that is adequately powered to detect significant changes in improvement across all outcomes. Such research would also independently ascertain ASD diagnostic status and perhaps include a subset of participants with intellectual disabilities to determine whether the intervention would also be effective in this population.

Footnotes

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This project is funded by a Project Grant from the Australian National Health and Medical Research Council (NHMRC, 607362). Dr. Sciberras is funded by an NHMRC Early Career Fellowship in Population Health (1037159). Associate Professor Hiscock is funded by an NHMRC Career Development Award (607351).

Author Biographies

Nicole Papadopoulos is a psychologist and research fellow at Deakin University, Australia.

Emma Sciberras is a Post-Doctoral Research Fellow and Clinical Psychologist at the Murdoch Children’s Research Institute and The Royal Children’s Hospital, Melbourne, Australia. She is also an Honorary Research Fellow at the Department of Paediatrics, the University of Melbourne.

Harriet Hiscock is a consultant paediatrician and postdoctoral research fellow. She is co-director of the Unsettled Babies Clinic and Co-Group Leader of the Community Health Services Research group at the Murdoch Childrens Research Institute.

Melissa Mulraney completed her PhD in psychology at Monash University in Australia. She is involved in research at the Murdoch Childrens Research Institute investigating ways improve outcomes for children with ADHD.

Jane McGillivray is the Director of Professional Programs and Chair of the Doctor of Clinical Psychology in the School of Psychology at Deakin University, Australia.

Nicole Rinehart is a professor of clinical psychology at Deakin University, Australia and an honorary fellow at the Murdoch Children’s Research Institute, Melbourne, Australia.

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