Children Insomnia Neurodevelopmental disorders Treatment accessibility
A B S T R A C T
Background/aims: Insomnia is highly prevalent in children with neurodevelopmental disorders (NDDs), yet little research exists on sleep treatment access, utilization, and provision in this population. This study explores barriers and facilitators to access, use, and provision of treatment for sleep problems as experienced by parents of children with NDDs, including Autism Spectrum Disorder (ASD), Attention-Deficit/Hyperactivity Disorder (ADHD), Cerebral Palsy (CP) and Fetal Alcohol Spectrum Disorder (FASD), and health care professionals who work with children with these conditions.
Abbreviations: BNBD, Better Nights, Better Days (name of intervention); BNBD-NDD, Better Nights, Better Days for Children with Neuro- developmental Disorders (name of intervention); TD, typically developing; NDD, neurodevelopmental disorder; ADHD, attention-deficit/hyperac- tivity disorder; ASD, autism spectrum disorder; CP, cerebral palsy; FASD, fetal alcohol spectrum disorder; HCP, health care professional; BCBA, Board-Certified Behaviour Analyst; BIQ, Behavioural Insomnia Questionnaire; SILS, Single Item Literacy Scale; PSQ, Pediatric Sleep Questionnaire; RCT, randomized controlled trial.
* Corresponding author at: Department of Psychology & Neuroscience, Dalhousie University, Life Sciences Centre, Rm 2521, 1355 Oxford Street, Halifax, NS, B3H 4R2, Canada.
E-mail address: [email protected] (P.V. Corkum).
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Research in Developmental Disabilities
journal homepage: www.elsevier.com/locate/redevdis
https://doi.org/10.1016/j.ridd.2020.103792 Received 15 May 2020; Received in revised form 22 September 2020; Accepted 5 October 2020
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Barriers Parents Method: Transcripts from online focus groups and interviews, conducted separately with parents
of children with NDDs (n = 43) and health care professionals (n = 44), were qualitatively analyzed using content analysis for key themes. Results: Barriers included limited access to/availability of treatment, lack of knowledge/training, NDD-specific factors (e.g., symptoms, medications, and comorbidities), parent factors (e.g., ca- pacity to implement treatment, exhaustion), and the challenging, intensive nature of sleep treatment. Facilitators included positive beliefs and attitudes, education, support, and ability to modify treatments for NDD symptoms. Barriers and facilitators were similar across all four NDDs. Conclusions: Results highlight a need for more education about sleep in NDDs and to develop accessible interventions, as well as the potential of a transdiagnostic approach to sleep treatment in this population.
What this paper adds
This paper contributes to our understanding of parents’ experiences of seeking, accessing, and using treatments for behavioural sleep problems, such as insomnia, for their children with neurodevelopmental disorders (NDDs). At the same time, this paper provides information on front-line health care professionals’ experiences with providing treatments for insomnia for children with NDDs. By engaging these key stakeholders, this study informs our understanding of unmet needs in the areas of sleep treatment accessibility, delivery, and use, as well as professional development and training needs related to sleep treatment. Findings from this study also add to a growing body of evidence that supports a transdiagnostic approach to treating sleep problem in children with NDDs.
1. Introduction
1.1. Background
Neurodevelopmental disorders (NDDs) emerge in early childhood and are linked to disturbances in central nervous system func- tioning, which can cause impaired cognition, communication, motor skills, and/or behaviour, and functional impairment in a variety of daily life domains (American Psychiatric Association, 2013). Sleep problems are highly prevalent in children with NDD, with rates ranging from 40 to 86% (Robinson-Shelton & Malow, 2016; Romeo et al., 2014). Insomnia, the most common sleep problem expe- rienced by children with NDD, includes difficulty falling and staying asleep (American Academy of Sleep Medicine, 2014). Throughout this paper, we will use the terms insomnia and sleep problems interchangeably.
Sleep problems have been shown to increase the severity of NDD symptoms as well as behavioural and emotional problems, and to have negative effects on children’s daytime functioning (Tudor, Hoffman, & Sweeney, 2012; Goldman et al., 2011; Newman, O’Regan, & Hensey, 2006). Children’s sleep problems occur within a broad psychosocial context and may affect the whole family; for example, parents of children with NDDs and sleep problems experience high levels of stress (Doo & Wing, 2006).
Development of effective treatments for insomnia in children with NDDs is important, given the high prevalence and negative effects of sleep problems. Behavioural interventions are the first-line recommendation for pediatric insomnia in both NDD and typi- cally developing (TD) populations (Malow et al., 2012). Research on effective sleep interventions for children with NDDs is expanding, with several recent randomized controlled trials (RCTs) (e.g., Hiscock, Sciberras, & Mensah, 2015). A recent systematic review found support for a transdiagnostic behavioural approach to treating sleep problems in children with NDDs (Rigney et al., 2018), wherein the same behavioural treatment principles are applied across multiple diagnoses, with minor modification of strategies originally developed for TD children (e.g., psychoeducation, healthy sleep practices, extinction).
Emerging research suggests that access to and uptake of behavioural sleep interventions by families of children with NDDs is limited (e.g., Bessey, Coulombe, Smith, & Corkum, 2013; Boerner, Coulombe, & Corkum, 2014). Additionally, front-line health care professionals (HCPs) are generally not well trained to provide sleep interventions (e.g., Boerner et al., 2014), much less for special populations such as children with NDDs. There is a great need to explore factors influencing families’ seeking of, access to, and uptake of treatment for sleep problems in their children with NDDs, as well as the factors influencing HCPs’ ability to provide such treatment. This information will provide a foundation for the development of effective sleep interventions for this population.
Focus groups were conducted (or interviews when participants were not able to attend focus groups) to gather the perspectives of parents of children with NDDs and HCPs on barriers and facilitators to access, uptake, and provision of sleep treatments for children. Four prevalent NDDs that encompass a range of symptoms and functional impairments were included: Attention-Deficit/Hyperactivity Disorder (ADHD), Autism Spectrum Disorder (ASD), Cerebral Palsy (CP), and Fetal Alcohol Spectrum Disorder (FASD). The results of the study will identify unmet needs in the areas of treatment delivery and use, accessibility, and professional development and training in order to inform the development of a sleep intervention for children with these four NDDs.
1.2. Research objectives
The research objectives were to explore the barriers and facilitators experienced by 1) parents, in seeking, accessing, utilizing, and implementing treatments for sleep problems in children with NDDs; and 2) HCPs, in their access to information about and provision of
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sleep treatments for children with NDDs. We predicted that lack of knowledge, training, and time may be barriers reported by HCPs. We expected that both parents’ and HCPs’ beliefs and attitudes about the nature of sleep problems in NDDs and their treatability would influence responses regarding treatment seeking, access, uptake, and provision.
2. Method
2.1. Participants
This study was approved by the Research Ethics Board of the IWK Health Centre in Halifax, Nova Scotia, Canada. Informed consent was obtained from all participants, who were recruited online via social media, through sharing of recruitment advertisements by NDD-related parent and health organizations, and through the authors’ professional networks. Conducting individual interviews became necessary for some participants, due to difficulty accommodating time zones and schedules.
Fig. 1. Parent study flow diagram.
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2.1.1. Parent participants The final sample included 43 parents or caregivers (hereafter, parents) of children aged 4–12 years with parent-reported diagnoses
of ADHD (n = 9), ASD (n = 20), CP (n = 6), and/or FASD (n = 8), as well as behavioural sleep problems confirmed by a screening
Table 1 Demographic Information for Parent Participants and their Children.
Total Primary NDD Group
Parent Participant Demographics N = 43 ASD (n = 20)
ADHD (n = 9)
CP (n = 6)
FASD (n = 8)
Participants’ relationship to child Biological Mother 32 (74.4 %) 18 (90 %) 9 (100 %) 5 (83.3 %) Biological Father 3 (7%) 2 (10 %) 1 (16.7 %) Adoptive Mother 8 (18.6 %) 8 (100 %)
Participant (Parent/caregiver) Mean Age in Years (SD, range)
38.5 (SD = 7.1, 25− 65)
36.5 (SD = 5.2, 25− 47)
38.2 (SD = 4.7, 32− 45)
38.7 (SD = 8.5, 28− 52)
43.6 (SD = 10.5, 32− 65)
Participants’ relationship status Married/Common-law 33 (75.7 %) 15 (75 %) 6 (66.7 %) 5 (83.3 %) 7 (87.5 %) Single/Never legally married 3 (7%) 1 (5%) 1 (16.7 %) 1 (12.5 %) Separated/Divorced 6 (14.0 %) 4 (20 %) 2 (22.2 %) 1 (16.7 %) 1 (12.5 %)
Community of residence Rural 12 (27.9 %) 5 (25 %) 2 (22.2 %) 1 (16.7 %) 4 (50 %) Town 5 (11.6 %) 3 (15 %) 1 (11.1 %) 1 (16.7 %) 1 (12.5 %) City 26 (60.4 %) 12 (60 %) 6 (66.7 %) 4 (66.7 %) 3 (37.5 %)
Ethnic or Cultural Heritage White/Caucasian 39 (90.7 %) 19 (95 %) 8 (88.9 %) 6 (100 %) 6 (75 %) Aboriginal – Metis 1 (2.3 %) 1 (12.5 %) Other 2 (4.7 %) 1 (5%) 1 (12.5 %)
Highest Level of Education High school equivalent or less 3 (7%) 2 (10 %) 1 (16.7 %) Diploma or certificate from college, university, trade/technical/vocational school, or less
18 (41.2 %) 7 (35 %) 5 (55.5 %) 2 (33.3 %) 4 (50 %)
Bachelor’s/Undergraduate Degree (e.g., BA, BSc, BEd)
13 (30.2 %) 7 (35 %) 2 (22.2 %) 2 (33.3 %) 2 (25 %)
Graduate degree (e.g., MA, MSc, MEd, PhD, DSc, EdD)
8 (18.6 %) 4 (20 %) 1 (11.1 %) 1 (16.7 %) 2 (25 %)
Participant’s Current Employment Status Full Time 21 (48.8 %) 7 (35 %) 6 (66.7 %) 4 (66.7 %) 4 (50 %) Part Time 7 (16.3 %) 6 (30 %) 1 (12.5 %) Unemployed 1 (2.3 %) 1 (12.5 %) Student 1 (2.3 %) 1 (5%) Homemaker 8 (18.6 %) 6 (30 %) 2 (33.3 %) Other 4 (9.3 %) 2 (22.2 %) 2 (25 %)
Estimated Household Income Less than $30,000 3 (7%) 2 (10 %) 1 (16.7 %) $30,000 – $59,999 7 (16.3 %) 3 (15 %) 1 (11.1 %) 1 (16.7 %) 2 (25 %) $60,000 – $99,999 16 (37.2 %) 6 (30 %) 4 (44.4 %) 1 (16.7 %) 3 (37.5 %) $100,000 + 16 (37.2 %) 9 (45 %) 2 (22.2 %) 2 (33.3 %) 3 (37.5 %) $100,000 – $149,999 9 (20.9 %) 6 (30 %) 1 (11.1 %) 2 (33.3 %) $150,000 – $199,999 5 (11.6 %) 1 (5%) 1 (11.1 %) 3 (37.5 %) $200,000 and over 2 (4.7 %) 2 (10 %)
Average Number of Other Children in Home (mean; SD; range)
1.95 (.90, 1− 5) 1.85 (.49; 1− 3) 2.44 (1.13, 1− 5) 1.50 (.55, 1− 2) 2.00 (1.41, 1− 5)
Child Demographics N = 43 ASD (n = 20) ADHD (n = 9) CP (n = 6) FASD (n = 8) Child Sex
Male 29 (67.4 %) 15 (75 %) 6 (66.7 %) 3 (50 %) 5 (62.5 %) Female 14 (32.6 %) 5 (25 %) 3 (33.3 %) 3 (50 %) 3 (37.5 %)
Child Mean Age in years (SD, range) 8.5 years (SD = 2.5, 4.3–12.6)
9 years (SD = 2.5, 4.8–12.6)
6.9 years (SD = 2.3, 4.3–11.4)
8.3 years (SD = 2.3, 4.9–11.7)
9 years (SD = 2.3, 4.8–11.5)
Years Since NDD Diagnosis (SD, range) 4.2 (2.5, 1− 10) 4.1 (SD = 1.8, 2− 8)
2.6 (SD = 2.3, 1− 7)
5.8 (SD = 1.9, 4− 9)
5 (SD = 3.6, 1− 10)
Comorbid Diagnoses: Additional NDD, Mental Health, and Physical Disorders (may have multiple diagnoses) Presence of Parent-reported Diagnosis 30 (69.8 %) 12 (60 %) 5 (55.6 %) 5 (83.3 %) 8 (100 %)
Another NDD (ADHD, ASD, CP, or FASD) 9 (20.1 %) 2 (10 %) 1 (16.7 %) 6 (75 %) Learning Disability 7 (16.3 %) 2 (10 %) 1 (11.1 %) 2 (33.3 %) 2 (25 %) Intellectual Disability / Developmental Delay 9 (20.9 %) 3 (15 %) 2 (33.3 %) 4 (50 %) Mental Health disorder (e.g., at least one of: anxiety, mood, obsessive compulsive disorder, oppositional defiant disorder)
26 (60.5 %) 10 (50 %) 5 (55.6 %) 4 (66.7 %) 7 (87.5 %)
Note: One participant did not complete; multiple participants missed or skipped questions.
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questionnaire; only participants whose children’s diagnoses were made by a physician or psychologist (self-reported) were eligible to participate. In cases where children had comorbid ADHD with ASD, CP, or FASD, the ASD/CP/FASD diagnoses were considered primary for assigning them to a disorder group (e.g., comorbid FASD and ADHD = FASD group). As such, children of parents in the ADHD group could not have comorbid ASD, CP, or FASD. Parents were required to live in Canada, have access to a computer, internet, web-camera and microphone (or telephone), and be comfortable speaking/reading English. Parent-reported formal diagnoses of sleep disorders other than insomnia (e.g., sleep apnea or sleep-disordered breathing) were an exclusion criterion due to the potential confound with behavioural insomnia. Information about children’s comorbid diagnoses (e.g., NDD, neurological, physiological, mental health) and medication use was recorded but not used as exclusionary criteria.
Fig. 1 depicts parent participation, and Table 1 contains demographic information. Twenty-seven parents participated in focus groups (which ranged from 2 to 5 participants each) and 16 parents completed individual interviews. Most parents were biological mothers (74.4 %). The mean age of parents was 38.5 years (SD = 7.1, range = 25− 65), and most parents were married/common-law (n = 33, 75.7 %). Most lived in cities (n = 26, 60.4 %), were of Caucasian heritage (n = 39, 90.7 %), and had completed high school and some post-secondary education (n = 39, 90.7 %). The average reported number of other children in the home was 1.95 (SD = .9, range 1–5). Most parents were from Ontario (n = 17, 39.5 %), British Columbia (n = 8, 18.6 %), and Alberta (n = 7, 16.3 %), with the remainder from Nova Scotia (n = 4), New Brunswick (n = 2), Newfoundland and Labrador (n = 2), and Prince Edward Island, Manitoba, and Quebec (each n = 1).
Fig. 2. HCP study flow diagram.
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Most children were male (n = 29, 67.4 %) and mean age was 8.5 years (SD = 2.5, range 4.3–12.6). Most children had at least one other parent-reported diagnosis (n = 30, 69.8 %), including other comorbid NDDs (ASD, ADHD, CP, or FASD; n = 9, 20.1 %) or mental health diagnoses (n = 26, 60.5 %); anxiety was common (n = 16, 37.2 %). Children also had a range of parent-reported physical health conditions (n = 19, 44.2 %), most frequently epilepsy/seizure disorders (n = 5, 11.6 %), other neurological disorders (n = 7, 16.3 %), gastrointestinal disorders (n = 6, 14 %), and respiratory disorders (n = 4, 9.3 %).
In terms of behavioural insomnia (Anders & Dahl, 2007), fifteen (34.9 %) children met criteria for bedtime resistance/sleep onset problems, six (14 %) met criteria for night waking problems, and 18 (41.9 %) met criteria for both. Four children (9.3 %) were below threshold for behavioural insomnia, but were included as their parents reported high severity/impact of sleep problems. Twenty parents (46.5 %) reported that their children woke too early in the morning. Frequently reported problems were: problems falling asleep (n = 38, 88.4 %), lying awake in bed after lights out for more than 20 min (n = 38, 88.4 %), problems staying asleep (n = 34, 79.1 %), getting out of bed once expected to stay in bed for the night (n = 32, 74.4 %), and waking during the night with difficulty falling back asleep (n = 32, 74.4 %).