Behavioral Sleep Interventions For Children 0-6 Age Group: A Systematic Review
Izel Guvensoy
12251437 Word count: 6067
Abstract
Sleep problems affect nearly 20-30 % of children, resulting in undesirable outcomes such as inattention or negative cognitive development. This aim of this review was to determine whether behavioral sleep interventions reduce children’s sleep problems. Secondly, this review aimed to identify the most effective strategies of these behavioral sleep interventions. We viewed 1516 articles and selected seventeen papers that fit the eligibility criteria for inclusion in the analyses. A structured data extraction sheet was used in summarizing the eligible studies. The findings indicate that behavioral sleep interventions are effective in reducing sleep problems. Across all studies, 76% reported that behavioral sleep interventions are efficacious. In particular, strong support was found for graduated extinction and bedtime routine strategies. Majority of children respond positively to these behavioral techniques and showed fewer sleep problems. Intervention developers should pay particular attention to the results of this study and tailor interventions that have large effects. It is important that a meta-analysis will be done to replicate these results and empirical studies on behavioral sleep interventions need to include methods beyond subjective measurements for measuring sleep outcomes.
Behavioral Sleep Interventions For Children 0-6 Age Group: A Systematic Review Sleep problems are highly prevalent among children. Nearly 20-30% of children experience sleep difficulties such as frequent waking, sleep onset latency, etc. (Mindell & Durand, 1993; Burnham, Goodlin-Jones, Gaylor, & Anders, 2002). The definition of sleep problems varies in terms of age, frequency, severity, and duration of symptoms. The International Classification of Sleep Disorder uses “Behavioral Insomnia of Childhood” terminology to refer to sleep problems which has two categories: limit-setting and sleep-onset association (Mindell, Kuhn, Lewin, Meltzer, & Sadeh, 2006). Limit-setting category includes bedtime problems which are mainly seen in two years old children and older (Mindell et al., 2006). Bedtime refusal behaviors can be described as stalling, verbal protests, crying, clinging, refusing to go to bed, getting out of bed, attention-seeking behaviors, and multiple requests for food, drinks, and stories (Mindell et al., 2006). Regarding the sleep-onset associations, night awakenings, need for rocking, cuddling, and parental presence to fall asleep are included in this category (Mindell et al., 2006). In this study, I will use "sleep problems" to refer to the sleep-onset association category of Behavioral Insomnia of Childhood terminology.
Sleeping is a child’s primary activity and undoubtedly important for the health and well-being of children (Matricciani, Blunden, Rigney, Williams, & Olds, 2013). It allows the child’s mind and body to rest and recover. Moreover, sleeping plays a role in the growth, learning, processing of memory, and central nervous system repair (Zee & Turek, 1999). The American Academy of Pediatrics recommends 12-16 hours of sleep for infants under 1 year old, 11-14 hours for children 1-2 years old and 10-13 hours for the children 3-5 age group (Dawkins, 2018). Infants’ sleep-wake cycle begins to develop at about six weeks of age, and by three to six months infants should have a regular sleep cycle. At the age of two, most children spend more time asleep than
awake. Overall, a child spends 40 % of their childhood asleep, which is vital for their mental and physical development (Sickkids, 2019).
Regarding the effects of sleep the scientific literature shows that good sleep during infancy and childhood is preventive for health problems (e.g., hypertension, diabetes, obesity) and ensures a healthy life into adulthood (Bruni, 2010). It is also known that sleeping is essential for children's optimal functioning and healthy development (Bruni, 2010). Child's sleep quality may be affected by a range of factors such as socio-demographic variables, parenting style and emotional/ behavioral factors including the child's age, gender, and socioeconomic status, parent education level, family income and sleep environment (e.g. sleeping arrangements) (Liu et al., 2013). Moreover, caregivers' sleep-related knowledge, behavior and attitudes, interparental inconsistency, and cognitions are other factors that are associated with sleep quality of children (Johnson & McMahon, 2008; Mindell, Sadeh, & Kohyama, 2010; Owens et al., 2011; Liu et al., 2014).
Insufficient quality of sleep during childhood causes behavioral (e.g., attention deficit hyperactivity disorder) and health problems (e.g., heart disease) and reduces the quality of life (Bruni, 2010). The inadequate sleep time in children is related to several undesirable functional outcomes such as sleepiness, inattention, negative cognitive development (e.g., learning, memory consolidation, executive function), negative mood regulation (e.g., chronic irritability, poor modulation of effect) as well as behavior problems (e.g., aggressiveness, hyperactivity, poor impulse control) (Beebe, 2011; Keren, Feldman, & Tyano, 2001; Gais, Plihal,Wagner, & Born, 2000; Lavigne et al., 2000; Sadeh, Gruber, & Raviv, 2002). Moreover, negative psychiatric and health outcomes including obesity and metabolic disorders, risk of depression, suicide attempts and self-harm behaviors are the other consequences of inadequate sleep time (Roberts, Roberts, &
Chen, 2002; Singareddy et al., 2013; Wong, Brower, & Zucker, 2011; Bell & Zimmerman, 2010; Magee & Hale, 2012). In addition, there are secondary effects on parents such as maternal depression, as well as on family functioning such as family stress (Hiscock & Wake, 2002). Unlikely to popular belief, child sleep problems do not disappear as the child grows (Wiggs, 2007). According to longitudinal studies, sleep problems often continue to occur after childhood and adolescence (Byars, Yolton, Rausch, Lanphear, & Beebe, 2012; Jenni, Fuhrer, Iglowstein, Molinari, & Largo, 2005; Meltzer, Plaufcan, Thomas, & Mindell, 2014; Roberts, Roberts, & Duong, 2008). Regarding the prevalence rates of persistent sleep problems, 21% to 35% of children shows persistent sleep problems (Byars et al., 2012). Similarly, another study results indicated that between 17% to 26% of children showed persistent sleep problems (Meltzer et al., 2014). Given the high prevalence rates and negative consequences of children’s sleep problems, effective treatment is essential.
According to the empirical literature, there are pharmacological and behavioral treatments that can help to reduce the sleep problems among children (Kuhn & Weidinger, 2000). Pharmacological treatments include antihistamines, chloral hydrate, benzodiazepines, and melatonin (Kuhn & Weidinger, 2000), however, these treatments are found to be temporarily effective in reducing the sleep problems of children (Ortiz & McCormick, 2007). Furthermore, there are adverse effects that may occur such as unwanted side effects when the medication is discontinued (Ortiz & McCormick, 2007). Even though there are some cases that the medication may be helpful for childhood sleep problems, considering the etiology of sleep problems, there is no solid basis for the use of pharmacological treatment for young children (Ortiz & McCormick, 2007). Approximately 82% of pediatricians reported using behavioral treatment instead of pharmacological treatment (Heussler et al., 2013). Regarding the behavioral approach, several
treatment strategies exist such as graduated extinction, positive bedtime routines, scheduled awakenings (Kuhn & Weidinger, 2000). The theoretical basis of this behavioral approach is that children who have sleep problems will be trained by the behavior of their parents or caregivers (Ortiz & McCormick, 2007). These efforts to create change in infants' behaviors can be viewed in the context of health behavior change models and theories (Moon, Hauck, & Colson, 2016). The behavioral interventions expect to end the undesired behaviors of the children, or at least significantly decrease the intensity and frequency of these behaviors (Ortiz & McCormick, 2007). Behavioral approaches attempt to create a sleep environment that a child would want to go to sleep willingly (Ortiz & McCormick, 2007). Moreover, these approaches teach parents specific skills such as the ways of settling baby at sleep time, how to respond crying and other strategies to prevent undisturbed sleep (Crichton & Symon, 2015) in order to create a smooth and peaceful sleep environment for the children (Ortiz & McCormick, 2007). Since the behavioral approach is mostly preferable by the pediatricians due to the temporary treatment of medicine and the possibility of treatment without medicine, this study will focus on behavioral treatment of sleep problems.
The number of studies have examined whether these behavioral sleep interventions were effective in reducing sleep problems among children. Many studies found positive outcomes of behavioral sleep interventions (e.g., Honaker, Schwichtenberg, Kreps, & Mindell, 2018; Salm Ward, & Balfour, 2015) whereas some studies found a short term or negligible effects (e.g., Ahlers-Schmidt et al., 2015; Stremler et al., 2013). A review study including 52 studies examined the effectiveness of behavioral interventions (Mindell et al., 2006). The results showed that 94% (49 of 52) of the studies found significant improvements in bedtime problems and night awakenings while the other three studies showed no effects (Mindell et al., 2006). A meta-analysis found
significant improvements in infants nocturnal total sleep time. However, no evidence was found for reducing night awakenings (Kempler, Sharpe, Miller, & Bartlett, 2016). Overall, it seems like behavioral sleep interventions are effective. This study aims to find evidence for this statement and provide an up-to-date review. Moreover, it is not clear in which domains these interventions are effective. This study will explore the effectiveness of behavioral interventions in three domains which are sleep duration, night awakenings and sleep onset latency. As indicated above contrasting results are present. There are several factors that could have caused contrasting results. Firstly, subjective measurements are mostly preferred to measure sleep outcomes. This could have affected the results. As the majority of RCT studies included self-reports to measure sleep-related outcomes, this study will also include subjective measurements. Secondly, the contextual factors such as delivery setting and structural factors such as the duration of the intervention might influence the results. Since there are contrasting findings, it is hard to conclude whether behavioral interventions are effective or not.
Regarding the components of behavioral interventions, unmodified extinction (parents ignore all negative behaviors until a set time in the morning), graduated extinction (includes brief parental checks after lights out and ignores all negative behavior) and parental education were identified as the most effective strategies in two comprehensive reviews (Mindell, 1999; Kuhn & Elliot, 2003). Other intervention strategies including bedtime routines (moving the child’s bedtime later to match when the child is currently falling asleep, and stimulus control techniques) and scheduled awakenings (waking and then consoling a child 15–30 min before the child wakes up in the middle of the night) were found to be probably efficacious as a result of lack of empirical research to identify these as efficacious (Mindell, 1999; Kuhn & Elliot, 2003). As these review studies were conducted more than ten years ago there is a need to conduct a new research in this
area. Although a recent meta-analysis results indicated behavioral sleep interventions as effective, the study was unable to identify which strategies were the most effective (Kempler et al., 2016). Thus, it is crucial to examine the most effective strategies and add up-to-date information to the literature. Identifying the essential components is important in order to eliminate ineffective features and improve intervention effectiveness.
In the present study, I aim to examine whether behavioral sleep interventions for children aged 0-6 years old are effective in reducing sleep problems. As the previous studies indicated contrasting results about the effectiveness of behavioral sleep interventions, this study will shed light upon this matter. Moreover, this study will provide an up-to-date review about the effectiveness of behavioral sleep interventions. Secondly, I aim to identify the most effective components of these interventions. There is a lack of this matter in the literature. This study will fill the gap and identify the most effective components of behavioral sleep interventions. In doing so, important insight will be gained to be able to see what components contribute to the effectiveness of interventions and how interventions can be best designed to reach potential effectiveness. This study will provide evidence to include the most effective components while designing behavioral sleep interventions. Therefore, research questions of this study include (1) Are behavioral sleep interventions effective in reducing sleep problems among children aged between 0-6? (2) If so, what are the most effective components of behavioral sleep interventions to reduce sleep problems among children aged between 0-6? I hypothesized that sleep interventions are effective in reducing sleep problems among children aged between 0-6. Secondly, I hypothesized that graduated extinction and sleep education are the most effective components of sleep interventions.
Method Literature search
The electronic databases PsychINFO, Medline, Web of Science were searched for articles, books, chapters, dissertations, and reports. Studies were collected by using the following combination of keywords in the title or abstract: sleep OR bed time OR bedtime OR insomni OR dyssomni OR intervention OR program OR training OR coaching OR treat OR control group OR random OR RCT OR therapy OR therapies OR workshop OR course OR manage OR fade OR fading OR extinction OR cry it out OR routine OR infant OR baby OR babies OR toddler OR child OR pediat OR paediat OR kid OR kids OR girl OR boy OR preschool OR parent OR mother OR maternal OR father OR paternal OR caregiver OR caretaker OR course OR manage OR fade OR fading OR extinction OR cry it out OR routine. The search provided 1516 relevant studies.
Study selection and inclusion criteria
Studies were selected if they met the following criteria. Firstly, studies that included behavioral sleep interventions designed for children 0-6 years of age and non-clinical group. Second criteria was studies must contain at least one sleep-related outcome of the behavioral sleep intervention. Third, only randomized control trial (RCT) studies were included. Randomized control trials were selected in order to increase the statistical power of the study. RCT's can be considered as the golden standard for studies that examine the effectiveness of interventions (Farrington 2003). Due to the limited time allowed for this research, only the past 10 years' studies had been included for the analysis. Lastly, studies written in English were selected.
Of the 1516 studies, 1437 of them were excluded. Reasons for exclusion included; school and medical interventions, outcomes that are not related to sleep, behavioral sleep interventions for children older than 6 years of age, the clinical group (disorders, etc.) of children and studies
more than ten years ago. The screening of 20% of articles was done duplicate. 79 articles were identified which met the eligibility criteria. Of the 79 articles, 63 of them were excluded due to insufficient information, not including a control group and wrong age group. The selection criteria resulted in 17 studies. Flowchart of selection of studies for inclusion in this study presented (see Figure 1).
Data analysis
A meta-analysis was not conducted due to the limited time allowed for this research. In contrast, a systematic review was conducted. A structured data extraction sheet was used in order to summarize the eligible studies. The data extraction sheet outlined the following: author, year, study design, age of children, number of children, intervention strategies, measurement, intervention effects, and limitations. As the study is limited to behavioral sleep interventions for children 0-6 years of age, the age of children was identified in each study. To answer the research questions, special attention was paid to the calculation of effect sizes in each study and identification of the intervention strategies. Comparisons were made between three different outcomes (sleep onset latency, night awakenings, sleep duration).
Results Eligible Studies According to Inclusion Criteria
Seventeen articles were identified which met the inclusion criteria. Table 1 illustrates a summary of the articles reviewed, including the author, year, study design, age of children, number of children, intervention strategies, measurement, intervention effects, and limitations.
Study Design/Measurement
All study designs included randomized controlled trials (RCT). The studies differed in terms of measurement number and method. Eight studies used two measurements (Yoong et al., 2018; Gradisar et al., 2016; Hall et al., 2015; Hiscock et al. 2014; Mindell, Telofski, Wiegand, & Kurtz, 2009; Galland et al., 2017; Stremler et al., 2013; Mindell et al., 2011) whereas nine studies used a single measurement to measure sleep outcomes (Hiscock et al., 2018; Donmez & Temel, 2019; Mindell et al., 2018; Wilson, Miller, Bonuck, Lumeng, & Chervin, 2014; Paul et al., 2016; Quach, Hiscock, Ukoumunne, & Wake, 2011; Salisbury et al., 2012; McRury & Zolotor, 2010; Price, Wake, Ukoumunne, & Hiscock, 2012). Different measurement combinations were made in the studies. Of the eight studies that used multiple measurements, four of them used both sleep diary and actigraphy (Gradisar et al., 2016; Hall et al., 2015; Galland et al., 2017; Stremler et al., 2013); one study utilized both accelerometers and sleep diary (Yoong et al., 2018); one study used both survey and Infant Behavior Diaries (Hiscock et al. 2014); one study used both sleep diary and Brief Infant Sleep Questionnaire (BISQ) (Mindell et al., 2009); and one study used both Brief Infant Sleep Questionnaire (BISQ) and Pittsburgh Sleep Quality Index (PSQI) (Mindell et al., 2011) for the measurement method. Although the studies used a different combination of measurements, sleep diaries appeared to be the most preferable measurement in the studies.
Regarding the studies utilized single measurement, three of them used sleep diaries (Hiscock et al., 2018; Donmez & Temel, 2019; Wilson et al., 2014); two studies used the Brief Infant Sleep Questionnaire (BISQ) (Mindell, Lee, Leichman, & Rotella, 2018; Paul et al., 2016); two studies used Child Sleep Habits Questionnaire (Quach et al., 2011; Price et al., 2012); one study used Baby’s Day Diary (McRury & Zolotor, 2010); and one study used Infant Behavior Diaries (Salisbury et al., 2012).
Overall, the results show that there are different methods to measure children’s sleep outcomes and the studies mostly relied on parent reports. Only five of the studies included objective measurements (Gradisar et al., 2016; Hall et al., 2015; Galland et al., 2017; Stremler et al., 2013; Yoong et al., 2018). Parent report of child sleep problems and intervention outcomes may lead to response bias which could affect the study results. Therefore, it is important to know whether the studies used subjective or objective measurements in order to discuss the accuracy of the studies.
Age of children
As one of the inclusion criteria was that behavioral sleep interventions designed for children 0-6 years of age, the age of children in each study were identified. It is interesting to note that most studies included newborns or infants in the study (Galland et al., 2017; Gradisar et al., 2016; Hall et al., 2015; Hiscock et al., 2014; Paul et al., 2016; Salisbury et al., 2012; Stremler et al., 2013; Dönmez & Temel, 2019; McRury & Zolotor, 2010; Mindell et al., 2011). The remaining four studies included children at preschool age (Yoong et al., 2018; Hiscock et al., 2018; Wilson et al., 2014; Quach et al., 2011); two studies included both infants and toddlers (6 months- 36 months) (Mindell et al., 2018; Mindell et al., 2009); and one study included both infants and pre-schoolers (4 months- 6years) (Price et al., 2012). Identification of age groups in the interventions shows that studies mostly included newborns or infants which could be due to the prevalence of sleep problems in younger ages (Meltzer et al., 2015).
Intervention strategies
Intervention strategies varied widely, and four different strategies were identified including sleep education, graduated extinction, bedtime routines, and nightly massages. Most commonly used intervention strategy was sleep education (Hall et al., 2015; Hiscock et al., 2014; Hiscock et
al., 2018; Paul et al., 2016; Quach et al., 2011; Salisbury et al., 2012; Stremler et al., 2013; Wilson et al., 2014; Dönmez & Temel, 2019; McRury & Zolotor, 2010; Price et al., 2012; Galland et al., 2017). Sleep education interventions mostly focused on the benefits of sleep, the recommended amount of sleep for young children, effects of inadequate sleep for infants and parents, sleep cycles, sleep hygiene strategies, and responses to night awakenings (Hall et al., 2015; Hiscock et al., 2014; Hiscock et al., 2018; Paul et al., 2016; Quach et al., 2011; Salisbury et al., 2012; Stremler et al., 2013; Wilson et al., 2014; Price et al., 2012; Galland et al., 2017). Moreover, 4S soothing techniques (swaddling, holding at side or stomach position, shushing-white noise, and swinging) were taught in two of the interventions (Dönmez & Temel, 2019; McRury & Zolotor, 2010). Sleep education strategies differed in terms of the delivery method. Several interventions used group teaching method (Hall et al., 2015; Donmez & Temel, 2019; Salisbury et al., 2012; Wilson et al., 2014; Galland et al., 2017) and one on one consultation method (Hiscock et al., 2018; Paul et al., 2016; Quach et al., 2011; Price et al., 2012) to deliver sleep education interventions. Remaining studies used a videotape (McRury & Zolotor, 2010) and multiple delivery methods which were one on one consultation and a booklet (Stremler et al., 2013) and DVD, booklet and group teaching method (Hiscock et al. 2014). Since the sleep education strategies are various, identifying them by given information and delivery methods would make it possible to examine which sleep education strategy has more effect on a child's sleep problems.
Regarding the other intervention strategies, two of them utilized multiple components including “nightly massage and bedtime routines” (Mindell et al., 2018) and “sleep education and bedtime routines” (Yoong et al., 2018). Although the multiple components can help to improve the uptake of safe sleep messaging, it makes it difficult to determine which component was more effective. Gradisar et al. (2016) tested two different interventions, which were graduated extinction
intervention and bedtime routines intervention, with a control group. The remaining intervention strategies included bedtime routines (Mindell et al., 2009; Mindell et al., 2011). Since there were different interventions with different components, identification of these interventions allows to determine which intervention strategy is the most effective by looking at their effect sizes.
Intervention effects
The intervention effects showed mixed results. Of the ten studies that included newborns or infants, seven of them found positive effects (Gradisar et al., 2016; Hall et al., 2015; Paul et al., 2016; Dönmez & Temel, 2019; Mindell et al., 2011; Hiscock et al., 2014; Salisbury et al., 2012) whereas three found non-significant effects (Galland et al., 2017; Stremler et al., 2013; McRury & Zolotor). The four studies that included preschoolers found positive effects (Hiscock et al., 2018; Quach et al., 2011; Yoong et al., 2018; Wilson et al., 2014). Similarly, two studies which included both infants and toddlers found positive effects (Mindell, Lee, Leichman, & Rotella, 2018; Mindell et al., 2009). Lastly, the study that included both infants and preschoolers found non-significant effects (Price et al., 2012). Overall, thirteen studies indicated positive effects (Gradisar et al., 2016; Hall et al., 2015; Hiscock et al., 2018; Mindell, Lee, Leichman, & Rotella, 2018; Paul et al., 2016; Quach et al., 2011; Salisbury et al., 2012; Yoong et al., 2018; Dönmez & Temel, 2019; Mindell et al., 2011; Mindell et al., 2009; Wilson, Miller, Bonuck, Lumeng, & Chervin, 2014; Hiscock et al., 2014) while four studies found non-significant effects on sleep outcomes (Galland et al., 2017; Stremler et al., 2013; McRury & Zolotor, 2010; Price et al., 2012). Effect sizes were calculated in each study in order to identify the most effective intervention strategy. However, the calculation of effect sizes was not possible in two studies due to insufficient information (Salisbury et al., 2012; Wilson et al., 2014). The outcomes of the studies varied, and in the upcoming paragraphs,
the effect sizes for three different outcomes (sleep onset latency, night awakenings, and sleep duration) will be discussed (see for an overview: Table 2).
Sleep onset latency. The largest effect for sleep onset latency was found in the study of Mindell et al. (2009) for bedtime routines intervention for toddlers’ sample (d=1.09) however, this association was small for infant sample in the same study (d=0.29) (Mindell et al., 2009). Gradisar et al. (2016) also found large effects for both bedtime routines intervention and graduated extinction intervention (d=1.04, d=0.87, respectively). Although the two studies found large effects for bedtime routines interventions, Mindell et al. (2011) found small effects (d=0.20). In addition, one sleep education intervention showed small effects on sleep onset latency (d=0.1) (Hiscock et al., 2018). The largest improvements for sleep onset latency were provided by interventions with bedtime routines and graduated extinction interventions.
Night awakenings. The largest effect for night awakenings was found for graduated extinction intervention (d=1.98) (Gradisar et al., 2016). The remaining studies that found an effect for night awakenings only indicated small effects. Mindell et al. (2009) found small effects for bedtime routines intervention for both infants and toddlers (d=0.47, d=0.48, respectively). Similarly, other bedtime routines intervention also showed small effects in reducing night awakenings (d=0.21) (Mindell et a., 2011). For interventions with sleep education strategy only small effects were found (d=45, d=0.11, d=0.04) (Hall et al., 2015; Donmez & Temel, 2019; Hiscock et al., 2018). Lastly, Mindell et al. (2018) found small effects for bedtime routines and nightly massages intervention (d=0.08).
Sleep duration. The largest effect for sleep duration was found in the intervention with sleep education and bedtime routines strategies (d=0.9) (Yoong et al., 2018). Medium effect size was found for sleep education intervention (d=0.52) (Donmez & Temel, 2019). The remaining
sleep education interventions found only small effects (d=0.2, d=0.42, d=0.42) (Hiscock et al., 2018; Paul et al., 2016; Quach et al., 2011, respectively). It is notable that medium effect sizes were found with the group teaching method while small effects were found with one on one consultation method for sleep education strategies. Similarly, one bedtime routines intervention showed small effects for sleep duration (d=0.34) (Mindell et al., 2011).
All strategies showed small to large effects in reducing sleep problems among children. Graduated extinction, bedtime routines and combination of sleep education and bedtime routines strategies showed the largest effect sizes. Graduated extinction showed large effects in two domains which are sleep onset latency and night awakenings. Bedtime routines strategy showed large effects for sleep onset latency in two studies. Intervention with multiple strategies (bedtime routines and sleep education) indicated large effects for sleep duration. Although the intervention with sleep education and bedtime routines strategies indicated large effects, sleep education strategy alone showed only small improvements in the studies. This could show that education itself might not be enough for the improvements. The given information should be supported by practice.
To conclude, as the graduated extinction strategy showed large effects in two domains it seems like the most effective strategy compared to other strategies. For the sleep duration improvement intervention with multiple strategies (bedtime routines and sleep education) showed the greatest effect. However, it is not clear whether bedtime routines or sleep education strategy was the most effective. As indicated above sleep education interventions showed only small improvements, therefore the large effect size for the intervention with multiple components (sleep education and bedtime routines) could be the effect of bedtime routines strategy. For the overall
most effective components, it can be concluded that graduated extinction and bedtime routines are the most effective strategies of behavioral sleep interventions.
Study Limitations
All articles reported study limitations. The limitations included: (1) self-report of caregiver (Hiscock et al., 2014; Hiscock et al., 2018; Mindell et al., 2018; Mindell et al., 2009; Paul et al., 2016; Quach et al., 2011; Salisbury et al., 2012; Wilson et al., 2014; Mindell et al., 2011; Yoong et al., 2018; Dönmez & Temel, 2019) which may result in biased responses; (2) small sample size (Gradisar et al., 2016; Wilson et al., 2014; Yoong et al., 2018; McRury & Zolotor, 2010) which may result in reduced generalizability; (3) lack of blinding (Hall et al., 2015; Hiscock et al., 2014) which may underestimate the effect size of the outcome, (4) lack of follow-up (Mindell et al., 2018; Mindell et al., 2009; Mindell et al., 2011) or large lost-to follow-up numbers (Price et al., 2012) which may result it not knowing whether the improvements in sleep were maintained, (5) limited generalizability due to only English speaking sample (Hall et al., 2015; Hiscock et al., 2018; Hiscock et al., 2014), well-educated sample (Galland et al., 2017) and socially advantaged sample (Stremler et al., 2013) or low-income sample (Wilson et al., 2014) and homogenous sample (Paul et al., 2016); (5) not measuring sleep onset outcomes (Hall et al., 2015) or measuring only a specific construct (bedtime) (Mindell et al., 2018; Mindell et al., 2009).
By looking at the limitations of the studies the generalizability of results can be discussed. Study limitations may have an impact on outcomes. Therefore, it is important to consider this while interpreting the intervention outcomes. The results of this study indicate that many of the studies have the same limitations. The most common limitation was self-report of a caregiver. This limitation may have resulted in biased intervention outcomes. Hence, it is an important point to take into consideration while interpreting the effectiveness of interventions.
Discussion
The goal of this study was to investigate the effectiveness of behavioral interventions and identify the most effective strategy. The results of this study indicated that 76% (13 of 17) of the studies found positive effects of sleep interventions ranging from small to large, while four studies showed no significant effects. This result is in line with the first hypothesis which was sleep interventions are effective in reducing sleep problems among children aged between 0-6. Secondly, it was hypothesized that graduated extinction and sleep education are the most effective strategies of sleep interventions. The results are partly in line with the second hypothesis. Graduated extinction and bedtime routines were found to be the most effective strategies of behavioral sleep interventions. Although the most common strategy was sleep education, only small improvements were found for sleep education interventions.
According to the study results, behavioral sleep interventions are effective in reducing sleep problems among children aged between 0-6. All of the identified intervention strategies (graduated extinction, bedtime routines, sleep education, and nightly massages) showed effects ranging from small to large. The results of this study suggest that graduated extinction and bedtime routines are the most effective strategies of behavioral sleep interventions. The findings are consistent with the results of three systematic reviews. All of the systematic review studies indicated positive effects of behavioral sleep interventions in reducing sleep problems among children (Mindell, 1999; Kuhn & Elliot, 2003; Mindell et al., 2006). For the most effective strategies, Mindell (1999) found that sleep education strategy was a well-established intervention, graduated extinction strategy was probably efficacious and bedtime routines strategy was considered as a promising intervention. In another review, graduated extinction and sleep education strategies were considered as well-established interventions and bedtime routines
intervention was a promising intervention (Kuhn & Elliot, 2003). Furthermore, Mindell et al. (2006) found support for both graduated extinction and sleep education strategies. Two studies indicated sleep education strategy as a well-established intervention and one study found strong support. However, this strategy did not find strong support in the present study. This study suggests that sleep education strategy alone may not be enough to improve sleep problems of children. Sleep education interventions should provide practical strategies along with the given information in order to be more successful. The graduated extinction strategy found strong evidence in all reviews as expected. For the bedtime routines, although the previous studies considered this strategy as a promising intervention, stronger support was found for this strategy in the present study. As an explanation for these consistent findings, it seems more effective to enable the child to develop self-soothing skills with graduated extinction strategy and to teach children appropriate behaviors and control of arousal with bedtime routines strategy. Moreover, it is also possible that more actions were shown by parents with accurate strategies that showed directions such as checking on the child after waiting 10 minutes (graduated extinction), doing similar activities in a similar way each night (bedtime routines). Thus, children were trained more effectively with these interventions. Overall, the existing literature indicated behavioral sleep interventions as beneficial in reducing sleep problems and graduated extinction and bedtime routines found support in all reviews. Unlikely to present study, sleep education strategies showed better results in two review studies. The difference between the existing reviews and the present study could be due to missing information about the effect sizes of two sleep education interventions in the present study.
This study has limitations to be considered. First, despite a large number of studies in the initial search, I was able to identify only seventeen studies for inclusion. Many studies were not RCTs therefore, needed to be excluded to make sure the review was based on the best available
evidence. Moreover, only studies of the past ten years were included which resulted in having a limited number of studies to include. Time limitation criteria was made in order to eliminate the articles due to insufficient time to identify all of them. This could have affected the results. The eliminated studies might have indicated large results for other strategies. Intervention effectiveness ratio and the most effective strategy could be different if all the studies were included. Second, the reliability and objectivity of the sleep data was another obstacle. The studies mainly used parent reports to record child sleep, which are not as reliable as objective measurements (e.g., actigraphy) and more open to bias. Only five studies used both subjective and objective measurements and it is notable that objective measurement results were identical to the results with subjective measurements (e.g., sleep diaries) only in three studies. It is possible that children could have learned not to cry immediately when they wake up during the night rather than learn to improve their sleep (Kempler et al., 2016). Third, this study only included the non-clinical group of children therefore, the results are not generalizable for the clinical group. Lastly, the effect sizes of the two interventions couldn't be calculated as the F statistics (Salisbury et al., 2012) and standard deviation (Wilson et al., 2014) were not given. If it was possible to calculate the effect sizes in these studies the results might have shown a difference.
Despite these limitations, this study has some strengths. Multiple databases were searched including PsychINFO, Medline, Web of Science in order to find all related studies. Inclusion and exclusion criteria were clearly stated. This provided external and internal validity of the study as well as increased the likelihood of finding a true relationship between intervention and outcomes. The study included only RCTs which is the most powerful study design in intervention research. This has improved the strength of the study. Moreover, the screening of 20% of articles was done
duplicate in order to increase the reliability of the study. The selected studies were presented with a flowchart to show the selection process clearly.
There are multiple possible directions for future research. A meta-analysis is needed to examine the most effective components of effective behavioral sleep interventions. Furthermore, the addition of objective assessment tools, such as actigraphy, is needed in future studies in order to increase reliability. The clear descriptions of the interventions are mostly missing in the studies. Future studies should include more information about the interventions. This could be helpful to identify the components and be able to make better comparisons. Identification of effective strategies has important implications for clinical practice. This information could be used to select interventions with strategies that are associated with greater intervention effectiveness. Moreover, it could be possible to eliminate the ineffective components thus, minimizing the burden on practitioners and families. As explained above high prevalence rates of sleep problems among children is a concerning problem. Many behavioral sleep interventions exist, and it is important to identify the most effective ones in order to reduce sleep problems among children. In the present study, graduated extinction and bedtime routines strategies showed the greatest improvements for sleep problems of children. This study suggests that intervention planners should opt these most effective strategies to train and educate parents better. Moreover, parents may show greater confidence to imply the strategies which were proved to have greater effects. Hereby, children's sleep problems could show more reductions and effectiveness of behavioral sleep interventions could increase. In addition, the intervention planners should pay attention to the generalizability of existing study findings.
To conclude, this review has indicated that behavioral sleep interventions have small to large effect on children’s sleep problems. Specifically, graduated extinction strategy and bedtime
routines strategy have received strong empirical support. Majority of children respond positively to these behavioral techniques and showed fewer sleep problems. Intervention planners should pay particular attention to these results and tailor interventions that have larger effects. It is essential that future research includes creative methods for measuring sleep outcomes, beyond just subjective measurements.
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Figure 1 Flowchart of selection of studies for inclusion in systematic review Records identified through
database searching (n = 1605) S cr ee n in g In clu d ed E li g ibi li ty Id en tif icat ion
Records after duplicates removed (n = 949 + Loes Screening)
Records screened
(n = ? ) Records excluded
(n = 1588)
Full-text articles assessed for eligibility
(n = 60)
Full-text articles excluded, with reasons (n = 43 ) Reasons: 1. Not enough information 2. No control group 3. Wrong age
4. Wrong study design Studies included in
qualitative synthesis (n = 17)
Abstracts not meeting inclusion criteria
, T ab le 1 Studi es include d in s ynthesis
T ab le 2 Summ ary of effec t si ze s o f the studi es
