The Lighter Touch: Less-Restriction in Sequentially Implemented Behavioral Sleep Interventions for Children with Rare Genetic Neurodevelopmental Conditions

Abstract

Purpose The prevalence of sleep difficulties among children with rare genetic neurodevelopmental conditions (RGNC) is high. Behavioral interventions are commonly used in the treatment of sleep difficulties in children with neurodevelopmental conditions such as autism, however, research is scarce in children with RGNC. The range of co-occurring complexities within this population, means there is a need for research to not only determine the effectiveness of behavioral sleep interventions, but also which components might be the least restrictive (i.e., intensive/aversive) and minimally sufficient. Methods This study used a single-case multiple baseline design to investigate the effectiveness and acceptability of behavioral sleep interventions, indicated within a Functional Behavior formulation in eight children with RGNC (M = 7.3 years). Intervention components were sequentially administered across up to three phases, based on the principle of less restriction (from least to relatively more intensive) to determine what might be minimally sufficient. Results Results showed an improvement in sleep onset latency, night wakings, early morning waking and unwanted bed-sharing for 7/7, 6/7, 3/3 and 3/3 children respectively. Improvement was observed for most participants following the less restrictive phases of intervention (circadian modifications, antecedent modifications and positive reinforcement), however, more restrictive, albeit modified, extinction procedures were still implemented for five participants. Improvements were maintained at follow-up and interventions were deemed acceptable to parents. Conclusions Less restrictive function-based behavioral strategies are an effective, and in some cases sufficient, contribution to a sequence of interventions for a range of sleep difficulties. They should be implemented first, before more restrictive strategies.

Behavioral Sleep Interventions for Children with Rare Genetic Neurodevelopmental Conditions: A Retrospective Analysis of Overall Outcomes for 26 Cases

“…This study is a retrospective analysis of case data collected within a series of single case experimental studies (e.g., Woodford et al, 2022;2024a), similar to that conducted by McLay et al (2020). Case data was gathered in the context of non-concurrent, multiple-baseline across participants experimental designs wherein all participants were randomly allocated pre-determined baseline lengths (namely, 7, 14, or 21 days).…”

“…Of the 26 participants (16 males; 10 females), 13 were aged ≤ 6 years, 9 were between 6 and 12 years, and 4 were > 12 years (M = 7.0, SD = 4.2). This included 13 participants from previously published studies (Woodford et al, 2022;2024a) and 13 participants who did not meet the specific study criteria of these previous studies (N = 6) and/or who withdrew during intervention (N = 9). Inclusion of all participants regardless of whether they adhered to or completed intervention was important for determining the overall, relatively unbiased clinical effectiveness of behavioral sleep intervention (Lachin, 2000).…”

“…Parents were also able to contact the researchers for ad hoc advice as required. The principle of less restriction (Johnston & Sherman, 1993) was used to guide intervention planning in most cases, so that consequence modifications were the last option in the sequence of interventions given their complexity and difficulty (Woodford et al, 2022(Woodford et al, , 2024a. Parents were provided revised recommendations both verbally and in written format via email, with systematic formative evaluation of intervention progress.…”

“…The measures described contributed to varying components of data analysis for the 26 participants. In summary, data from sleep diaries and the derived SPS scores were used as the primary outcome measures, while CSHQ global scores were used as a secondary outcome measure in this study; data from sleep diaries and video recordings were the primary measures used for individual time-series analyses such as in (Woodford et al, 2022;2024a) and interobserver agreement and treatment fidelity calculations; and data from the questionnaires assessing child psychological variables were used for analysis of potential moderators of intervention response in this study and for the analysis of collateral effects in a separate study (Woodford et al, 2024b).…”

“…Existing research is limited to individual case studies or small N projects (Curfs et al, 1999;Didden et al, 1998;Woodford et al, 2022) and has rarely included adolescents, and interventions have typically been generic rather than being individualized for each case (Allen et al, 2013;Weiskop et al, 2005). The current study is a retrospective review of 26 participants with RGNC who received function-based behavioral sleep interventions evaluated with experimental, single-case research designs (e.g., Woodford et al, 2022;2024a). Overall outcomes, possible moderators of intervention response (demographic and child characteristics) and the social validity of the interventions were examined.…”