Longitudinal change in sleep, functional, and behavioural characteristics in a cohort of children with Down syndrome.

This paper describes the longitudinal change in sleep, functional, and behavioural characteristics in a cohort of children with Down syndrome, including the effect of sleep interventions in a subset. A prospective longitudinal cohort study was undertaken in children with Down syndrome aged 3-16 years comparing (1) children referred to a tertiary sleep medicine clinic who received sleep hygiene advice and an additional sleep treatment (DSref_I) with (2) children attending the same clinic who only received sleep hygiene advice (DSref_N) and (3) children recruited from the community who, were not receiving any treatment (DScomm). Data collected included demographic and medical history information, Child Sleep Habits Questionnaire-Abbreviated (CSHQ-A), Life-Habits Questionnaire (Life-H) and Child Behaviour Checklist (CBCL) at baseline and then 6-monthly for a total of 18 months. Any sleep interventions during this time were recorded. A total of 57 children were included (DSref_I, n = 16; DSref_N, n = 25; DScomm, n = 16). At recruitment, the median CSHQ-A total score was high (>41) in all three subgroups, but highest in the DSref_I subgroup (median [interquartile range] Dsref_I score 58 [53-66] versus DSref_N score 49 [43-53], p = 0.019). Although improved, 80% of participants in the DSref_I subgroup still had a CSHQ-A total score >41 at the last assessment point. The median total Life-H and total CBCL scores were not significantly different between groups at baseline and there was no significant time, group, or interaction effect seen through the study. Over an 18-month period, sleep problems were seen to persist in children with Down syndrome. Treatment resulted in only modest improvements in sleep.

Parents' Experiences of Having a Child with Down Syndrome and Sleep Difficulties.

OBJECTIVES: Sleep disorders are prevalent in children with Down Syndrome (DS). However, sleep treatment is not always readily accessed by this group. This study aims to understand families' experiences of having a child with DS and sleep difficulties, and in particular, their healthcare experiences, with the goal of informing practice improvements. METHODS: We conducted semi-structured interviews with 34 parents (fathers n = 4 and mothers n = 30) with open-ended questions about parents' experiences of sleep, family dynamics, and healthcare. We operationalized a reflexive Thematic Analysis. RESULTS: Parents normalized their experiences of having a child with DS and sleep problems. Parents acknowledged that sleep disruption has adverse and pervasive impacts on their wellbeing and family dynamics, but also found this difficult to identify as a health problem. They accepted sleep difficulties as a regular part of bringing up any child, particularly one with a disability. When they did seek treatment for their child's sleep difficulties, parents often reported encountering insensitive and inadequate care and described that, at times, healthcare professionals also normalized children's sleep difficulties, resulting in sub-optimal treatment. This included at times failure to refer to tertiary sleep medicine services when required. CONCLUSIONS: Parents' and healthcare professionals' normalization of sleeping difficulties denies that they are both deleterious and modifiable. Practice implications include raising healthcare professionals' awareness of the importance of proactively addressing sleep, with sensitivity to families' normalization strategies, recognizing that families may require prompting to report concerns.

Management of residual OSA post adenotonsillectomy in children with Down Syndrome: A systematic review.

STUDY OBJECTIVE: The aim of this systematic review is to summarise the existing literature regarding management of residual obstructive sleep apnoea post adenotonsillectomy in children with Down Syndrome. METHODS: PubMed, EMBASE, Cochrane and Scopus were searched to retrieve all studies published up to May 2020 regarding the management of obstructive sleep apnoea in children with Down Syndrome post adenotonsillectomy. RESULTS: Eight studies were identified including five retrospective studies, two prospective and one case series. Five articles addressed surgical treatments only, two articles addressed hypoglossal nerve stimulation and one article addressed both surgical and medical treatments. Findings were similar across most studies where the obstructive apnoea hypopnoea index (oAHI) was reduced post intervention however given the heterogeneity in methodology and patient demographics, results were not directly comparable. Limited evidence was found regarding BMI and outcome post-surgery, suggesting that those who were normal weight or overweight had a significant reduction in the oAHI, compared to those who were obese. CONCLUSION: The most optimal treatment for the management of residual obstructive sleep apnoea remains uncertain. There are various treatment options described in the literature, however, they have not been thoroughly studied, with only eight relevant articles found. Studies comparing outcomes following different treatment interventions and using consistent parameters are required to gain further understanding of the most optimal treatment for residual obstructive sleep apnoea in this population. Understanding if specific phenotypic features, such as weight, influence response to therapy will also be important. This information will help to develop treatment algorithms, which will assist in improving patient care by directing treatment and ensuring a more consistent approach to overall management. The high prevalence of residual obstructive sleep apnoea in children with Down Syndrome following first line treatment makes this a clinically important area to target in this particular cohort of children to avoid long term complications of untreated OSA.

Sleep, Function, Behaviour and Cognition in a Cohort of Children with Down Syndrome.

OBJECTIVE: To describe the sleep problems experienced by children with Down syndrome attending a tertiary sleep clinic and relationship with behaviour, function and cognition. METHODS: Data were collected from children with Down syndrome aged 3-18 years old. Carers completed the Abbreviated Child Sleep Habits Questionnaire, Child Behaviour Checklist and Life-Habits Questionnaire at enrolment. Cognitive assessment (Stanford-Binet 5) was undertaken by a trained psychologist. Children received management for their sleep problem as clinically indicated. RESULTS: Forty-two subjects with a median age of 6.8 years (Interquartile Range-IQR 4.5, 9.8) were enrolled. A total of 92% were referred with snoring or symptoms of Obstructive Sleep Apnoea (OSA), with 79% of those referred having had previous ENT surgery. Thus, 85% of all participants underwent a sleep study and 61% were diagnosed with OSA (OAHI ≥ 1/h). Based on questionnaires, 86% of respondents indicated that their child had a significant sleep disorder and non-respiratory sleep problems were common. Non-respiratory problems included: trouble going to sleep independently (45%), restless sleep (76%), night-time waking (24%) and bedtime resistance (22%). No significant correlations were found between sleep measures (behavioural and medical sleep problems) and the behavioural, functional or cognitive parameters. CONCLUSION: Sleep disorders were very common, especially non-respiratory sleep problems. OSA was common despite previous surgery. No association was found between sleep-related problems (snoring, sleep-study-confirmed OSA or non-respiratory sleep problem) and parent-reported behavioural problems, functional impairments or intellectual performance. This may reflect limitations of the measures used in this study, that in this population ongoing problems with daytime function are not sleep related or that a cross-sectional assessment does not adequately take into account the impacts of past disease/treatments. Further research is required to further evaluate the tools used to evaluate sleep disorders, the impact of those disorder on children with Down syndrome and interventions which improve both sleep and daytime function.

Sleep problems in Australian children with Down syndrome: the need for greater awareness.

BACKGROUND: Children with Down Syndrome (DS) have a high prevalence of obstructive sleep apnoea (OSA). Non-respiratory sleep disorders also occur commonly but are less well recognised. This cross-sectional study evaluates the prevalence of sleep difficulties in a community sample of Australian children with DS (DScomm), using the Children's Sleep Habits Questionnaire (CSHQ), and compares them to children referred to the sleep clinic (DSref). To our knowledge this is the first study to have reported prevalence of sleep problems in Australian children with DS and to compare a community and referred group of children with DS directly. METHODS: The CSHQ was completed by parents of children with DS recruited from the community (DScomm) via survey distributed by Down syndrome Queensland and Australia. A second group was recruited through the tertiary sleep clinic at our institution (DSref) and completed the same questionnaire on enrolment. Data from these groups was compared. RESULTS: There were 76 participants in the DScomm group (57% male; median age 9.7yrs) and 42 participants in the DSref group (50% male; median age 6.97yrs). The overall prevalence of sleep disturbances was 90.9% in the DScomm group, and 85.7% in the DSref group (p = 0.54). There was a statistically significant difference in the mean total CSHQ score, with the DScomm having the higher score (p = 0.023). CONCLUSIONS: This study reports a high prevalence of sleep problems in both a community and referred group of Australian children with DS and suggests that there are many children with DS with sleep problems, particularly non-respiratory difficulties, who are potentially not receiving adequate treatment.

The impact of sleep problems on functional and cognitive outcomes in children with Down syndrome: a review of the literature.

STUDY OBJECTIVES: The aim of this review was to summarize the existing literature on the association between sleep problems and cognition, function, and behavior in children with Down syndrome. METHODS: Embase, PubMed, CINAHL, and PsychINFO databases were searched to retrieve all studies published between 1990 and 2018 that evaluated the relationship between sleep and cognition, function, or behavior in children with Down syndrome. RESULTS: Fifteen articles were included, which were mostly of a cohort or case-controlled design. Five articles addressed sleep and cognition only, 6 reported on sleep and behavior, and only 1 reported on sleep and functional ability. Three papers evaluated sleep and both cognition and behavior. Findings varied across studies with methodological differences, making it difficult to directly compare results. The association between sleep and behavior or cognition in children with Down syndrome remains uncertain, but a large study in 110 children provides strong evidence of a negative impact of sleep disorders on the accomplishment of daily life habits. CONCLUSIONS: The impact of coexisting sleep disorders in children with Down syndrome has not been widely studied, with only 15 relevant studies found through an extensive literature review. Large well-designed studies are required to fully understand this relationship further. This is important as sleep-disordered breathing and difficulties with sleep patterns and routines are highly prevalent in children with Down syndrome. Sleep may be one of the few treatable factors that can assist in improving long-term outcomes in this population.