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Summary 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. [ABSTRACT FROM AUTHOR]

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. 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. 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. 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. [ABSTRACT FROM AUTHOR]

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. [ABSTRACT FROM AUTHOR]

Obstructive Sleep Apnoea (OSA) is common in children with Down Syndrome (DS). Adenotonsillectomy is regarded as first line treatment for OSA but does not always lead to resolution of symptoms in this group of children. Management of residual OSA is variable with no existing recommendations to guide clinical practice. To describe the experience of a large tertiary sleep service in managing residual OSA in children with DS following upper airway surgery (adenotonsillectomy, adenoidectomy or tonsillectomy). A retrospective study of children who were under evaluation at the Queensland Children's Hospital sleep medicine department between October 2013 to April 2022 for residual OSA, after upper airway surgery was undertaken. 148 children with DS who underwent polysomnography for evaluation of OSA were identified. 100 were included in this study and of these, 77 underwent adenotonsillectomy, 19 adenoidectomy, and 4 tonsillectomy. Post-surgical PSG data of all 100 children showed residual mixed sleep disordered breathing in 68 children. 41 were recommended CPAP following surgery, while 21 underwent further surgery. Residual OSA was confirmed to be highly prevalent in children with DS who had already undergone upper airway surgery for OSA. This study identified that CPAP is possible to establish in most children with DS and can be used to manage residual OSA in this population. Approximately one-third of this group were able to discontinue therapy at a median duration of 18 months, suggesting resolution of disease with time can occur in some children. • Improvement in OSA severity post-initial surgery seen in 62% of children. • Residual moderate-severe OSA seen following surgical management in 33% of children. • 82.9% of children initiated on CPAP therapy were able to sustain it for >12 months. • 31.7% of children commenced on CPAP were able to cease after 15 months. [ABSTRACT FROM AUTHOR]

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. 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. 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. 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. [ABSTRACT FROM AUTHOR]

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. [ABSTRACT FROM AUTHOR]

Families of children with Down syndrome experience complex lives and needs, yet the few existing studies on these families are written in conventional academic prose that is not optimal for knowledge translation beyond academia, particularly for busy healthcare professionals. In this paper, we Depart Radically in Academic Writing (DRAW) (Mackinlay, 2022) and present data poetry and two case studies that draw upon semi-structured interviews with mothers, fathers, and siblings, who were interviewed separately about their experiences of having a child/sibling with Down syndrome. We introduce our interdisciplinary team that includes academics and clinicians to contextualise our focus on research translation. We demonstrate that writing with creative criticality (i.e. 'DRAWing') contributes an embodied and affective understanding of research participants' stories, which is largely lacking in the academic literature on families of children with Down syndrome and the sociology of health and illness field more broadly. Moreover, DRAWing can impact audiences emotionally as well as intellectually (Richardson, 2003, p. 924), which has important knowledge translation implications for both healthcare professionals and these families. DRAWing can capture healthcare professionals' attention, prompting them to critically reflect on their practices and opportunities for improving care and treatment for these families. [ABSTRACT FROM AUTHOR]

Describe the spectrum of OSA across time in infants and children with Down syndrome. Retrospective records review of children who underwent formal polysomnography (PSG) in one of two Australian tertiary sleep centres over selected 3.5-year periods. 152 children were identified, then all sleep study and treatment records were retrieved for the lifetime of the child through 2018. 3.8 ± 3.2 studies (range 1–17) were retrieved per child and 38.2% had mild disease at worst. Children having only 1 study were more likely to have a normal or mild result than those having ≥2 (chi-square 11.25, p -value 0.0008) Studies were more often severe in children age <2 compared to those ≥2 years, (chi-square 12.87, p = 0.005). After age 2 years, OSA severity increased with age. Amongst 91 (56.4%) children with ≥2 studies, 71 (78.0%) had moderate or severe disease at some time. Studies evaluating the effects of surgery (most often adenotonsillectomy) showed resolution of disease to mild or normal in 53.3%. Where ≥2 studies were evaluated, the last study polarised towards normal or mild disease 40 (44.0%), or treatment titrations 34 (37.4%) with moderate or severe disease in 17 (18.7%). In a tertiary sleep unit, a full spectrum of sleep disordered breathing in Down syndrome was seen from infancy onwards. Children having only one study were more likely to have normal results. Children with multiple studies reflected disease surveillance, including follow-up after treatment interventions. • Moderate/severe OSA is more common in DS children attending specialist referral centres (44.4%) than in population studies. • Moderate/severe OSA is highest in infants <12 months old (68%) compared to 42% of those >2 years of age. • Reviewing published cohorts, 29.4% of children with Down syndrome studied at specialist centres do not have OSA. • Children with only one study are more likely to have normal results. • Children with multiple studies progress towards normal or treated studies over time. [ABSTRACT FROM AUTHOR]