Sleep-Related Rhythmic Movement Disorder in Young Children with Down Syndrome: Prevalence and Clinical Features

Published on Oct 1, 2021in Brain Sciences3.394
· DOI :10.3390/BRAINSCI11101326
Ceren Kose , Izabelle Wood + 6 AuthorsCatherine M. Hill39
Estimated H-index: 39
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Abstract
Sleep-related Rhythmic Movement Disorder (RMD) affects around 1% of UK pre-school children. Little is known about RMD in Down syndrome (DS). We aimed to determine: (a) the prevalence of RMD in children with DS aged 1.5–8 years; (b) phenotypic and sleep quality differences between children with DS and RMD and sex- and age-matched DS controls; and (c) night-to-night variability in rhythmic movements (RMs). Parents who previously reported RMs from a DS research registry of 202 children were contacted. If clinical history suggested RMD, home videosomnography (3 nights) was used to confirm RMs and actigraphy (5 nights) was used to assess sleep quality. Phenotype was explored by demographic, strengths and difficulties, Q-CHAT-10/social communication and life events questionnaires. Eight children had confirmed RMD. Minimal and estimated maximal prevalence were 4.10% and 15.38%, respectively. Sleep efficiency was significantly lower in RMD-cases (69.1%) versus controls (85.2%), but there were no other phenotypic differences. There was considerable intra-individual night-to-night variability in RMs. In conclusion, RMD has a high prevalence in children with DS, varies from night to night and is associated with poor sleep quality but, in this small sample, no daytime phenotypic differences were found compared to controls. Children with DS should be screened for RMD, which is amenable to treatment.
References0
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#1Anna Joyce (Coventry University)H-Index: 6
#2Heather ElphickH-Index: 16
Last. Catherine M. Hill (University of Southampton)H-Index: 39
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ABSTRACTObjective/Background: Children with Down syndrome (DS) commonly experience difficulties with executive function (EF). They are also vulnerable to obstructive sleep apnoea (OSA). OSA is asso...
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#1Soonyiu Yau (University of Southampton)H-Index: 1
#2Ruth M. Pickering (University of Southampton)H-Index: 39
Last. Catherine M. Hill (University of Southampton)H-Index: 39
view all 12 authors...
Abstract Aims To compare sleep in infants and toddlers with Down syndrome (DS) to typically developing controls, including differences in snoring and sleep ecology (sleep setting and parent behaviors). Methods Parents of 104 children with DS and 489 controls aged 6–36 months completed the Brief Infant Sleep Questionnaire (BISQ). We explored group differences, controlling for demographic variables. Results Parents of children with DS reported more sleep problems (45% v 19%), snoring (19% vs 2%), ...
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#1Markus Gall (Austrian Institute of Technology)H-Index: 6
#2Bernhard Kohn (Austrian Institute of Technology)H-Index: 10
Last. Catherine M. Hill (University of Southampton)H-Index: 39
view all 13 authors...
Unlike other episodic sleep disorders in childhood, there are no agreed severity indices for rhythmic movement disorder. While movements can be characterized in detail by polysomnography, in our experience most children inhibit rhythmic movement during polysomnography. Actigraphy and home video allow assessment in the child’s own environment, but both have limitations. Standard actigraphy analysis algorithms fail to differentiate rhythmic movements from other movements. Manual annotation of 2D v...
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#1Liliana Ruta (National Research Council)H-Index: 17
#2Flavia ChiarottiH-Index: 49
Last. Filippo Muratori (UniPi: University of Pisa)H-Index: 54
view all 16 authors...
Background The Q-CHAT is parent-report screening questionnaire for detecting threshold and sub-threshold autistic features in toddlers. The Q-CHAT is a dimensional measure, normally distributed in the general population sample and able to differentiate between a group of children with a diagnosis of autism and unselected toddlers. Objectives We aim to investigate the psychometric properties, score distribution and external validity of the Q-CHAT in an Italian clinical sample of young children wi...
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#1Rosemary S.C. Horne (Hudson Institute of Medical Research)H-Index: 51
#2Poornima Wijayaratne (Hudson Institute of Medical Research)H-Index: 1
Last. Lisa M. Walter (Hudson Institute of Medical Research)H-Index: 23
view all 4 authors...
Summary Down syndrome (DS), the most common human chromosomal malformation, has an estimated annual incidence of one in 1000 live births worldwide. Sleep problems are common in children with DS, reported by parents in up to 65% of school-aged children, significantly higher rates than in typically developing (TD) children. Problems include difficulty in sleep initiation and maintenance together with obstructive sleep apnoea (OSA) which affects up to over 90%, of DS children compared with 1–5% in ...
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#1Emily Gogo (University of Southampton)H-Index: 1
#2Rachel M. van Sluijs (ETH Zurich)H-Index: 4
Last. Catherine M. Hill (University of Southampton)H-Index: 39
view all 7 authors...
Abstract Objective Childhood sleep-related rhythmic movement disorder (RMD) (ie, sleep-related repetitive movements involving large muscle groups) can impair sleep quality, cause local injury, and disturb household members. Previous parental reports indicate prevalence rates in children under three years of age between 5.5 and 67%. We studied the prevalence of RMD with objective home videosomnography. Methods Parents of 707 children having their one-year routine health check (357 male), 740 chil...
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#1Anna J. Esbensen (Cincinnati Children's Hospital Medical Center)H-Index: 23
#2Emily K. Hoffman (Cincinnati Children's Hospital Medical Center)H-Index: 7
Last. Rebecca C. Shaffer (Cincinnati Children's Hospital Medical Center)H-Index: 10
view all 4 authors...
There is a need for rigorous measures of sleep in children with Down syndrome as sleep is a substantial problem in this population and there are barriers to obtaining the gold standard polysomnography (PSG). PSG is cost-prohibitive when measuring treatment effects in some clinical trials, and children with Down syndrome may not cooperate with undergoing a PSG. Minimal information is available on the validity of alternative methods of assessing sleep in children with Down syndrome, such as actigr...
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#1Amy R.M. Gwyther (University of Southampton)H-Index: 1
#2Arthur S. Walters (VUMC: Vanderbilt University Medical Center)H-Index: 64
Last. Catherine M. Hill (University of Southampton)H-Index: 39
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Summary Rhythmic movement disorder consists of repetitive stereotypic movements, such as head banging or body rocking, that recur every second or so and may last from a few minutes to hours, usually prior to sleep onset. This review of childhood rhythmic movement disorder highlights the lack of systematic research into core aspects of the condition, relying heavily on small case series or case reports. Interpretation is further limited by almost universal failure to confirm the core diagnostic c...
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#1Maurice M. Ohayon (Stanford University)H-Index: 89
#2Emerson M. Wickwire (UMB: University of Maryland, Baltimore)H-Index: 21
Last. Michael V. Vitiello (UW: University of Washington)H-Index: 71
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Abstract Objectives To provide evidence-based recommendations and guidance to the public regarding indicators of good sleep quality across the life-span. Methods The National Sleep Foundation assembled a panel of experts from the sleep community and representatives appointed by stakeholder organizations (Sleep Quality Consensus Panel). A systematic literature review identified 277 studies meeting inclusion criteria. Abstracts and full-text articles were provided to the panelists for review and d...
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#1Catherine M. Hill (University of Southampton)H-Index: 39
#2Hazel J. Evans (Boston Children's Hospital)H-Index: 8
Last. Paul Gringras (Boston Children's Hospital)H-Index: 26
view all 12 authors...
Abstract Background Children with Down syndrome (DS) are vulnerable to obstructive sleep apnoea (OSA) because of their unique craniofacial anatomy and hypotonia. Understanding the predictors of OSA in DS may enable targeted screening. Methods Children with DS (n = 202) aged from six months to below six years (110 boys) were recruited from three UK children's hospitals. The clinical assessment included height, weight and tonsillar size. The parents either set up cardiorespiratory polygraphy at ho...
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