Sleep in �Children with Down Syndrome �

Anna J Esbensen, PhD

Richard & Jane Thomas Center for Down Syndrome

November 10, 2018

Sleep in Down syndrome

• Obstructive sleep apnea (OSA)
• 31-63% in clinic samples
• 24-59% in community samples

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• Rate of OSA in general population:
• 3-7%

Sleep in Down syndrome

• Sleep problems (31-54%)
• Rating scales
• Later sleep onset
• Sleep anxiety
• Bedtime resistance
• Difficulties with sleep maintenance
• Night waking
• Parasomnias
• Frequent night waking / fractured sleep

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• Polysomnography
• Fractured sleep
• Longer time in bed
• Lower sleep efficiency
• Less time in REM
• More movement

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• Children with DS averaging less sleep than recommended
• PSG = 6.9hrs
• Actigraphy home = 7.9hrs
• Recommended 6-13yo = 9-11hrs, 14-17yo = 8-10hrs
• Sleep efficiency poor
• PSG 85%
• Actigraphy home 82%

Most amount sleep

Least amount sleep

Minimum recommended

8hrs, 9hrs sleep

How do we measure�sleep?

• Parent-reports

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• Behavioral Evaluation of Disorders of Sleep (BEDS)
• Children’s Sleep Habits Questionnaire (CSHQ)
• Sleep Disturbance Scale for Children (SDSC)

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• 7-night sleep diary, behavior diary

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• Pilot with 30 caregivers of children with DS 6-17 years

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How do we measure�sleep?

• PSG gold standard
• Not realistic for cost / access
• Actigraphy

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• Actigraphy vs PSG (clinic) (n=27, age 5-17 years, M=10.7)
• Actigraphy vs CSHQ (community) (n=30, age 6-17 years, M=11.7)

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• Depends on what you measure
• Total sleep time
• Waking after sleep onset
• Sleep efficiency

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How Do We Screen for & Treat Sleep Problems?

• Electronic Medical Records (EMR) query (2009-2013)
• 954 children with DS ages 5-21
• Classified as:
• No sleep problems (n=581, 60.9%)
• Behavioral sleep disorder (n=33, 3.5%)
• OSA (n=258, 27.0%)
• Both (n=82, 8.6%)

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39.1%

PSG recommended by age 4 years

47.7%

81.2%

Impact of sleep �in the general population

In DS:

• Age
• Gender
• Behavioral regulation
• Verbal IQ
• Cognitive flexibility

Ashworth et al., 2013; Breslin et al., 2011; Breslin et al., 2014; Churchill et al., 2014; Maris et al., 2016; Stores, 1993

Impact of sleep in �adults with DS

• 20-year follow-up
• Caregiver report on 75 adults with DS
• Medical and behavioral questions about sleep
• Health conditions, medical care, daytime behavior

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• Different correlates for OSA and behavioral sleep problems

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Impact of sleep in �adults with DS

• OSA
• More respiratory concerns
• More frequent visits to physician

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• Behavioral sleep problems
• Poorer health
• More frequent overnight hospital and ED visits
• More common cardiac conditions
• Less common thyroid condition
• More common mental health conditions (anxiety, depression, dementia)
• Higher rate daytime behavior problems

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Impact of sleep on behavior in �children with DS

• 30 children with DS, 6-17 years (M=11.7, SD=2.7)

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Sleep duration

Sleep movement

Sleep duration

Sleep efficiency

Behavior

Anxiety

Inattention

Hyperactivity

Behavior

Anxiety

Inattention

Hyperactivity

Impact of sleep on executive functioning in children with DS

• Same sample as behavior

Sleep movement

Sleep duration

Sleep efficiency

Inhibit

Shift

Working Memory

Inhibit

Shift

Working Memory

No impact of sleep on performance on neuropsychology battery of assessments

Sleep Intervention in DS

• 5 week behavioral intervention
• CON: Enhanced standard of care general-education program
• BST: Behavioral sleep treatment

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• Aims/Outcomes
• Improve child sleep
• Improve child behavior
• Improve parental sleep & well-being

Outcomes

Treatment effects at 18-week follow-up

Preliminary findings R21 - behavior

• BST works! It reduces behaviors on:
• ABC hyperactivity
• Vanderbilt hyperactivity
• Vanderbilt oppositional behaviors
• But so does CON

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• Thus, is our intervention effective? Or is it a placebo effect of therapist’s time and attention?

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Exciting news

• INCLUDE project
• Conduct targeted, high-risk, high reward basic science studies on chromosome 21
• Assemble a large study population of individuals with DS
• Deep phenotyping and study co-existing conditions
• Include individuals with DS in existing clinical trials

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https://www.nih.gov/include-project

More exciting news!

• R01 HD093754 (Esbensen)
• Purpose
• Evaluating metrics of cognition and executive functioning
• Characterize natural development
• Sample
• 6-17 year olds
• Study
• 5 visits over 1 year
• Standardized testing
• Parent-rating forms

More exciting news!

• R01 HD093654 (Thurman)
• Purpose
• Evaluating metrics of prelinguistic skills, spoken language ability
• Characterize natural development
• Sample
• 2.5 – 7 year olds
• Study
• 3-4 visits over 1 year
• Semi-structured play
• Standardized testing
• Parent-rating forms

Acknowledgements

Funding

• NICHD – R03 HD059848
• NICHD – R21 HD082307, R01 HD093754
• Jack H Rubinstein Foundation
• Emily Ann Hayes Research Fund
• Jérôme Lejeune Foundation

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Colleagues

• Dean Beebe, PhD
• Kelly Byars, PsyD
• Jeff Epstein, PhD
• Emily Hoffman, MEd
• Cynthia Johnson, PhD
• Rebecca Shaffer, PsyD

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Community

• DSAGC
• Families who participated in this research
• Families/Individuals who educate me every day

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Questions?