Post on 25-Jul-2020
transcript
Luci Wiggs
Oxford Brookes University
Department of Psychology
Sleep disturbance in children with Autism
Spectrum Disorder (ASD)
Outline
• Nature of sleep
problems
• Associations
• Management
Sleep abnormalities and ASD
• Physiological sleep abnormalities eg. melatonin profile (Tordjman 2005;2012; Kulman et al 2000; Melke 1998; Nir et al 1995)
Kulman et al, (2000)
Sleep abnormalities and ASD
• Physiological sleep abnormalities eg. melatonin profile (Tordjman 2005;2012; Kulman et al 2000; Melke 1998; Nir et al 1995)
• Sleep disorders
eg. sleep/wake cycle disorders (Inanuma 1984; Glickman 2010)
• ‘Sleeplessness problems’ (Souders et al 2009; Malow et al 2006; Wiggs & Stores 2004; see Richdale 2001)
‘sleeplessness’ –
bedtime difficulties
difficulty getting to sleep
waking in night
early waking
irregular sleep
short duration sleep
Sleeplessness and ASD
• Sleeplessness rate in ASD about 66% (range 49-89%) (Richdale 2001; Wiggs & Stores 2004; Krakowiak et al 2008, Souders et al 2009)
• Compared to typically developing children, more of a decrease in night sleep (later bedtime, night wakes, early waking) from 30 months – 11 years (Humphreys et al 2013)
• High rates in children intellectual disabilities (Patzold et al 1998; Krakowiak et al 2008)
• IQ positively predictive of sleep anxiety (n=1583) (Hollway et al 2013)
Sleep disturbance and ASD: associations • Behaviour: Increased challenging behaviour and externalizing
problems (e.g. Sikora 2012; Mayes & Calhoun, 2009; DeVincent et al, 2007, Allik et al, 2006, Patzold et al,
1998; Hoshino et al 1984)
• Mental health: Increased anxiety and affective problems (Hollway et al
2013; Sikora 2012, Malow et al, 2006;Tani et al, 2004; Wiggs & Stores, 2004; Tani et al, 2003)
• Cognition: Impaired perception/visual response, and cognitive
procedural memory (Limoges et al 2013; Taylor, Schreck & Mulick 2012; Elia et al, 2000)
• Motor function: Low sensory-motor memory, hand-eye co-
ordination and adaptive skills (Limoges et al 2013; Taylor, Schreck & Mulick 2012; Elia et al,
2000)
• Features of ASD: Severity of autistic symptoms, communication
abnormalities, social skills, routines and rituals (May et al 2014; Hollway et al
2013; Taylor et al 2012; Alik et al 2006; Liu et al 2006; Malow et al 2006; Hoffman et al 2005; Schreck et al 2004;
Elia et al 2000)#
• Parental sleep disturbance: (Lopez-Wagner et al 2008; Meltzer 2008; Hodge et al 2013)
Significance of
successful resolution of
sleep disturbance
• Improved sleep associated with improvements in child behaviour, mental
health, parents mental health and family functioning (in typically developing
children and those with developmental disorders)
• ASD – limited studies; range of sleep disorders/interventions
• Some studies suggesting successful intervention associated with increased
ease of management. e.g.
– improved aspects of child, behaviour (e.g. hyperactivity, self injurious behaviour,
rigid/repetitive behaviour)
– mood, internalising problems
– communication, social interaction
– child quality of life
– parenting sense of competence
(Malow et al 2014; 2012 Wright et al, 2010; Reed et al, 2009; Malow et al 2006; Paavonen et al 2003; De Leon et al, 2004)
Sleep disturbance is common
in a range of conditions associated
with intellectual disabilities and autism
Down syndrome
Smith-Magenis syndrome
Prader-Willi syndrome
Angelman syndrome
Williams syndrome
Fragile X syndrome
Cornelia de Lange syndrome
Cri du Chat syndrome
Rett syndrome
Mucopolysaccharidoses
Sleep disturbance -
comorbidity common Comorbidity with:
• Neurodevelopmental disorders
• Medical/neurological problems
• Emotional/behavioural disorders
Direct effects (e.g. discomfort, physical
features of Down syndrome)
Indirect effects (e.g. treatment)
(See Wiggs (2012) for discussion)
Management approaches
• Reassurance/explanation
• Safety measures
• Sleep hygiene
• Psychological
* Behavioural
* Cognitive
• Chronotherapy
• Medication (see Hollway & Aman 2011)
* Hypnotics
* Stimulants
* Melatonin
* Others
• Physical measures
• Surgery
(See Wiggs. L. (2012). Sleep Disturbances and Learning Disability (Mental Retardation) In
C. M. Morin and C. Espie (Eds.), The Oxford Handbook of Sleep and Sleep Disorders.
New York: Oxford University Press)
Sleeplessness Management in ASD
• Sleep hygiene
• Behaviour therapy
• Melatonin
• Other interventions
• Weighted blankets
Sleep Hygiene
“A set of sleep-related behaviours that exposes the
individual to activities and cues that prepare them for and
promote appropriately timed and effective sleep”
(Meltzer & Mindell 2004)
• Environment - familiar, comfortable, dark, quiet
• Scheduling – consistency in timing, daytime activities
• Sleep practices – calming routine
• Physiologic – naps, caffeine, TV/PC use
Sleep Hygiene and ASD
• Poor evidence base for special considerations in children
with developmental disorders (Jan 2008) and ASD (Vriend et al
2011)
• Unusual/inconsistent bedtime routines in children with
ASD - maladaptive for promoting good sleep hygiene?
(Henderson et al 2011)
• TV/computer in room and time spent playing video games
in boys with autism more strongly associated with
reduced sleep than for boys with ADHD and controls
(Englehardt et al 2013)
Elements of sleep are learnt behaviours including…
• How we prepare for bed
• How we settle to sleep
• Where we settle to sleep
• What we do when we wake up
Behaviour therapy likely to play a
role where elements of sleep
behaviour have been learnt
‘incorrectly’ or ‘not learnt’ at all
Behaviour Therapy
• Number of ‘well
established’ techniques
for TD infants (see Mindell
2006; American Academy of Sleep
Medicine 2006)
• Helpful for children with
developmental disorders
inc. ASD Vriend et al (2011)
Richdale & Wiggs (2005)
Interventions for sleeplessness likely to
include
• Appropriately timed bedtime routine (cueing)
• Appropriate bed time (linked with sleep onset)
(conditioning)
• Extinction/stimulus fading/checking (removing positive
reinforcement for undesired behaviour)
• Positive reinforcement (shaping)
Behaviour therapy: general
considerations
• intervention based on functional assessment
• use diary
• pick good time to start
• support
Behaviour therapy: special considerations
• Making the room/house safe/secure
• Changes as gradual as required (for parent and child)
• Communication difficulties with child
– social stories
– visual schedules
– use of all senses
• Use of school/drivers
• Creative use of reinforcement
Reports of behaviour therapy for
sleeplessness in ASD • 4 RCTs
– 36 children 2-10 years (Adkins et al, 2012)
– 39 children, 4-16 years (Wiggs & Stores, in preparation)
– 33 children 2-6 years (Johnson et al, 2013)
– 144 children (33 CBT), 4-10 years (Cortesi et al 2012)
• 2 uncontrolled trials, 20 children 3-10 years (Reed et al 2009); 80 children, 2-10 years (Malow et al, 2014)
• 1 multiple baseline study, 6 children 3-7 years (Weiskop et al 2005)
• 9 case reports, 18 children, 3-12 years
• 36 children 2-10 years
• Information pamphlet : no effect
on sleep latency
• Parents needed to know ‘how’
not just ‘what’ to do
(Adkins et al, 2012)
• 80 children, 2-10 years
• Group (4x1 hr) vs individual (1hr)
parent education (+2 follow up calls):
both statistically reduced sleep
latency
(Malow et al, 2014)
http://www.autismspeaks.org/science/resources-programs/autism-treatment-
network/tools-you-can-use/sleep-tool-kit
Randomised control trial (RCT) of behaviour
therapy (BT) in children with ASD
PE
BT
Children with ASD and sleep disturbance
(2-6 years).
RCT of manualised BT (n=15) vs non-sleep
parent education (PE) (n=18) over 8 weeks
The BT group improved significantly more
than the comparison group based on
composite sleep index (CSI) parent report.
No change in objective sleep (actigraphy)
(Johnson, Kylan, Foldes, Kronk, & Wiggs 2013)
Same pattern of findings in older children with ASD (5-16 years)
(Wiggs & Stores, in preparation)
PE
Some parents’ comments…
“our child sleeps the best now that he’s ever done in his life”
“massive improvement – thank you!”
“has made our lives a lot easier as it’s so much calmer in the house”
“we have our evenings back! Thank you”
“for the first time I am able to say ‘good night’ to my child and it’s a pleasant experience”
(Wiggs & Stores, in preparation)
Cortesi et al (2012) 134 children (4-10y)-ASD (no ID) and sleeplessness
3mg controlled release
Multifactorial BT – Combined (n=35)
– Melatonin (n=34)
– BT (n=33)
– Placebo (n=32)
Mean % change from baseline Combined Melatonin BT Placebo
Sleep onset latency 60.75 44.33 22.54 -0.02
Total sleep time 22.01 17.31 9.31 0.07
Sleep latency
Melatonin
• Used with varying dose (0.5 - 24mg) for unspecified ‘sleep difficulties’
(Gringras 2005)
• Suggested as being useful to reduce sleep latency and possibly increase continuous sleep duration in children with developmental disorders inc. ASD
(Tordjman et al (2013); Rossignol & Frye (2011); Braam et al (2009))
• Issues
– Sample (small, age, heterogenous)
– Melatonin administration
• Loss of response in 15 patients (7/15 ASD)… Slow metabolism of melatonin? (Braam et al, 2013)
Melatonin in ASD
Double blind placebo controlled RCTs
• 144 children (34 melatonin) (4-10 years) – 3mg (controlled release); actigraphy and questionnaire (Cortesi et al 2012)
• 146 children (70 melatonin; 30 ASD) (3-16 years) – 0.5-12mg (immediate release); actigraphy, diary and questionnaire (Gringras et al 2012)
• 17 children (3-6 years) – 2-10mg. Diary report of improved sleep latency and total sleep time. No effect on night wakes (Wright et al 2010)
• 12 children with ASD (2-15). 3mg. Actigraphy and diary. Improvement in sleep, especially sleep onset time (Wirojanan et al, 2009)
• 7 children (5-15 yrs) - 5mg melatonin. Parent report of improved sleep latency, reduced night wakes and increased total sleep
(Garstang & Wallis, 2006)
Open label trials (Malow et al, 2011; Giannotti, 2006; Paavolen, 2003) and Retrospective trial (Andersen, 2008)
Gringras et al (2012) 263 children registered 146 randomised (8 declined/93 no longer met criteria/16 other reasons)
Sleep diary (adjusted difference and 95% CI)
Actigraphy (adjusted difference and 95% CI)
Sleep latency -37.5 mins ** (-55.3 to -19.7)
-45.3 mins ** (-68.8 to -21.9)
Sleep duration 22.4 mins * (0.5 to 44.3)
13.3 mins
(-15.4 to 42.2)
Melatonin resulted in earlier waking times
Most effective for children with longest sleep onset latency
Treatment effects not modified by presenting sleep disturbance (p=0.56) or
presence of autism (p=0.85)
* p<0.05; ** p<0.0001
Montgomery, Wiggs & Stores (2004)
http://www.researchautism.net/publicfiles/good_sleep_ha
bits.pdf
Other interventions
• ‘Insomnia’ medications generally poor evidence base in children (Hollway & Aman 2011; Gringras 2008)
– Trimeprazine tartrate/diphenhydramine/chloral hydrate (only short
term)
– Atypical antipsychotics
– Trazodone/mirtazapine
– Ramelteon
– Clonidine
– Zolpidem/Zopiclone/Zalepon
– Benzodiazepines
• Currently a lack of quality evidence for other approaches such as exclusion diets, white noise, aromatherapy, exercise, relaxation, dietary supplements, massage etc.
(McLay & France 2014)
Gringras, Green, Wright, Rush, Sparrowhawk, Pratt, Allgar, Hooke, Moore, Zaiwalla, Wiggs
(2014); Funded by Research Autism
Snuggledown (The use of sensory weighted blankets
in children with autistic spectrum disorders: a
randomised crossover study)
– Southpaw weighted blanket (ROMPA) (NHS preferred supplier and OT approved)
– Standard size (147 by 76 cm - 2.25kg ) for height <135cm; Large size (152 by 152 cm - 4.5kg) for taller. The blanket that approximates best to 10% of child’s body weight.
– Identical weighted and non-weighted placebo blanket provided
Results • 67 children completed the trial (73 recruited)
• No change in total sleep time, sleep latency, night wakings
or sleep efficiency (both actigraphy and diary measures).
• Other outcomes:
– Parents and children preferred the weighted blanket
– Parents said children calmer and their sleep better with weighted
blankets
– No blanket-related change in sensory response or child
behaviours
– Age, weight, severity of sleep problems, autism severity or profile
of sensory response not related to response to treatment
Conclusions
• Sleep disturbance in children with ASD and ID is a big clinical and family problem
• Coexisting ID does not appear to be a particular risk factor but might have management implications
• Autism Treatment Network Practice Pathway (Malow et al, 2012); Autism NICE clinical guidelines (NICE, 2013)
– Preliminary insomnia screening enquiries should be made routinely
– Screening/treatment for contributory factors
– With appropriate assessment and diagnosis of underlying sleep disorder there are various management options from which to select
– Behaviour therapy as first-line approach
– Pharmacological intervention in addition where necessary
• Behaviour therapy has various levels; if ‘booklets’ don’t work-don’t give up!
• Need for quality intervention studies and to understand factors
contributing/related to outcome in order to guide management decisions for individual children
Thank you