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