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doi:10.1111/jpc.12933
ORIGINAL ARTICLE
Sleep concerns in children and young people with cerebral palsy in their home setting Susan M McCabe,1,2 A Marie Blackmore,2 Chris R Abbiss,1 Katherine Langdon3 and Catherine Elliott4,5 1
School of Exercise and Health Sciences, Edith Cowan University, Joondalup, 2Ability Centre, 4Faculty of Health Sciences, Curtin University of Technology, 5Child and Adolescent Health Services, Perth, 3Department of Paediatric Rehabilitation, Princess Margaret Hospital, Subiaco, Western Australia, Australia
Aims: The aims were to identify in-home concerns about sleep in children and young people with cerebral palsy (CP) across age and Gross Motor Function Classification Scale (GMFCS) levels. Methods: This was a retrospective review of clinical notes of 154 children and young people with CP, aged 1–18 years (M = 7.8; standard deviation = 5.4) who received a home-based sleep service. Reported concerns were synthesised, for analysis according to age groups (1–5, 6–13, 14–18) and GMFCS levels. Results: Sixteen factors of concern were derived from the home-based assessment reports. Most children and young people had multiple factors of concern. These varied across age groups and GMFCS levels. Body position was of concern across all age groups, for over 90% at GMFCS levels IV and V, and for 10% at GMFCS level I. Settling routines were of concern for more than 90% at GMFCS levels I and II, but for less than 50% at GMFCS levels IV and V. Settling routines were of concern to over 65% of those under 6 years but less than 25% of those over 14 years. Conversely, pain and pressure care concerned less than 10% of children under 6, and more than 35% of those over 14 years. Conclusions: Concerns about sleep vary across ages and GMFCS levels of children and young people with CP. Concerns relate to impairment of body structure and function, activity, environment, and personal supports. Multi-disciplinary, home-based assessment and interventions are recommended to address these concerns. Key words:
adolescent; cerebral palsy; child; preschool; sleep.
What is already known on this topic
What this paper adds
1 Children with cerebral palsy (CP) have a higher incidence of sleep difficulties than their typically developing peers. 2 Children’s sleep problems have significant impact on their health and well-being, and that of their caregivers. 3 There are diverse and complex reasons for their sleep problems.
1 Sleep problems are evident in children and young people with CP of all ages and across all levels of classification of gross motor function. 2 Children and young people with mild physical impairment, and younger children, have concerns related to their environment, activities and personal factors. 3 Children and young people with severe physical impairment have concerns related to their lying position, movement control, breathing, reflux, pain, pressure care, body temperature and safety.
Cerebral palsy (CP) is the commonest cause of physical disability in childhood,1 affecting 2–2.5 in 1000 live births.2 Children with CP are a highly heterogeneous group. They have mild to severe motor impairments affecting functional movement3 and may also have disturbances of sensation, perception, cognition, communication and behaviour, epilepsy, and secondary muscuCorrespondence: Ms Susan M McCabe, Ability Centre, 106 Bradford Street, Coolbinia, WA 6050, Australia. Fax: +61 8 9443 0356; email: [email protected] Conflict of interest: There is no conflict of interest to report. Accepted for publication 21 April 2015.
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loskeletal problems.4,5 Children with ‘mild’ CP may have learning disabilities, attention deficit/hyperactivity disorder and pervasive developmental disorder.6 Irritability, anxiety, overactivity and poor attention have been reported in over 50% of children with hemiplegic CP.7 Children with CP commonly experience sleep disorders.3,6 A survey of 216 parents of children with CP found that 48% reported sleep problems, 23% of them serious.8 A study using the Sleep Disturbance Scale for Children9 found that 23.5% of children with CP had a ‘pathological’ sleep score compared with 5% of the normative population.10 Difficulties with all aspects of sleep have been reported in children with CP, including difficulties with sleep initiation and maintenance, sleep–wake transitions,
Journal of Paediatrics and Child Health 51 (2015) 1188–1194 © 2015 The Authors Journal of Paediatrics and Child Health © 2015 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
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breathing during sleep and daytime sleepiness.8,11–13 Sleep disturbances have adverse effects on child development, health, growth, behaviour, learning, memory, mood and performance.14–18 In children with CP, the effects of sleep disturbance compound with other impairments and reduce their wellbeing, daily functioning and quality of life.12,13,19–21 Furthermore, children’s sleep disturbance affects their parents’ sleep, and is associated with increased parental daytime sleepiness, stress, depression, fatigue, and impairment of health and social functioning. 22–27 Diverse factors affect the sleep of children with CP. Indeed, studies utilising parent questionnaires, scales and interviews have revealed that children with CP have sleep difficulties related to breathing,8,10,12,13,28–33 pain, cramps and muscle spasm,8,10,13,34,35 postural discomfort and need for assistance to change position,8,10,13,35 epilepsy and its treatment,8,13,28,32,34 circadian difficulties,8,12,28,31 use of splints, and positioning equipment for musculoskeletal support,10,13,34 toileting needs,8,13 bedtime resistance or anxiety,8,10,13,28,32 gastro-oesophageal reflux,6,10,13,19,29,33,36 and other swallowing and digestion difficulties,8,13 skin conditions such as itchiness or pressure sores,13 disturbances of vision or hearing,13 and thermoregulation difficulties such as cold extremities13 and hyperhydrosis.11 Sleep difficulties affect all areas of children’s lives, and it is important to consider sleep of children with CP and their families using a holistic perspective of the child’s health condition, environment and personal factors.37 However, there has been little comprehensive exploration of children’s sleep in their home environment. The aims of the present study were to identify the concerns around sleep of children and young people with CP in their home setting, and to describe the incidence of these factors across ages and levels of classification of gross motor function.
Methods This was a review of records of a clinical sleep service for children and young people with disabilities, which undertakes assessments in their home. This multi-disciplinary sleep assessment and intervention service are based at Ability Centre in Perth, Western Australia, and available to all individuals with disabilities in Western Australia. Referrals come from clinical teams from the health and disability sector. The service is provided in the home setting by an occupational therapist or physiotherapist, alongside the referring team. There is no cost to individuals eligible for state disability services. The number of sessions varies depending upon families’ needs.
Design This was an observational retrospective review of referral data and clinical case notes of children and young people seen by the clinical sleep service at TCCP between January 2007 and December 2012. Ethics approval was received from the Human Research Ethics Committee at Edith Cowan University, Western Australia.
Participants Participants were included in the study if they (i) were referred for the first time during the study period (2007 – 2012), (ii)
Sleep in children with cerebral palsy
were aged 1–18 years, (iii) had CP and (iv) proceeded with clinical services from the sleep team.
Procedures The data for this study were extracted from referral information, clinical notes from the sleep team assessment and medical records. The referral data included the participant’s name, age, sex, diagnostic information and sleep problems reported by the referrer. The sleep team’s home-based assessment included a safety checklist, postural assessment, data collection during sleep, a ‘sleep profile’ interview proforma, and parent-recorded daytime and nighttime data. The safety checklist was administered in all cases, but use of other components depended upon the concerns of each family. The safety checklist was developed by clinicians in this sleep service. It facilitates discussion about pain, pressure care, breathing, gastro-intestinal function, management of hygiene, continence, thermoregulation, safety (e.g. risk of falling from bed, being caught in side rails), mobility into and within bed, general comfort, sleep disturbance, postural care and medical issues (e.g. epilepsy, surgery, medications) and carers’ needs. Postural assessment and observation of the child’s or young person’s movements and positions in bed were undertaken if pain, breathing, pressure care, thermoregulation, mobility, safety, general comfort or postural care were reported as concerns. Data collection during sleep included photographs, actigraphy, overnight video and pulse oximetry. The sleep profile interview was used if sleep onset and maintenance, early wakening and daytime sleepiness were reported as concerns. This interview profile guides the discussion about the child’s or young person’s activities and routines, household and bedroom environment, sensory regulation, behaviour and communication, and general health. Daytime and night-time data, including sleep diaries, activity logs, actigraphy, evening and overnight video, and room thermometers, were used to enhance assessment of home-based activity and environment. The third source of data was a review of participants’ medical records. These confirmed the classification of CP and Gross Motor Function Classification Scale (GMFCS) level, and identified diagnoses of co-morbidities of epilepsy, intellectual disability, vision impairment, gastro-oesophageal reflux and scoliosis. The GMFCS classified gross motor function from Level I (most able) to Level V (least able).38,39
Data extraction and management A clinician in the sleep team (SM) reviewed all data sources, and noted the factors reported for each participant. When there was doubt about factors, these were cross-checked with another clinician in the sleep team, and agreement was reached through discussion. The factors were allocated to categories of best fit. Data were analysed in EXCEL 2010 (Microsoft, Redmond, WA, USA).
Results There were 476 referrals to the clinic during the period studied. Of these, 291 did not meet the inclusion criteria. The 31 who did not proceed with services withdrew due to participant’s illness or domestic concerns. This left 154 participants in the study (Fig. 1).
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Table 2 Percentages of participants at each GMFCS level with diagnosed comorbid conditions Diagnosed comorbid conditions
GMFCS
Epilepsy Intellectual disability Vision impairment Gastro oesophageal reflux disease Scoliosis
I 25 5 0 5 0
II 38 38 8 8 31
III 35 41 12 6 6
IV 50 39 29 18 29
V 72 57 29 29 46
Table 3 Numbers of categories of concerns expressed by participants (n = 154)
Fig. 1
Percentage of participants
1 2 3 4 5 6 7 8 9
7 11 19 17 19 12 8 2 3
Referrals to Sleep Solutions service included in study.
Table 1 Demographic characteristics of participants (n = 154) Participant characteristics Sex Male Female Age 1–5 years 6–12 years 13–18 years GMFCS I II III IV V
N
%
83 71
54 46
72 39 43
47 25 28
20 13 17 28 76
13 8 11 18 49
Participants’ ages ranged from 1 to 18 years (mean = 7.8; standard deviation = 5.4). Their age and GMFCS levels are shown in Table 1. Most participants had co-morbid conditions (Table 2). Participants at all GMFCS levels were diagnosed with epilepsy, intellectual disability and gastro-oesophageal reflux disease. Participants at all except GMFCS level I were diagnosed 1190
Number of categories
with vision impairment and scoliosis. The highest proportions of co-morbid conditions were at GMFCS levels IV and V. Reported concerns related to the participants’ sleep were assigned to 16 categories. These categories gradually evolved over time and were based on published literature, comprehensive clinical experience of clinicians in the sleep team, and a flexible in-home assessment that allowed participants to raise a very wide range of concerns regarding sleep. A description of each category is given in the Appendix. Nearly all participants (93%) had multiple concerns, with 25% having concerns in six or more categories (Table 3). The categories of concern varied between age groups (Table 4). In children aged 1–5 years, the main categories were settling routines, posture, sleep environment, communication and behaviour, temperature and perspiration, and reflux and digestion. For children aged 6–13 years, the main categories were posture, settling routines, movement control, reflux and digestion, temperature and perspiration, and safety. In individuals aged 13–18 years, the main categories were posture, pain, pressure, breathing, and temperature and perspiration. Categories of concern also varied across GMFCS groups (Table 5). For individuals with mild motor impairment (GMFCS levels I and II), sleep routines, settling routines, daytime activities, behaviour and sensory regulation predominated. Children and young people with severe motor impairment (GMFCS IV and V) had concerns mostly related to posture, movement control, breathing, reflux and digestion, and temperature and perspiration.
Journal of Paediatrics and Child Health 51 (2015) 1188–1194 © 2015 The Authors Journal of Paediatrics and Child Health © 2015 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
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Table 4 Percentages of participants at each age group in each category of concern Age (years) Categories of concerns
1–5
Body position Movement Breathing Reflux/digestion Pain Pressure care Temperature/perspiration Seizures/medications Safety Continence Health issues Sleep environment Settling routines Daytime activity and routines General behaviour and communication Sensory regulation responses
6–12
67 26 25 32 8 3 33 22 29 6 22 42 68 28 35 34
13–18
69 36 68 31 18 13 31 15 31 5 10 28 51 13 28 15
86 26 33 9 47 37 33 5 23 5 12 14 23 9 14 9
Black shading indicates very high frequency (75–100%), dark grey shading high frequency (50–74%), light shading moderate frequency (25– 50%) and white shading low frequency (0–25%).
Table 5 Percentages of participants at each GMFCS level in each category of concern GMFCS Categories of factors of concern
I
II
III
IV
V
Body position Movement Breathing Reflux/digestion Pain Pressure care Temperature/perspiration Seizures/medications Safety Continence Health issues Sleep environment Settling routines Daytime activity and routines General behaviour and communication Sensory regulation responses
10 5 5 10 0 0 20 5 10 10 25 60 100 60 50 40
31 0 8 8 23 0 31 15 15 0 8 38 92 38 69 23
71 6 0 6 6 6 24 12 18 12 18 29 53 12 29 41
86 46 21 14 18 11 29 7 21 4 14 21 43 7 11 14
92 38 46 41 25 25 39 22 39 4 16 25 34 11 20 7
Black shading indicates very high frequency (75–100%), dark grey shading high frequency (50–74%), light shading moderate frequency (25– 50%) and white shading low frequency (0–25%).
Discussion The purpose of this study was to describe the factors concerning sleep of children and young people with CP, and their families,
as identified by individualised assessment in their home setting. The main findings were that: (i) most children and young people had multiple factors of concern, (ii) factors of concern differed with age, and (iii) there were concerns regarding sleep for children and young people at every GMFCS level. In this study, most participants (over 80%) had three or more factors of concern relating to their sleep (Table 3). Given the complex presentations of CP, this is not surprising. The participants in this study do not represent a random sample of the CP population but were referred to the sleep service because of reported difficulties. Therefore, sleep difficulties would be expected to be higher in this sample than in the CP population. Factors were included only if they were reported as concerns at the time of assessment. Factors that did not interfere with sleep (e.g. well-managed incontinence) were not recorded. Therefore, this is not a study of the overall prevalence of these factors in children and young people with CP, nor does it address the unique psychosocial or emotional aspects that determine families’ capacity to deal with the associated challenges of everyday life. Nevertheless, this study adds a home-based perspective to the previous studies which report the wide diversity of concerns in relation to sleep for this population.8,10–13,19,20,28–30,32–34,40–42 It shows that, in clinical practice, identification of only one or two sleep concerns may greatly underestimate the extent of the problems. Effective sleep assessment must be broad enough to screen for all kinds of sleep concerns, and focused enough to target the specific areas of concern for each individual. This study found that factors of concern for sleep in children and young people with CP vary with age (Table 4). Young children had frequent concerns related to sleep environment, settling routines, daytime activities and routines, behaviour and communication, and sensory regulation responses. The reasons for this were not investigated in the study but reflect expected developmental factors. Elsayed et al.42 reported that in typically developing children, more preschool children than older children had difficulty getting to sleep. Likewise, Annaz et al.43 reported that psycho-social factors affecting sleep tend to improve with age. In the present study, some medical factors (management of reflux and digestion, effects of seizures and medications, and general health issues such as ear infections) were identified as concerns for more of the younger children. Parents and medical specialists may develop a better understanding of the children’s health-care needs as they get older, enabling them to tailor care and reduce the burden of concern. Management of posture, uncontrolled movements, and temperature and perspiration were of concern for all age groups, possibly reflecting the unchanging neurophysiology of CP. It is not surprising to see that breathing issues were of concern across all age groups. Sleep breathing problems are well described for this population, with previous studies reporting difficulties related to upper airway obstruction, oropharyngeal motor problems, gastro-oesophageal reflux, poor cough and airway clearance and respiratory muscle weakness.29,30,33,41 Management of pain and of pressure injuries was noted as concerns for over a third of the older age group. This reflects the serious impact of musculo-skeletal changes as children with CP grow.44,45 The present study also shows that there are concerns regarding sleep for children and young people at every GMFCS level.
Journal of Paediatrics and Child Health 51 (2015) 1188–1194 © 2015 The Authors Journal of Paediatrics and Child Health © 2015 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
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Individuals at all GMFCS levels were referred for assistance with sleep difficulties. Those with less severe gross motor impairment (GMFCS I and II) had factors of concern mostly related to their settling routines, sleep environment, daytime activities and routines, behaviour and communication, and sensory regulation responses. It is important to recognise that children and young people with less severe gross motor impairment have sleep difficulties and should not be overlooked in screening, assessment and interventions. There is a risk of under-identification in this group because parents may not report their concerns,46 and health professionals may be less inclined to ask about the sleep of individuals who appear to have a ‘mild’ disability. Indeed, individuals with minor motor disabilities were excluded from the study by Hemmingsson et al.8 because those with moderate to severe motor disabilities were expected to have the more serious sleep problems. As noted by Jan et al.,19 it is especially important to recognise that children with ‘mild’ CP have sleep difficulties that stem from, and contribute to, their known difficulties with behaviour and attention. Clinicians need to ensure strong working links with psycho-social teams to help to manage these behavioural aspects of sleep and daytime function. Children and young people with more severe gross motor impairment (GMFCS IV and V) had factors of concern related to impairment of their body structure and function, with most frequent concerns related to their posture, movement control, breathing, digestion, pain, pressure care, temperature and safety. For these individuals, it is especially important that allied health and medical teams are well linked for effective co-ordination of the assessment and management of these factors in both the clinical and home setting. There is possible referral bias in this retrospective study, with 50% of participants at GMFCS level V (Table 1). Nearly all children and young people at GMFCS V (92%) were referred to the service on the basis of concerns regarding their posture and positioning. This reflects prevailing concerns of the referring agents (mainly physiotherapists and occupational therapists) about the need for postural care, which is most apparent in this group (Table 5). This limits the generalisability of the results. Nonetheless, all levels of GMFCS are represented in this study, and concerns were expressed at all levels. The present study indicates that factors of concern for sleep of children and young people with CP are diverse and complex, with effects at all ages, and all levels of gross motor function. They cross all bio-psychosocial domains, with physiological, environmental and personal factors interacting and affecting sleep. Medical, allied health and psycho-social clinicians need to ask about, screen for and provide targeted assessment and well-co-ordinated intervention for sleep for all children and young people with CP. Although medical concerns may be addressed in clinical setting, a home-based approach is also needed to ensure that unique and diverse needs are well met. This study does not report on effectiveness of the interventions that were provided for this very heterogeneous group. Intervention is multi-faceted, individualised and provided alongside educational, recreational, therapy, social and medical services, to meet unique and varied family life priorities. Clinicians are encouraged to seek opportunities for targeted research to measure and report on the effectiveness of home-based interventions. 1192
Acknowledgements We gratefully acknowledge the co-operation of the participants and their families in this study, and the assistance of Linda Mercer and Penny Walker from the clinical sleep service at Ability Centre. This study was undertaken with financial support of the Developmental Occupational Therapy Interest Group of Western Australia.
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Appendix I Categories of Factors of Concern Reported by Parents of Children with Cerebral Palsy Who Were Referred to the Sleep Service Category of factor of concern
Description
1
Body position
2
Movement
3
Breathing
4
Reflux/digestion
5
Pain
6
Pressure care
7
Temperature/perspiration
8
Seizures/medications
9
Safety
Concern regarding the child’s posture when lying in bed: Therapists report increasing severity of scoliosis or hip dislocation, or positioning support required following orthopaedic surgery. Concern regarding involuntary, uncontrolled movements: Parents report that the child slides down the bed, rolls against side rails or hits arms or legs against wall. Concern regarding the child’s breathing during sleep: Parents report that the child has poorly controlled oral secretions, poor swallow, identified apnoeas, history of chest infection. Concern regarding the child’s ability to manage liquid and oral intake: Parents report concerns about reflux and vomiting during night; need to manage night-time feeding regimes. Concern related to the management of pain: Parents report that the child has musculo-skeletal and/or gastro-intestinal pain when lying in bed. Concern regarding risk of pressure injury: Child has signs of skin breakdown due to pressure or shearing forces from lying and moving in bed. Concern regarding management of the child’s temperature or excessive perspiration: parents report that the child has cold extremities, or seems hot during sleep, with evidence of discomfort, agitation, flushed, sweaty. Concern regarding the effects of seizures, or epilepsy medications: Parents report that the child has excessive daytime sleepiness following seizures, or following administration of anti-epileptic medication. Concern regarding the child’s safety in bed: Parents report that the child inadvertently moves into potentially dangerous positions in bed, falls out of bed or actively climbs out of bed. Concern regarding the management of continence: Parents report that they need to tend to the child during the night, due to the need to use toilet or inadequacy of continence aids. Concern regarding general health issues: Parents report that the child has ear infections, urinary tract infections, asthma, hay fever, allergies, skin conditions which they believe are affecting their comfort and sleep. Concern regarding the environment for settling and for sleep: Parents report that household factors (space available, other people in the household, evening and night time activities in the house, cooling and heating) affect their capacity to provide a quiet, dark and comfortable sleep environment for the child. Concern regarding the child’s settling routines: Parents report that the child does not engage in calming activities before bedtime, and requires parental attention and devices as they fall asleep or settle back to sleep during the night (e.g. being held, singing, massage, drinking from a bottle, falling asleep in parents’ bed). Concern regarding the effects of child’s participation in daily activities and routines: Therapists and parents note that the child spends time sleeping at school, does not participate in daytime physical activity and does not have regular time spent outdoors, in sunlight. Concern regarding the child’s general behaviour and communication: Therapists and parents report that the child has difficulty expressing their needs, understanding instructions and routines, or has behaviour management difficulties which affect daytime as well as sleep time. Concern regarding the child’s management of alertness and arousal: Therapists and parents report that the child has difficulty regulating their activity levels; they are ‘revved up’ at bedtime and were identified through sensory assessment as having sensory modulation difficulties.
10
Continence
11
Health issues
12
Sleep environment
13
Settling routines
14
Daytime activity and routines
15
General behaviour and communication
16
Sensory regulation responses
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Journal of Paediatrics and Child Health 51 (2015) 1188–1194 © 2015 The Authors Journal of Paediatrics and Child Health © 2015 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
doi:10.1111/jpc.12933
ORIGINAL ARTICLE
Sleep concerns in children and young people with cerebral palsy in their home setting Susan M McCabe,1,2 A Marie Blackmore,2 Chris R Abbiss,1 Katherine Langdon3 and Catherine Elliott4,5 1
School of Exercise and Health Sciences, Edith Cowan University, Joondalup, 2Ability Centre, 4Faculty of Health Sciences, Curtin University of Technology, 5Child and Adolescent Health Services, Perth, 3Department of Paediatric Rehabilitation, Princess Margaret Hospital, Subiaco, Western Australia, Australia
Aims: The aims were to identify in-home concerns about sleep in children and young people with cerebral palsy (CP) across age and Gross Motor Function Classification Scale (GMFCS) levels. Methods: This was a retrospective review of clinical notes of 154 children and young people with CP, aged 1–18 years (M = 7.8; standard deviation = 5.4) who received a home-based sleep service. Reported concerns were synthesised, for analysis according to age groups (1–5, 6–13, 14–18) and GMFCS levels. Results: Sixteen factors of concern were derived from the home-based assessment reports. Most children and young people had multiple factors of concern. These varied across age groups and GMFCS levels. Body position was of concern across all age groups, for over 90% at GMFCS levels IV and V, and for 10% at GMFCS level I. Settling routines were of concern for more than 90% at GMFCS levels I and II, but for less than 50% at GMFCS levels IV and V. Settling routines were of concern to over 65% of those under 6 years but less than 25% of those over 14 years. Conversely, pain and pressure care concerned less than 10% of children under 6, and more than 35% of those over 14 years. Conclusions: Concerns about sleep vary across ages and GMFCS levels of children and young people with CP. Concerns relate to impairment of body structure and function, activity, environment, and personal supports. Multi-disciplinary, home-based assessment and interventions are recommended to address these concerns. Key words:
adolescent; cerebral palsy; child; preschool; sleep.
What is already known on this topic
What this paper adds
1 Children with cerebral palsy (CP) have a higher incidence of sleep difficulties than their typically developing peers. 2 Children’s sleep problems have significant impact on their health and well-being, and that of their caregivers. 3 There are diverse and complex reasons for their sleep problems.
1 Sleep problems are evident in children and young people with CP of all ages and across all levels of classification of gross motor function. 2 Children and young people with mild physical impairment, and younger children, have concerns related to their environment, activities and personal factors. 3 Children and young people with severe physical impairment have concerns related to their lying position, movement control, breathing, reflux, pain, pressure care, body temperature and safety.
Cerebral palsy (CP) is the commonest cause of physical disability in childhood,1 affecting 2–2.5 in 1000 live births.2 Children with CP are a highly heterogeneous group. They have mild to severe motor impairments affecting functional movement3 and may also have disturbances of sensation, perception, cognition, communication and behaviour, epilepsy, and secondary muscuCorrespondence: Ms Susan M McCabe, Ability Centre, 106 Bradford Street, Coolbinia, WA 6050, Australia. Fax: +61 8 9443 0356; email: [email protected] Conflict of interest: There is no conflict of interest to report. Accepted for publication 21 April 2015.
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loskeletal problems.4,5 Children with ‘mild’ CP may have learning disabilities, attention deficit/hyperactivity disorder and pervasive developmental disorder.6 Irritability, anxiety, overactivity and poor attention have been reported in over 50% of children with hemiplegic CP.7 Children with CP commonly experience sleep disorders.3,6 A survey of 216 parents of children with CP found that 48% reported sleep problems, 23% of them serious.8 A study using the Sleep Disturbance Scale for Children9 found that 23.5% of children with CP had a ‘pathological’ sleep score compared with 5% of the normative population.10 Difficulties with all aspects of sleep have been reported in children with CP, including difficulties with sleep initiation and maintenance, sleep–wake transitions,
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breathing during sleep and daytime sleepiness.8,11–13 Sleep disturbances have adverse effects on child development, health, growth, behaviour, learning, memory, mood and performance.14–18 In children with CP, the effects of sleep disturbance compound with other impairments and reduce their wellbeing, daily functioning and quality of life.12,13,19–21 Furthermore, children’s sleep disturbance affects their parents’ sleep, and is associated with increased parental daytime sleepiness, stress, depression, fatigue, and impairment of health and social functioning. 22–27 Diverse factors affect the sleep of children with CP. Indeed, studies utilising parent questionnaires, scales and interviews have revealed that children with CP have sleep difficulties related to breathing,8,10,12,13,28–33 pain, cramps and muscle spasm,8,10,13,34,35 postural discomfort and need for assistance to change position,8,10,13,35 epilepsy and its treatment,8,13,28,32,34 circadian difficulties,8,12,28,31 use of splints, and positioning equipment for musculoskeletal support,10,13,34 toileting needs,8,13 bedtime resistance or anxiety,8,10,13,28,32 gastro-oesophageal reflux,6,10,13,19,29,33,36 and other swallowing and digestion difficulties,8,13 skin conditions such as itchiness or pressure sores,13 disturbances of vision or hearing,13 and thermoregulation difficulties such as cold extremities13 and hyperhydrosis.11 Sleep difficulties affect all areas of children’s lives, and it is important to consider sleep of children with CP and their families using a holistic perspective of the child’s health condition, environment and personal factors.37 However, there has been little comprehensive exploration of children’s sleep in their home environment. The aims of the present study were to identify the concerns around sleep of children and young people with CP in their home setting, and to describe the incidence of these factors across ages and levels of classification of gross motor function.
Methods This was a review of records of a clinical sleep service for children and young people with disabilities, which undertakes assessments in their home. This multi-disciplinary sleep assessment and intervention service are based at Ability Centre in Perth, Western Australia, and available to all individuals with disabilities in Western Australia. Referrals come from clinical teams from the health and disability sector. The service is provided in the home setting by an occupational therapist or physiotherapist, alongside the referring team. There is no cost to individuals eligible for state disability services. The number of sessions varies depending upon families’ needs.
Design This was an observational retrospective review of referral data and clinical case notes of children and young people seen by the clinical sleep service at TCCP between January 2007 and December 2012. Ethics approval was received from the Human Research Ethics Committee at Edith Cowan University, Western Australia.
Participants Participants were included in the study if they (i) were referred for the first time during the study period (2007 – 2012), (ii)
Sleep in children with cerebral palsy
were aged 1–18 years, (iii) had CP and (iv) proceeded with clinical services from the sleep team.
Procedures The data for this study were extracted from referral information, clinical notes from the sleep team assessment and medical records. The referral data included the participant’s name, age, sex, diagnostic information and sleep problems reported by the referrer. The sleep team’s home-based assessment included a safety checklist, postural assessment, data collection during sleep, a ‘sleep profile’ interview proforma, and parent-recorded daytime and nighttime data. The safety checklist was administered in all cases, but use of other components depended upon the concerns of each family. The safety checklist was developed by clinicians in this sleep service. It facilitates discussion about pain, pressure care, breathing, gastro-intestinal function, management of hygiene, continence, thermoregulation, safety (e.g. risk of falling from bed, being caught in side rails), mobility into and within bed, general comfort, sleep disturbance, postural care and medical issues (e.g. epilepsy, surgery, medications) and carers’ needs. Postural assessment and observation of the child’s or young person’s movements and positions in bed were undertaken if pain, breathing, pressure care, thermoregulation, mobility, safety, general comfort or postural care were reported as concerns. Data collection during sleep included photographs, actigraphy, overnight video and pulse oximetry. The sleep profile interview was used if sleep onset and maintenance, early wakening and daytime sleepiness were reported as concerns. This interview profile guides the discussion about the child’s or young person’s activities and routines, household and bedroom environment, sensory regulation, behaviour and communication, and general health. Daytime and night-time data, including sleep diaries, activity logs, actigraphy, evening and overnight video, and room thermometers, were used to enhance assessment of home-based activity and environment. The third source of data was a review of participants’ medical records. These confirmed the classification of CP and Gross Motor Function Classification Scale (GMFCS) level, and identified diagnoses of co-morbidities of epilepsy, intellectual disability, vision impairment, gastro-oesophageal reflux and scoliosis. The GMFCS classified gross motor function from Level I (most able) to Level V (least able).38,39
Data extraction and management A clinician in the sleep team (SM) reviewed all data sources, and noted the factors reported for each participant. When there was doubt about factors, these were cross-checked with another clinician in the sleep team, and agreement was reached through discussion. The factors were allocated to categories of best fit. Data were analysed in EXCEL 2010 (Microsoft, Redmond, WA, USA).
Results There were 476 referrals to the clinic during the period studied. Of these, 291 did not meet the inclusion criteria. The 31 who did not proceed with services withdrew due to participant’s illness or domestic concerns. This left 154 participants in the study (Fig. 1).
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Table 2 Percentages of participants at each GMFCS level with diagnosed comorbid conditions Diagnosed comorbid conditions
GMFCS
Epilepsy Intellectual disability Vision impairment Gastro oesophageal reflux disease Scoliosis
I 25 5 0 5 0
II 38 38 8 8 31
III 35 41 12 6 6
IV 50 39 29 18 29
V 72 57 29 29 46
Table 3 Numbers of categories of concerns expressed by participants (n = 154)
Fig. 1
Percentage of participants
1 2 3 4 5 6 7 8 9
7 11 19 17 19 12 8 2 3
Referrals to Sleep Solutions service included in study.
Table 1 Demographic characteristics of participants (n = 154) Participant characteristics Sex Male Female Age 1–5 years 6–12 years 13–18 years GMFCS I II III IV V
N
%
83 71
54 46
72 39 43
47 25 28
20 13 17 28 76
13 8 11 18 49
Participants’ ages ranged from 1 to 18 years (mean = 7.8; standard deviation = 5.4). Their age and GMFCS levels are shown in Table 1. Most participants had co-morbid conditions (Table 2). Participants at all GMFCS levels were diagnosed with epilepsy, intellectual disability and gastro-oesophageal reflux disease. Participants at all except GMFCS level I were diagnosed 1190
Number of categories
with vision impairment and scoliosis. The highest proportions of co-morbid conditions were at GMFCS levels IV and V. Reported concerns related to the participants’ sleep were assigned to 16 categories. These categories gradually evolved over time and were based on published literature, comprehensive clinical experience of clinicians in the sleep team, and a flexible in-home assessment that allowed participants to raise a very wide range of concerns regarding sleep. A description of each category is given in the Appendix. Nearly all participants (93%) had multiple concerns, with 25% having concerns in six or more categories (Table 3). The categories of concern varied between age groups (Table 4). In children aged 1–5 years, the main categories were settling routines, posture, sleep environment, communication and behaviour, temperature and perspiration, and reflux and digestion. For children aged 6–13 years, the main categories were posture, settling routines, movement control, reflux and digestion, temperature and perspiration, and safety. In individuals aged 13–18 years, the main categories were posture, pain, pressure, breathing, and temperature and perspiration. Categories of concern also varied across GMFCS groups (Table 5). For individuals with mild motor impairment (GMFCS levels I and II), sleep routines, settling routines, daytime activities, behaviour and sensory regulation predominated. Children and young people with severe motor impairment (GMFCS IV and V) had concerns mostly related to posture, movement control, breathing, reflux and digestion, and temperature and perspiration.
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Table 4 Percentages of participants at each age group in each category of concern Age (years) Categories of concerns
1–5
Body position Movement Breathing Reflux/digestion Pain Pressure care Temperature/perspiration Seizures/medications Safety Continence Health issues Sleep environment Settling routines Daytime activity and routines General behaviour and communication Sensory regulation responses
6–12
67 26 25 32 8 3 33 22 29 6 22 42 68 28 35 34
13–18
69 36 68 31 18 13 31 15 31 5 10 28 51 13 28 15
86 26 33 9 47 37 33 5 23 5 12 14 23 9 14 9
Black shading indicates very high frequency (75–100%), dark grey shading high frequency (50–74%), light shading moderate frequency (25– 50%) and white shading low frequency (0–25%).
Table 5 Percentages of participants at each GMFCS level in each category of concern GMFCS Categories of factors of concern
I
II
III
IV
V
Body position Movement Breathing Reflux/digestion Pain Pressure care Temperature/perspiration Seizures/medications Safety Continence Health issues Sleep environment Settling routines Daytime activity and routines General behaviour and communication Sensory regulation responses
10 5 5 10 0 0 20 5 10 10 25 60 100 60 50 40
31 0 8 8 23 0 31 15 15 0 8 38 92 38 69 23
71 6 0 6 6 6 24 12 18 12 18 29 53 12 29 41
86 46 21 14 18 11 29 7 21 4 14 21 43 7 11 14
92 38 46 41 25 25 39 22 39 4 16 25 34 11 20 7
Black shading indicates very high frequency (75–100%), dark grey shading high frequency (50–74%), light shading moderate frequency (25– 50%) and white shading low frequency (0–25%).
Discussion The purpose of this study was to describe the factors concerning sleep of children and young people with CP, and their families,
as identified by individualised assessment in their home setting. The main findings were that: (i) most children and young people had multiple factors of concern, (ii) factors of concern differed with age, and (iii) there were concerns regarding sleep for children and young people at every GMFCS level. In this study, most participants (over 80%) had three or more factors of concern relating to their sleep (Table 3). Given the complex presentations of CP, this is not surprising. The participants in this study do not represent a random sample of the CP population but were referred to the sleep service because of reported difficulties. Therefore, sleep difficulties would be expected to be higher in this sample than in the CP population. Factors were included only if they were reported as concerns at the time of assessment. Factors that did not interfere with sleep (e.g. well-managed incontinence) were not recorded. Therefore, this is not a study of the overall prevalence of these factors in children and young people with CP, nor does it address the unique psychosocial or emotional aspects that determine families’ capacity to deal with the associated challenges of everyday life. Nevertheless, this study adds a home-based perspective to the previous studies which report the wide diversity of concerns in relation to sleep for this population.8,10–13,19,20,28–30,32–34,40–42 It shows that, in clinical practice, identification of only one or two sleep concerns may greatly underestimate the extent of the problems. Effective sleep assessment must be broad enough to screen for all kinds of sleep concerns, and focused enough to target the specific areas of concern for each individual. This study found that factors of concern for sleep in children and young people with CP vary with age (Table 4). Young children had frequent concerns related to sleep environment, settling routines, daytime activities and routines, behaviour and communication, and sensory regulation responses. The reasons for this were not investigated in the study but reflect expected developmental factors. Elsayed et al.42 reported that in typically developing children, more preschool children than older children had difficulty getting to sleep. Likewise, Annaz et al.43 reported that psycho-social factors affecting sleep tend to improve with age. In the present study, some medical factors (management of reflux and digestion, effects of seizures and medications, and general health issues such as ear infections) were identified as concerns for more of the younger children. Parents and medical specialists may develop a better understanding of the children’s health-care needs as they get older, enabling them to tailor care and reduce the burden of concern. Management of posture, uncontrolled movements, and temperature and perspiration were of concern for all age groups, possibly reflecting the unchanging neurophysiology of CP. It is not surprising to see that breathing issues were of concern across all age groups. Sleep breathing problems are well described for this population, with previous studies reporting difficulties related to upper airway obstruction, oropharyngeal motor problems, gastro-oesophageal reflux, poor cough and airway clearance and respiratory muscle weakness.29,30,33,41 Management of pain and of pressure injuries was noted as concerns for over a third of the older age group. This reflects the serious impact of musculo-skeletal changes as children with CP grow.44,45 The present study also shows that there are concerns regarding sleep for children and young people at every GMFCS level.
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Individuals at all GMFCS levels were referred for assistance with sleep difficulties. Those with less severe gross motor impairment (GMFCS I and II) had factors of concern mostly related to their settling routines, sleep environment, daytime activities and routines, behaviour and communication, and sensory regulation responses. It is important to recognise that children and young people with less severe gross motor impairment have sleep difficulties and should not be overlooked in screening, assessment and interventions. There is a risk of under-identification in this group because parents may not report their concerns,46 and health professionals may be less inclined to ask about the sleep of individuals who appear to have a ‘mild’ disability. Indeed, individuals with minor motor disabilities were excluded from the study by Hemmingsson et al.8 because those with moderate to severe motor disabilities were expected to have the more serious sleep problems. As noted by Jan et al.,19 it is especially important to recognise that children with ‘mild’ CP have sleep difficulties that stem from, and contribute to, their known difficulties with behaviour and attention. Clinicians need to ensure strong working links with psycho-social teams to help to manage these behavioural aspects of sleep and daytime function. Children and young people with more severe gross motor impairment (GMFCS IV and V) had factors of concern related to impairment of their body structure and function, with most frequent concerns related to their posture, movement control, breathing, digestion, pain, pressure care, temperature and safety. For these individuals, it is especially important that allied health and medical teams are well linked for effective co-ordination of the assessment and management of these factors in both the clinical and home setting. There is possible referral bias in this retrospective study, with 50% of participants at GMFCS level V (Table 1). Nearly all children and young people at GMFCS V (92%) were referred to the service on the basis of concerns regarding their posture and positioning. This reflects prevailing concerns of the referring agents (mainly physiotherapists and occupational therapists) about the need for postural care, which is most apparent in this group (Table 5). This limits the generalisability of the results. Nonetheless, all levels of GMFCS are represented in this study, and concerns were expressed at all levels. The present study indicates that factors of concern for sleep of children and young people with CP are diverse and complex, with effects at all ages, and all levels of gross motor function. They cross all bio-psychosocial domains, with physiological, environmental and personal factors interacting and affecting sleep. Medical, allied health and psycho-social clinicians need to ask about, screen for and provide targeted assessment and well-co-ordinated intervention for sleep for all children and young people with CP. Although medical concerns may be addressed in clinical setting, a home-based approach is also needed to ensure that unique and diverse needs are well met. This study does not report on effectiveness of the interventions that were provided for this very heterogeneous group. Intervention is multi-faceted, individualised and provided alongside educational, recreational, therapy, social and medical services, to meet unique and varied family life priorities. Clinicians are encouraged to seek opportunities for targeted research to measure and report on the effectiveness of home-based interventions. 1192
Acknowledgements We gratefully acknowledge the co-operation of the participants and their families in this study, and the assistance of Linda Mercer and Penny Walker from the clinical sleep service at Ability Centre. This study was undertaken with financial support of the Developmental Occupational Therapy Interest Group of Western Australia.
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Appendix I Categories of Factors of Concern Reported by Parents of Children with Cerebral Palsy Who Were Referred to the Sleep Service Category of factor of concern
Description
1
Body position
2
Movement
3
Breathing
4
Reflux/digestion
5
Pain
6
Pressure care
7
Temperature/perspiration
8
Seizures/medications
9
Safety
Concern regarding the child’s posture when lying in bed: Therapists report increasing severity of scoliosis or hip dislocation, or positioning support required following orthopaedic surgery. Concern regarding involuntary, uncontrolled movements: Parents report that the child slides down the bed, rolls against side rails or hits arms or legs against wall. Concern regarding the child’s breathing during sleep: Parents report that the child has poorly controlled oral secretions, poor swallow, identified apnoeas, history of chest infection. Concern regarding the child’s ability to manage liquid and oral intake: Parents report concerns about reflux and vomiting during night; need to manage night-time feeding regimes. Concern related to the management of pain: Parents report that the child has musculo-skeletal and/or gastro-intestinal pain when lying in bed. Concern regarding risk of pressure injury: Child has signs of skin breakdown due to pressure or shearing forces from lying and moving in bed. Concern regarding management of the child’s temperature or excessive perspiration: parents report that the child has cold extremities, or seems hot during sleep, with evidence of discomfort, agitation, flushed, sweaty. Concern regarding the effects of seizures, or epilepsy medications: Parents report that the child has excessive daytime sleepiness following seizures, or following administration of anti-epileptic medication. Concern regarding the child’s safety in bed: Parents report that the child inadvertently moves into potentially dangerous positions in bed, falls out of bed or actively climbs out of bed. Concern regarding the management of continence: Parents report that they need to tend to the child during the night, due to the need to use toilet or inadequacy of continence aids. Concern regarding general health issues: Parents report that the child has ear infections, urinary tract infections, asthma, hay fever, allergies, skin conditions which they believe are affecting their comfort and sleep. Concern regarding the environment for settling and for sleep: Parents report that household factors (space available, other people in the household, evening and night time activities in the house, cooling and heating) affect their capacity to provide a quiet, dark and comfortable sleep environment for the child. Concern regarding the child’s settling routines: Parents report that the child does not engage in calming activities before bedtime, and requires parental attention and devices as they fall asleep or settle back to sleep during the night (e.g. being held, singing, massage, drinking from a bottle, falling asleep in parents’ bed). Concern regarding the effects of child’s participation in daily activities and routines: Therapists and parents note that the child spends time sleeping at school, does not participate in daytime physical activity and does not have regular time spent outdoors, in sunlight. Concern regarding the child’s general behaviour and communication: Therapists and parents report that the child has difficulty expressing their needs, understanding instructions and routines, or has behaviour management difficulties which affect daytime as well as sleep time. Concern regarding the child’s management of alertness and arousal: Therapists and parents report that the child has difficulty regulating their activity levels; they are ‘revved up’ at bedtime and were identified through sensory assessment as having sensory modulation difficulties.
10
Continence
11
Health issues
12
Sleep environment
13
Settling routines
14
Daytime activity and routines
15
General behaviour and communication
16
Sensory regulation responses
1194
Journal of Paediatrics and Child Health 51 (2015) 1188–1194 © 2015 The Authors Journal of Paediatrics and Child Health © 2015 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
