DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY

REVIEW

Association between motor functioning and leisure participation of children with physical disability: an integrative review PARIMALA S KANAGASABAI 1

| HILDA MULLIGAN 1 | BRIGIT MIRFIN-VEITCH 2 | LEIGH A HALE 1

1 Centre for Health, Activity and Rehabilitation Research (CHARR), School of Physiotherapy, University of Otago, Dunedin; 2 Donald Beasley Institute, Dunedin, New Zealand. Correspondence to Hilda Mulligan, School of Physiotherapy, PO Box 56, Dunedin 9016, New Zealand. E-mail: [email protected]

PUBLICATION DATA

Accepted for publication 31st May 2014. Published online ABBREVIATIONS

ABPI CAPE DCD ICF

LAQ-CP MACS MMAT

Acquired brachial plexus injury Children’s Assessment of Participation and Enjoyment Developmental coordination disorder International Classification of Functioning, Disability and Health Lifestyle Assessment Questionnaire Manual Ability Classification System Mixed Method Appraisal Tool

AIM The aim of this integrative review is to synthesize the evidence of association between motor functioning and leisure participation of children with physical disability. METHOD We conducted a systematic electronic search of key databases from the period 1 January 2001 to 30 April 2014 using relevant keywords. The Mixed Method Appraisal Tool was used to assess the quality of the qualitative and quantitative studies included and carried out in children with physical disability aged 6 to 12 years. We synthesized and compared information from both type of studies to identify the extent, and way by which, motor functioning influences leisure participation. RESULTS Thirty-five studies were included and analyzed for this review. Twenty-four studies used quantitative and 11 studies used qualitative methodology. We identified the association between motor functioning and six dimensions of participation including diversity, intensity, context, preferences, enjoyment and quality of participation. Motor functioning was found to have a weak to moderate association with involvement in specific leisure activity types and dimensions. We developed a conceptual framework to illustrate the influence of motor functioning on leisure participation from this review. INTERPRETATION Whether motor functioning poses a barrier to leisure participation for children with physical disability appears to depend on the functional movement required to participate in a particular leisure activity.

Involvement in leisure activities or non-mandated, outof-school activities is vital for all children, and no less so for the development of a child with physical disability. The development of physical, social, and psychological skills occurs by engaging children in a challenging activity with others. Social engagement improves confidence and develops a sense of self-competence and belonging.1 However, children with physical disability are found to be restricted in their participation in leisure activities, both in the frequency of participation and in the diversity of involvement in leisure activities.2,3 According to the International Classification of Functioning, Disability and Health (ICF), leisure participation is a major component of community, social, and civic life. It is defined as engagement in play, sports, art and culture, crafts, hobbies, or social activities. Such activities could be further classified into formal (organized activities involving rules) and informal (unorganized) activities, which impose different demands on a child with physical disability.4 Some authors also classify these into activity types such as recreational, physical, social, skill-based, and self-improvement activities.5 According to Palisano et al.,6 optimal participation is a subjective construct related to physical engagement (child’s involvement in an activity), social engagement (interpersonal interactions during an © 2014 Mac Keith Press

activity), and self-engagement (child’s enjoyment and learning). For children with physical disability, optimal participation occurs as a result of dynamic interaction of factors (child, family and environment) and the physical, social, and self-engagement dimensions of participation. Understanding the factors that influence leisure participation of children with disability is important in addressing the barriers to participation. Recent reviews suggest that functional ability, personal factors, family and peer support, and a physical, social, or attitudinal environment can all play an important role in influencing leisure participation in a child with physical disability.2,3,7–10 For example, in a study involving 427 children in Canada, structural equation modelling identified that functional ability (cognitive, communicative, and motor functioning), preferences, and family involvement in social and recreational activities directly predicted a child’s leisure participation, while factors such as family cohesion and supportive or unsupportive environments indirectly affected leisure participation.11 Further, factors affecting leisure participation change with increasing age. Thus, participation of school-aged children is different from that of pre-schoolers, who mostly depend on family for involvement in leisure activities, and adolescents, who are more focused and participate in fewer activities.7 DOI: 10.1111/dmcn.12570 1

Whether to focus on the child or modify the environment to improve participation is a dilemma for those wishing to provide intervention or interventional strategies.12 Many recent therapies are successfully using new forms of interventions to improve a child’s motor functioning to accomplish a task. These interventions include the practice of functional skills in natural environments and using adaptations and compensation strategies such as modifying the way an activity is performed.13–15 Children with high performance capacity may adopt more skilled compensatory and coping strategies to deal with a challenging environment.16 In recent years, functional training has become an important aspect of rehabilitation.17 In fact, functional therapy has been shown to significantly improve the social functioning of children with cerebral palsy (CP).18 A recently developed intervention termed ‘participation-based therapy’ also emphasizes the need to identify the physical skills required for an activity, measure current performance, and implement practice of necessary physical skills for the achievement of a goal.6 As modifying a child’s motor functioning is a basis for many therapeutic interventions, it is important to understand the association between motor functioning and leisure participation. Although recent reviews consider motor functioning as one of the determinants of leisure participation, none of the reviewed studies specifically explored the extent and phenomenon by which motor functioning influences leisure participation. Hence, the aim of this review is to examine the association between motor functioning and leisure participation, and to examine how and to what extent motor functioning influences leisure participation.

Operational definitions For the purpose of this review, we define physical disability as lack of ability to perform an activity or task required for daily living resulting primarily from a physical impairment. We define motor functioning as the ability to use the musculoskeletal system to execute a motor task for activity of daily life including self-care, mobility, or dexterity. In this review, we include measures of gross motor, fine motor, and self-care functioning assessed either as ability to function in a controlled environment (capacity) or as functioning in a real-life environment (performance). We define leisure participation as involvement in play, sports, art and culture, crafts, hobbies, or social activities.4 METHOD Design We used an integrative review to better understand the phenomenon of concern (i.e. to explore and understand the association between motor functioning and leisure participation). This approach allows for the inclusion and analysis of studies with diverse methodologies such as qualitative, quantitative, and mixed method studies. Unlike systematic reviews that synthesize evidence for the efficacy of specific interventions, such as randomized controlled trials, integrative reviews can be used for broader research questions. 2 Developmental Medicine & Child Neurology 2014

• • •

What this paper adds Motor functioning has weak to moderate association with specific leisure participation dimensions. Influence of motor functioning varies based on the leisure participation requirements. Successful leisure participation depends on choosing, adapting, and engaging in suitable leisure activity.

Moreover, integrative reviews can combine data from various types of research for a wider range of purposes, such as to define concepts, review theories and evidence, and to analyze methodological quality.19

Procedure The primary reviewer (PK) conducted electronic searches of the databases MEDLINE, EMBASE, PsycINFO, AMED, ERIC, CINAHL, and SCOPUS. Specifically, articles published between 1 January 2001 and 30 April 2014 were considered owing to the emergence of World Health Organization’s ICF model in 2001. The search terms used were ‘Children’ AND ‘Participation’ AND ‘Leisure activity OR recreation OR physical activity OR sports OR activities of daily living OR play’. After the removal of duplicates, two reviewers (PK, HM) independently screened the titles, abstracts, and full texts based on the selection criteria. Studies that investigated or explored the association between physical functioning and leisure activities in children with physical disability aged 6 to 12 years using either a cross-sectional, case–control, qualitative, or mixedmethod study design were eligible for inclusion. The following studies were excluded: (1) studies with child participants younger than 5 years or older than 13 years old; (2) review studies, theses, and conference proceedings; (3) studies published in languages other than English; (4) studies that investigated activities in exercise or school programmes; (5) quantitative studies on children with a wider age group where the mean age or more than 50% of participants were not in the age range 6 to 12 years; and (6) quantitative studies on children with disability in which more than 50% of participants did not fall into the category of physical disability as defined by this review. After the screening of studies according to the selection criteria, the two independent assessors disagreed on the inclusion of 39 articles. After discussion, a consensus was reached that only one of these 39 articles was eligible for inclusion in the review. The primary reviewer (PK) handsearched the references of the included studies for any additional studies deemed eligible for the review. Quality evaluation The first reviewer (PK) and two other assessors (HM and HD) then independently assessed the quality of the included articles using the Mixed Method Analysis Tool, version 2011 (MMAT, McGill University, Montreal, QC, Canada).20 This tool is used for the appraisal of the methodological quality of qualitative, quantitative, and mixed method studies. Although a newly developed tool, it has

been reported to have good reliability, with an inter-rater reliability of 0.72 pre- and 0.95 post-discussion among assessors.21 The MMAT tool has four criteria for each type of study and scores can vary from 25% (one quality criterion met) to 100% (all quality criteria met). Table I illustrates the methodological quality criteria of the MMAT tool. Following quality assessment, the primary reviewer (PK) extracted data into two tables, developed for quantitative and qualitative studies respectively. Data were extracted according to the study design, participant details, sample size, outcome measures, MMAT scores, themes (for qualitative studies), and main findings related to the association between motor functioning and leisure participation. The second reviewer (HM) rechecked data extraction in a random sample of five studies.

Data analysis and synthesis The included studies had investigated different dimensions of leisure participation. In order to organize the data into a manageable framework, we grouped together the studies investigating specific dimensions of leisure participation. We identified the dimensions of participation investigated directly from the outcome measures used in quantitative studies. The themes and illustrating quotes from qualitative studies were then mapped to these dimensions. A constant iterative comparison method19 was applied to the data. In this process, we compared the data from quantitative and qualitative studies and looked for similarities and differences in the findings of the included studies to explore the influence of motor functioning on leisure participation dimensions. RESULTS The initial search revealed 2196 studies after the removal of duplicates. Following title and abstract screening, 96 articles were selected for full-text screening. From the full

text, 31 articles were included for the review with the addition of three articles from the hand search of references and one article resulting from author contact. Figure 1 illustrates the selection process for the review in a flow chart. Of the 35 articles included in the review, 24 studies used quantitative methodology (18 cross-sectional, one longitudinal), 10 studies were qualitative, and one study used a mixed method study design. However, the quantitative component of this particular study was not eligible for the review (only six children completed the study survey and motor functioning was not measured) and so we considered only the qualitative part of the study in our analysis. The percentage agreement between first reviewer and the two second reviewers (HM and HD) for quality rating of studies was excellent (j 0.82). The quality of all qualitative studies was rated 75% or above. The quality of nine quantitative studies was rated 75% or above and 15 studies rated were 50%. The methodological quality assessment of the included studies is outlined in the data extraction table (Tables II and III) of included studies. The studies included in our review mainly explored the influence of motor functioning on six dimensions of leisure participation. These dimensions were ‘diversity’, ‘context’, ‘preferences’, ‘enjoyment’, ‘quality’, and ‘frequency’ of participation. A conceptual framework that illustrates how motor functioning relates to leisure participation, developed from our analysis, is shown in Figure 2. We describe the association of motor functioning to the six dimensions of leisure participation below.

Diversity of leisure participation Out of nine quantitative studies which investigated the association between motor functioning and leisure participation diversity, six studies included children with CP, two studies included children with developmental coordination

Table I: Mixed Method Appraisal Tool quality rating Study designsa

Methodological quality criteria

1. Qualitative

1.1. Are the sources of qualitative data (archives, documents, informants, observations) relevant to address the research question (objective)? 1.2. Is the process for analyzing qualitative data relevant to address the research question (objective)? 1.3. Is appropriate consideration given to how findings relate to the context, e.g. the setting, in which the data were collected? 1.4. Is appropriate consideration given to how findings relate to researchers’ influence, e.g. through their interactions with participants? 2.1. Are participants (organizations) recruited in a way that minimizes selection bias? 2.2. Are measurements appropriate (clear origin, or validity known, or standard instrument; and absence of contamination between groups when appropriate) regarding the exposure/intervention and outcomes? 2.3. In the groups being compared (exposed vs. non-exposed; with intervention vs. without; cases vs. controls), are the participants comparable, or do researchers take into account (control for) the difference between these groups? 2.4. Are there complete outcome data (80% or above), and, when applicable, an acceptable response rate (60% or above), or an acceptable follow-up rate for cohort studies (depending on the duration of follow-up)? 3.1. Is the sampling strategy relevant to address the quantitative research question (quantitative aspect of the mixed methods question)? 3.2. Is the sample representative of the population understudy? 3.3. Are measurements appropriate (clear origin, or validity known, or standard instrument)? 3.4. Is there an acceptable response rate (60% or above)?

2. Quantitative non-randomized

3. Quantitative descriptive

a

Study designs and quality criteria relevant to the included studies in this review taken from Mixed Method Appraisal Tool.

Review

3

AMED - 92; CINAHL - 449; EMBASE - 1093; ERIC - 102; MEDLINE - 1156; PSYCINFO - 141; SCOPUS - 421

Titles: 3454

Removal of duplicates (1258)

Titles: 2196

Title and abstract screening

96 studies

Reasons for exclusion (n = 65): Full text screening

Not relevant = 18 Not specific to age criteria = 9

31 studies

Not specific to population criteria = 6 3 hand-searched, 1 from author contact,

Not specific to leisure participation = 13 Motor functioning not measured = 7 Motor functioning not compared to

35 studies

leisure participation = 12

Figure 1: Flow chart on selection process of integrative review.

disorder (DCD), and one study included children with acquired brachial plexus injury (ABPI). In studies focusing on children with CP, one study suggested that the group of children with better walking ability (Gross Motor Function Classification System [GMFCS] level I) scored significantly more than the other two groups (GMFCS levels II/III) and (GMFCS levels IV/V) in diversity of participation scores in all activity types of the Children’s Assessment of Participation and Enjoyment (CAPE) questionnaire.22 In this study, the only exception was in social activities, for which the effect of GMFCS was not significant.22 In contrast, another study reported that children were involved in a variety of leisure activities unless they had severely impaired walking ability. The diversity of leisure participation was similar for all children in GMFCS levels I–IV, which differed from the participation of those in level V, who have severe mobility restrictions. Although in children with mild to moderate disability (GMFCS levels 4 Developmental Medicine & Child Neurology 2014

I–IV), walking ability strongly correlated with diversity in informal activity participation (r=0.75), manual ability was found to be the best predictor, explaining 22.6% of variance in diversity of participation in informal activities. Neither manual ability nor walking ability predicted diversity of formal leisure participation.23,24 In another study, gross motor functioning predicted 23% of the variance in diversity of participation in active physical activities (b=0.03).25 Moreover, motor functioning (b=0.04) together with IQ (b=0.08) predicted 21% of variance in participation in informal activities.25 Similar results were also found in a study in which GMFCS level moderately predicted diversity of participation in active physical activities (b= 0.46) and all types of activities except social activities.26 In children with ambulatory CP, ambulatory activity was significantly associated with sports club participation on weekend days and not on school days, suggesting that other factors influenced sports participation on school days.27

Review

5

Cross-sectional

Cross-sectional

Cross-sectional

Cross-sectional

Cross-sectional

Cross-sectional

Cross-sectional

Cross-sectional

Prospective, cross-sectional analytical study Cross-sectional

Cross-sectional

Cross-sectional

Cross-sectional

Imms, 200823

Imms, 200924

Majnemer, 200825

Longo, 201326

Van Wely, 201227

Jarus, 201128

Fong, 201129

Bae, 200930

Palisano, 200939

Majnemer, 201041

Kerr, 200744

Morris, 200645

Beckung, 200246

Orlin, 2010

Cross-sectional

Method

22

Author, year

CP/5–8y

CP/6–12y

Ambulant CP/5–16y

CP/6–12y

CP/6–12y

BPBP/6–18y

DCD/6–12y

Ambulatory CP GMFCS I–III/7–13y DCD/5–7y

CP/8–18y

CP/6y 1mo– 12y 11mo

CP/10–12y

CP/10–12y

CP/6–12y

Population/ age range

Table II: Data extraction of included quantitative studies

176

129

59

55

291

85

81

25

62

199

67

108

114

291

Sample size (n)

GMFCS, BMFM

GMFCS, MACS, ASK

GMFM

GMFM

GMFCS

PODCI (global functioning

MABC-2

Ambulatory activity (StepWatch Activity Monitor) MABC VMI–Visual motor integration

GMFCS

GMFM

GMFCS, MACS

GMFCS groups (level I, level II or III, and level IV or V) GMFCS, MACS

Functional skill

Outcome measures

LAQ-CP (social integration): Degree of impact LAQ-CP (social integration): Degree of impact ICF (social relations): Extent/magnitude of problem

PAC: Preferences

CAPE: Context: With whom, Where

Sports participation: Status (yes/no)

CAPE: Diversity, Intensity

CAPE: Diversity, Intensity, Context, Enjoyment PAC: Preferences

Sports participation: Status (yes/no)

CAPE: Diversity, Intensity

CAPE: Intensity, Diversity, Enjoyment

CAPE: Diversity

CAPE: Diversity, Intensity

CAPE: Diversity, Intensity

Participation (dimensions)

MABC scores correlated significantly with intensity and diversity of informal activities. No significant correlation between MABC and context of participation except social activities scores. Motor functioning influences preferences of active physical activity and formal activities Motor ability explained 7.6% of variance in participation diversity. Motor ability did not correlate with participation intensity scores of CAPE No statistical difference in PODCI scores between children with and without sports participation was observed Children in level I did a higher percentage of activities with friends and others and outside home than children in levels II and III and those in levels IV and V High GMFM scores along with younger age, female sex, and motivation significantly predicted 51% of variance in preference for skill-based activities Degree of correlation between GMFM and social integration was modest and accounted for only 27% of variation between measures Extent of problem with social integration correlated moderately with GMFCS and ASK (r=0.53 and r= 0.58 respectively) GMFCS correlated well with extent of problem in social relations (b=0.327)

For formal and informal activities: GMFCS I>II/III>IV/V. Walking ability may improve the opportunities but is not essential for leisure participation Diversity and intensity of participation was similar for children in each level of the MACS and the GMFCS, except for participants in level V Manual ability was the most notable predictor of activity. No evidence that GMFCS contributed to participation (r= 0.56, 0.47) in children with mild to moderate disability GMFM and aetiology explain 42% of variance in intensity of participation in active physical activities GMFM and higher IQ explain 21% of variance in diversity informal activities. GMFM also predicted diversity in active physical activity and skill-based activities GMFCS predicted weak-moderate association with diversity and intensity of participation in all activity types except social activities (b= 0.9) Low ambulatory activity associated with no sports club participation at weekends but not on weekdays

Findings

50

75

75

75

50

50

50

50

50

50

100

75

50

50

MMAT scores (%)

6 Developmental Medicine & Child Neurology 2014

Cross-sectional

Cross-sectional

Cross-sectional

Retrospective study

Cross-sectional

Cross-sectional

Cross-sectional

Longitudinal study

Cross-sectional

Cross-sectional

Parkes, 201047

Bjornson, 201448

Voorman, 200649

Foo, 201250

King, 200651a

Palisano, 201152

Kelly, 201153

King, 200954

Cairney, 200555

Poulsen, 201156 DCD/10–13y

DCD/9–14y

Physical disability/6–14y

Spina bifida/6–12y

CP/6–12y

Physical disability/6–14y

ABI/5–18y

CP/9–13y

Ambulatory CP/2–9y

CP/8–12y

Population/ age range

59

44

409

21

288

427

28

110

128

102

Sample size (n)

M-ABC, two groups: severe DCD (5th centile MABC)

BOTMPSF: to categorize DCD status (yes/no)

Ambulatory status, bladder and bowel needs ASK

GMFCS

ASK

PEDI (Self-care, mobility function)

GMFCS

Average strides/d (walking performance)

GMFCS

Functional skill

Outcome measures

Participation questionnaire (PQ) for Physical Activity (PA): Frequency Leisure survey questionnaire and 7-d diary: Frequency

CAPE: Intensity

CAPE: Intensity

CAPE: Intensity

Life-H (social roles – recreation, interpersonal relations): Degree of difficulty and type of assistance Life-H (recreation): Degree of difficulty and type of assistance PEDI (Subscale: Social life) and VABS (Subscale: Socialization): Capacity, Caregiver assistance CASP (community participation): Age expected participation CAPE: Intensity

Participation (dimensions)

Participation frequency was similar for children with moderate and severe DCD. Most participation in organized group activities with low energy expenditure (choir, band, or chess club) was linked with poor motor performance across areas

Physical functioning scores significantly correlated with intensity of participation in active physical activities (r=0.42), social activities (r=0.30), and recreational activities (r=0.15) GMFCS significantly predicted intensity of participation (b= 0.27) along with enjoyment, adaptive behaviour, age, family activity, and orientation, together explaining 32% of variance Participation intensity did not differ based on the motor level, ambulation status (yes/no) or bladder/bowel issues Physical functioning was not associated with the slope of change, indicating that physical functioning may not affect the rate of change In this model, 28% of the variance in children’s PA was predicted by generalized self-efficacy and DCD

No significant correlation between PEDI scores at discharge and community participation scores at least 3mo after discharge

No significant association was found between motor ability and social functioning

Average strides per day significantly associated with assistance required for recreational activity (b=0.35)

GMFCS related to social activities with the final models explaining about 39% of variance in recreation and 7% of variation in interpersonal relations

Findings

50

75

75

50

50

75

50

50

50

75

MMAT scores (%)

King, 2006: this article was included by contacting the author. MMAT, Mixed Method Appraisal Tool. Population: BPBP, brachial plexus birth palsy; CP, cerebral palsy; DCD, developmental coordination disorder; ABI, acquired brain injury; SCI, spinal cord injury. Motor function measures: GMFCS, Gross Motor Function Classification System; MACS, Manual Ability Classification System; ASK, Activity Scale for Kids; GMFM, Gross Motor Function Measure; BOTMP-SF, Bruininks–Ozeretsky Test of Motor Proficiency –Short Form; MABC, Movement Assessment Battery for Children; MABC-2, Movement Assessment Battery for Children–Second edition; PODCI, Pediatric Outcomes Data Collection Instrument; BMFM, bimanual fine motor function; PEDI, Pediatric Evaluation of Disability Inventory. Participation measures: CAPE, Children’s Assessment of Participation and Enjoyment; PAC, Preferences for Activities of Children; LAQ-CP, Lifestyle Assessment Questionnaire–Cerebral Palsy; Life-H, Life Habits Questionnaire; CASP, Child and Adolescent Scale of Participation; VABS, Vineland Adaptive Behaviour Scale. Quality Assessment using MMAT: 25%, one criterion met; 50%, two criteria met; 75% three criteria met; 100% four criteria met. Note: studies with convenience sampling which did not report the response rate were scored as ‘No’ for this particular criterion.

a

Method

Author, year

Table II: Continued

Review

7

Method

In-depth interviews (phenomenology)

Six focus groups

Interview (twice) (phenomenology)

Interviews (phenomenology)

Author, year

Sandberg, 200431

Jones, 200332

Missiuana, 200733

Mandich, 200334 DCD/7–12y

DCD/6–14y

Children with disability/5–35y

Visual disability (5), motor disability (5), Asperger syndrome (5)/25–76y

Population/age range

Table III: Data extraction of included qualitative studies

12 parents

13 parents

37 parents

15 adults

Sample size (n) Either peers adapt their play or the person with disability adapts themselves in order to play. Play was considered a physical challenge and children were motivated to explore and cope with different physical activities Play increased independency Disability did not affect the experience of play per se Community programmes were competitive and required athletic ability and skill which were not possessed by children with disability Children who lacked social and behavioural skills were excluded from programmes Play differences in early childhood led to peer problems in middle childhood and emotional problems in later childhood Sedentary play patterns, problems with ride-on toys and team sports were common play differences Activities such as swimming and skating which require less eye–hand coordination were easier Inability to cope with peers in playing sports during recess Failure to acquire simple skills affects the self-esteem and willingness to try new things Acquisition of new skills enabled participation with peers, improved socialization, self-esteem, and self-confidence

Themes

‘He likes to play and when he is late for recess because it takes him longer to tie his shoes or put on his coat it frustrates him and isolates him.’ (p. 590)

‘She felt very insecure and she always wanted to make sure that I was there to catch her. And I always felt the need to catch her and felt the need to be behind her.’ (p. 89) ‘She does not play. She either sits there and receives information from the TV or reads.’ (p. 89)

‘If it’s real competitive it doesn’t work for them, because they don’t have the ability to–I mean, she gives it her all, but she doesn’t have the ability to help her team out. She wants to, but it just doesn’t work.’ (p. 58)

‘And then they adapted a lot, so I could participate in twist and skipping-rope.’ (p. 118) ‘I could never be bossy with them.’ (p. 118) ‘I can say I hung on to everything. . . in a typical playground then, that’s a swing, slide and climbing frame.’ (p. 121)

Relevant quotes

100

75

100

75

MMAT sores (%)

8 Developmental Medicine & Child Neurology 2014

Method

Focus group/individual phone interviews (phenomenology)

10 focus groups (five for children, five for parents)

Individual interviews (twice) Pictorial data (narrative enquiry)

Author, year

Shimmell, 201335

Verschuren, 201236

Hynes, 201237

Table III: Continued

SCI/8–11y

CP (ambulatory without assistive device)/7–14y

CP/9–21y (9–11y, five children)

Population/age range

Three children

33 children

17 youth and few parents

Sample size (n)

Physical activities improved energy levels Motor challenges led to decreased peer acceptance Vulnerability to injury made them hesitant to engage in sports programmes Choice of leisure changed after injury Children developed new skills and abilities A ‘can-do attitude’ towards leisure leads to successful participation and improved self-perceptions Some leisure activities were all-consuming and connected to emotions

Children do activities they enjoy Difficulty in keeping up with peers leads to frustration Travelling long distance and transfer in or out of adaptive equipment leads to fatigue

Themes

Initially, learning to play cricket was difficult for Michael but he added, ‘you develop skills’. (p. 366) Tyson changed his leisure activity from camping to cinema. He says cinema was ‘. . .one of the options. . .that’s fun for me’. (p. 365) Tyson advised ‘just try’ and ‘see if you like something and. . .just go for it’. (p. 366)

‘I feel good like after I do a workout, like swimming . . . Cause I’m like building muscle, I’m getting stronger and faster.’ (p. 413) ‘. . . when he’s with his friends, and his friends can do things right away, and he is, at the beginning it takes a lot of frustration because he just, by the time he moves from one place to another, everybody’s already gone.’ (p. 413) ‘The time it will take to learn an activity is often too long to stay motivated or to be accepted by peers.’ (p. 3) ‘I can see he has gained a lot of energy from his football training. His energy level was very poor before he started. Moreover, it was very good for his self-esteem.’ (p. 3)

Relevant quotes

100

75

75

MMAT sores (%)

Review

9

Method

Online focus group interviews

CAPE Photographs Interviews of children

Individual interviews

Author, year

de Jong, 201238

Harding, 200940

Columna, 201142

Table III: Continued

Physical disability, emotional disability, intellectual disability/5–14y

Physical disability (3), cardiopulmonary (1), emotional disability (2) (one child with motor disability aged 10y)

UCBED, three groups (8–12, 13–16, 17–20y)

Population/age range

12 parents

Six children

77 parents and children, 17 children in 8–12y group

Sample size (n) Only few activities were limited because they required two arms Children used other body parts for playing, such as to fix Lego Such as tying skipping rope around the short arm to be able to jump Use adaptive device, sports or adaptive musical instruments Enjoyment of participation level depends on both experience of place (feeling accepted and having choice of activity) A positive attitude would enhance participation in out-of-school activities Participants spoke predominantly of supports rather than barriers. Social supports came in the form of friends, nice helpers, pets, and neighbours Non-sport-related activities which required minimal organization (e.g. playing at community, cycling, etc.) were preferred by the participants Parents lacked skills to make modifications to sports or games

Themes

‘I need more time and someone to help me with my child.’ (p. 223) ‘He needs to be more active because of his disability and for that he needs a person to teach him, a specialist that can tell him what he should do.’ (p. 223)

Participants valued independence in activity and valued being alone. ‘Well . . . a lot of it’s because I really like playing with my stuffed animals in there and also I just like playing in there mostly.’ (p. 140)

‘Playing with Lego, he fixes the pieces using his foot. He does this especially with his big toe.’ (p. 889) ‘When I was playing a computer game with friends, and I didn’t succeed in holding the controller, we thought that a sweatband could be the solution.’ (p. 889)

Relevant quotes

100

100

75

MMAT sores (%)

MMAT, Mixed Method Appraisal Tool. Population: CP, cerebral palsy; DCD, developmental coordination disorder; SCI, spinal cord injury; UCBED, unilateral congenital below elbow deficiency. Participation measure: CAPE, Children’s Assessment of Participation and Enjoyment. Quality Assessment using MMAT: 25%, one criterion met; 50%, two criteria met; 75%, three criteria met; 100%, four criteria met. Note: studies with convenience sampling which did not report the response rate were scored as ‘no’ for this particular criterion.

100 ‘It is no different for a person who is walking, than a person who is rolling. . .It does not matter where you have it as long as you get to play.’ (p. 91) Michelle explained why she liked to do things like cycling and trampolining, ‘I guess they are just really fun and they, um, it does not really matter that I have a disability to do these things or not.’ (p. 91) Physical disability does not affect the selfperception or enjoyment of play activities It provided a sense of freedom Dependence on new people to help in toilet activities could make leisure activities in community difficult 15 children Individual interviews (phenomenology) Anderson, 200543

Females with physical disability/10–16y (three females aged 10y, one female aged 11y)

MMAT sores (%) Relevant quotes Themes Sample size (n) Population/age range Method Author, year

Table III: Continued

10 Developmental Medicine & Child Neurology 2014

Among studies that investigated children with DCD, one study found that motor functioning correlated well with overall participation diversity scores (r=0.58) and informal activities (r=0.58) and correlated moderately with self-improvement activities (r=0.49), recreational activities (r=0.37), active physical activities, and social activities (r=0.42).28 Another study reported that motor functioning was weakly correlated with total participation diversity score (r=0.264), formal activities (r=0.291), recreational activities (r=0.249), and skill-based activities (r=0.235).29 Both these studies provided only weak evidence as in one study28 the sample size was small (n=25) and in the other study29 motor functioning explained only 7.6% of variance in leisure participation diversity. In a study on children with ABPI, although 88% of the children played sports, the authors found no significant difference in the global functioning scores between those who played sports and those who did not get involved in sports.30 The findings of the qualitative studies suggested that motor functioning may influence participation in specific activity types. In one study, three adults with motor disability shared their experiences of inclusion and exclusion from play in childhood. Although impairment per se did not greatly contribute to these experiences, it seemed to affect certain play contents. Play activities such as those involving cycling and running were identified to be difficult for the participants. Moreover, in order to play with their peers they had to adjust themselves in various ways. A participant stated, ‘I could never be bossy with them.’31 Moreover, parents of children with physical disability reported that some recreation programmes in their community were competitive and required athletic skills, and that this restricted their children in participating in such programmes.32 Children with DCD were identified in one study to be skilled at sports which did not involve good eye–hand coordination, examples of these being swimming and skating.33 Two studies identified that inability to play might lead to reduced self-esteem in children and that this could potentially inhibit their readiness to try new things.33,34 In particular, inability to keep up with their peers led to frustration in some children, which restricted their participation in certain activities.35 Moreover, some parents considered that children with CP were susceptible to injury, and this resulted in the children being hesitant to become involved in sports. For example, one mother stated, ‘Having cerebral palsy and all the problems that come with it, like balance problems, increases the chance of having an injury. When our daughter (with CP) falls, she has a greater chance of breaking something than our (typically developing) son.’36 It was also perceived that some motor activities took longer for such children to learn, which reduced their motivation.36 Although some activities were not feasible for children, those with a ‘cando attitude’ chose and participated in activities suitable for them, such as a modified sport. For instance, in one study children with spinal cord injury reported that they

Quantitative studies

Does influence

Qualitative studies

Association to leisure participation

Occurs as barrier Diversity: Risk of injury

Diversity of informal activities and active physical activities Quality of social life and integration Frequency of active physical activities, recreational activities, and social activities Motor functioning

Does not influence Diversity of formal activities Enjoyment of overall leisure activities Preference for recreational, activephysical, social, and selfimprovement activities Frequency of skill-based and selfimprovement activities, change in level of leisure participation

Frustration, anger and reduced self-esteem due to failure lead to reduced willingness to try new things Context: Inability to cope with peers

Reduced participation in specific activities Reduced play with peers Preference for solitary activities

Dependant on family and friends for negotiating environment, toileting needs

Does not occur as barrier Diversity: Positive or ‘can-do’ attitude

Enjoyment and fun

Develop new skills for a new activity

Self-engagement in participation

Choose suitable or modified leisure activities

Feelings of ‘normality’

Skill training Context: Self-adjustment to play

Figure 2: Conceptual framework of association between motor functioning and leisure participation.

developed skills for playing a new sport and explored different leisure activities with optimism.37 In another study on children with unilateral congenital below-elbow deficiency, most children did not experience limitations with leisure activities, except a few activities such as judo, playing guitar, and rope climbing. The children followed strategies such as using other body parts (using legs to fix Lego) and creating adaptations for playing (for example, taping drumstick on short arm to play drums).38

Context of leisure participation Two quantitative studies investigated the influence of motor functioning on the context (where and with whom) of leisure participation. One study included children with CP and the other included children with DCD. Children with CP in GMFCS level I were involved in more activities with friends and outside the home than children in more severely affected groups (GMFCS levels II/III) and (GMFCS levels IV/V). In the three groups, the percentage of activities performed with peers was reported to be 25.2%, 19.1%, and 17.0% respectively, and the percentage of activities performed in the community was reported to be 40.3%, 34.2%, and 35.6% respectively.39 However, in children with DCD, motor functioning did not significantly correlate with the companionship of playing except for a weak correlation with social activities (r= 0.29).28 Similarly, qualitative studies reported that children’s choice of with whom and where they play depended on motor functioning to some extent. Participants in a case study design reported that, although some environments could be negotiated to enable involvement in leisure activities, some environments were not accessible (an example

being steps leading to a cinema or theatre). However, the participants of the study had a positive attitude and spoke about the support from friends, neighbours, and even pets that helped them access the environment. However, because of the design of this study and its small sample size (n=3), the findings may not represent the experiences of all children.40 Issues with motor functioning such as increased time to tie shoes or perform toileting activities in school recess periods were also found to restrict play activities of children with disability with their peers. However, a client-centred, problem-solving intervention changed the performance competency of children, resulting in improved self-esteem and socialization.34 In a different study, a child’s independence in motor functioning was also one of the factors which determined with whom the children played. One mother expressed the need to be present with her child with DCD while playing to look after her child’s safety (for example in case of a fall).33 Further, children’s experiences of joy were reported to be associated with the experience of places. Children preferred places where they were accepted and free to choose an activity.40

Preferences for leisure participation Two quantitative studies considered the effect of motor functioning on preferences of leisure participation. One study included children with CP and the other children with DCD. In children with CP, greater motor limitation, younger age, and higher motivation significantly predicted preferences towards skill-based activities (swimming or dancing), explaining 52% of the variance. However, the contribution of motor functioning towards the model was low (b= 0.008). Motor functioning did not influence the Review

11

choice of recreational, physical, or social activities or overall formal and informal activities.41 In children with DCD, motor functioning of children correlated with preferences in both active physical activities (r=0.30) and formal activities (r=0.32).28 Qualitative studies also support the idea that leisure activity choices of children depend on their motor functioning. In a study involving children with spinal cord injury, a child expressed how his choice of holiday camping trips before injury changed to going to the cinema (as an option for fun) after injury. Moreover, those children who were motivated and wanted to retrieve their ‘able’ identity chose a modified sport. A child narrates that playing wheelchair soccer would be more satisfying because it would give him ‘the feeling . . . that (he) (could) play sports again’.37 A study of children with DCD described them as being different in play to typically developing children. As toddlers they did not prefer to ride on toys. A mother stated, ‘He wasn’t showing any signs of even trying to sit on a tricycle or trying to ride a tricycle.’ Moreover, at a young age these children preferred sedentary play activities such as reading books, watching television, or quiet imaginary play instead of team sports.33 Two studies found that some children wanted to do things independently and so preferred playing alone.31,37 As well as children having preferences, one study identified that parents preferred non-sport activities for their children with disability and that these were activities which required minimal organizational skill, such as cycling or playing in community parks.42 While some parents preferred inclusive group programmes for their children, some preferred private classes to improve their children’s skill development.35

Quality of leisure participation We identified six quantitative studies that measured the degree of difficulty or dependence of leisure participation using ordinal scales in children with CP. Two studies reported a moderate correlation (q= 0.27 and r=0.52) between motor functioning and social integration using the Lifestyle Assessment Questionnaire (LAQ-CP).44,45 However, the LAQ-CP is limited in its scope of measuring leisure participation. In another study, GMFCS levels were moderately predictive (r=0.327) of participation restrictions in social relations in a five-level ordinal ICF scale which consisted of dimensions including mobility, education, and social relations. Again, this scale does not cover all the dimensions of leisure participation.46 Parents reported that walking ability determined 39% of the variation in difficulty with recreation activities.47 Another study obtained similar results, increased day-to-day walking performance being associated with decreased difficulty with recreational activities (b=0.35).48 However, there was no significant relationship between walking ability (GMFCS level) and performance in social life and socialization.49 In addition, motor functioning scores in children with acquired brain injury at discharge did not correlate with limitations in age-expected community participation at 3 months after discharge from hospital.50 Qualitative studies suggest that one of the factors that make leisure participation difficult is the dependence on others for self-care activities. For instance, a female with disability reported social activities to be difficult if she relied on others for helping with toilet activities.43 Further, travelling long distances and transfers in or out of adaptive equipment lead to tiredness in children even before initiating an activity.35

Enjoyment in leisure participation Besides one quantitative study which indicated no significant correlation between the level of enjoyment in leisure activities and the children’s motor abilities,28 many qualitative studies identified that the crucial factor was interaction with other children as opposed to motor functioning that enhanced enjoyment. Children with physical disability experienced great fun, a sense of freedom and belongingness while playing with friends.31,43 In an interview study of females with disability, a female participant stated, ‘Sometimes I just like to forget all about my disability and just have fun.’43 Another female participant also expressed the feeling of ‘normality’ while playing sports.43 Furthermore, children with spinal cord injury (n=3) reported that they were happy about their leisure experiences. With a ‘can-do approach’ they tried a variety of leisure activities with optimism.37 Moreover, one child reported that he enjoyed the relaxing effects of sedentary leisure activity such as watching television.37 However, some parents reported that their child with DCD experienced frustration and anger because of inability to keep up with peers, and that this isolated them during play at school recess.33

Frequency of leisure participation Thirteen quantitative studies investigated the association between the frequency of participation and the motor functioning of children. The studies included children with CP (six studies), complex physical disabilities (two studies), spina bifida (one study), and DCD (four studies). One study reported that children with CP with better walking ability participated more frequently in leisure activities.22 In a study of children with complex physical disability, motor functioning correlated moderately with frequency of active physical activities (r=0.42) and social activities (r=0.30).51 In a similar study, GMFCS was found to have direct influence on frequency of participation (b= 0.27).52 Majnemer et al.25 reported that gross motor functioning (b=0.02) together with aetiological determination (b= 0.68) predicted 42% of variance in the frequency of participation in active physical activities in children with CP. However, the predictive aetiological factors were not reported. Similarly, in another study, GMFCS levels predicted frequency of participation in physical activities (b= 0.47) and other activity types except social activities.26 In contrast, another study reported that frequency of participation was similar in children in GMFCS and Manual Ability Classification System

12 Developmental Medicine & Child Neurology 2014

levels I–IV, but that participation frequency was severely reduced in children in level V.23 Similar results were found in children with spina bifida, in whom ambulatory status (yes/no) or presence of bladder and bowel issues did not affect participation frequency. However, as a result of the low sample size, this study may not have been able to identify group differences.53 One longitudinal study investigated the rate of change of leisure participation over time and identified factors other than motor functioning to be the determinants.54 In studies involving children with DCD, two studies found no direct correlation between motor functioning and frequency of participation.29,55 One study, however, illustrated that DCD status could indirectly influence participation in physical activity via self-efficacy. DCD status and self-efficacy together explained 28% of the variance in children’s physical activity.55 Another study reported that frequency of participation in unstructured (street games) and structured activities (choir and band) was similar in children with moderately and severely impaired motor functioning.56 In contrast, Jarus et al.28 conducted a study in younger children with DCD and found moderate positive correlations between motor functioning and frequency of informal activities (r=0.41), active physical activities (r=0.29), social activities (r=0.29), and self-improvement activities (r=0.38). Although the interview studies in our review provided information on how motor functioning act as a barrier to participation, these studies did not explore what influences the frequency of participation.

DISCUSSION Using information from both quantitative and qualitative studies, we have examined the association between motor functioning and six dimensions of leisure participation in children with physical disability. We have also identified the way in which motor functioning influences engagement in leisure activities. It is evident from the analysis and synthesis of studies that the extent to which motor functioning affects leisure participation depends on the motor functional skills required for the involvement in a particular activity. We illustrated our findings in a conceptual framework built from high-quality studies (quality rated ≥75%) included in this review. This is illustrated in Figure 2. Evidence suggests that limitations in motor functioning moderately restricted frequency of leisure participation in active physical activity51,55 and recreational activities.47,51 Increased frequency of leisure activities, especially physical activities, may be beneficial for health and fitness.22,57 However, some authors argue that activities that are important to the child may have a greater impact on their quality of life than frequency of performing such activities.9,22 Limitations in motor functioning also appear to restrict the variety of informal activities and physical activities chosen.23,25 Although walking ability is not mandatory for participation, it may influence where and with whom a child plays.39 Reduced walking speed and endurance might

result in an inability to keep up with friends, especially outdoors and in the community.39 Thus, motor functioning could lead to limitations in social integration.44,45 The results from qualitative studies suggest that motor functioning might be a barrier to involvement in particular activities, such as running, cycling,31 team activities,33 and sports that involve risk of injury, such as team sports which could threaten the balance of a child.36 Reasons for selecting or avoiding certain activities may be that successful attempts in some activities lead to self-efficacy and enjoyment, whereas unsuccessful attempts in certain activities might lead to avoidance.28,29 Moreover, the longer time taken to learn certain activities, inability to keep up with their peers, and dependence on self-care activities could reduce self-esteem and readiness to try new activities and restrict play with friends.33,34,43 In addition, the passion for a particular leisure activity could restrict participation in diverse activities.40 On the other hand, motor functioning did not correlate with certain leisure participation dimensions, such as the number of formal activities chosen or the frequency of skill-based and self-improvement activities. Moreover, children preferred recreational, active physical, social, and selfimprovement activities irrespective of their motor functioning.41 Enjoyment in leisure activities seems to be unrelated to motor functioning of children. Children expressed their feelings of fun, freedom, belongingness, normality, and competency when engaging in leisure activities.25,28,31,37,43 Furthermore, limitations in motor functioning were not perceived as a barrier by children with a positive attitude; they preferred games or activities that were suitable for them.33,37 Children with an optimistic approach tried new activities and developed new skills,37 and adapted themselves to play.31 Interest or motivation to choose a satisfying activity is necessary for psychological engagement in an activity with peers, resulting in enjoyment. Enjoyment provides intrinsic motivation to engage in an activity and may also increase frequency of participation.1,11 Furthermore, specific skill training also facilitates leisure participation by improving the fitness, competency, self-esteem, and socialization of children.34,36 The inconsistent results reported in the quantitative studies of weak to moderate associations in their regression models suggest that motor functioning does not influence leisure participation dimensions to a great extent. Multiple factors affecting leisure participation with bi-directional relationships or interaction between them could explain this finding. Moreover, motor functioning might restrict involvement in specific leisure activities depending on the functional skill requirements of the activity. To encourage participation in a wide variety of leisure activities among children one should consider some of the barriers leading to unsuccessful attempts, risk of injury, or reduced selfesteem. Moreover, strategies to improve the motivation of children are important as they can induce a positive attitude and preferences towards leisure and play.25 Such children could explore new physical activities, develop new Review

13

skills, and cope with challenging environments.37 Some of the motivational strategies reported in the studies include playing with children who have similar physical skills, choosing activities according to the child’s ability or susceptibility to injury and offering adapted, non-competitive recreational programmes.32,33,36,40 Further, goal-directed interventions enabling simple skills acquisition can make a huge difference to the quality of leisure participation of children.34 Children might also benefit from psychosocial interventions to improve coping strategies to tackle limited motor functioning or reduced self-efficacy.55 Additionally, improving independence in self-care and daily activities may be useful for leisure activities in the wider community. Many of the quantitative studies used a convenience sampling strategy, except six studies23,24,44–47 which may not be representative of the population under study. Although four studies sampled children using convenience sampling, the authors suggest that they were representative.25,41,51,54 The studies that recruited participants via regional health or disability databases (which could be representative of the population under study) had a low response rate of