Social participation in adults with cerebral palsy: a systematic review of the evidence-base

Abstract

Purpose

To identify and synthesise the current evidence on social participation in adults with cerebral palsy (CP).

Methods

Four databases (PubMed, CINAHL Plus, PsycINFO, Web of Science) were systematically searched between December 2021 and February 2022. Pre-specified eligibility criteria were applied to all identified studies resulting in the inclusion of 16 articles. Data extraction was performed using a standardised tool and quality appraisal was assessed using the Mixed Methods Appraisal Tool. A narrative synthesis approach was taken for data analysis.

Results

The 16 included studies were rated as high (n = 11) and medium quality (n = 5). Numbers of participants included in the studies ranged from 7 to 335. Definitions of social participation were discussed. Common themes were identified: the impact of home and work environments on social participation, the importance of age-appropriate support and interventions, and the impact of limited autonomy on social participation.

Conclusions

Adults with CP experience limited social participation due to lack of appropriate support in childhood, issues across the lifespan including physical limitations when ageing, and factors such as societal expectations and inaccessible environments which limit opportunities for autonomy. Social participation may be improved by supporting families to provide opportunities in childhood, providing timely interventions, and by enhancing autonomy.

IMPLICATIONS FOR REHABILITATION

• Considering the support needs of the wider family, in order to build a supportive family environment in childhood, could improve social participation opportunities for individuals with cerebral palsy (CP) in adulthood.

• Social participation in adulthood may be improved by encouraging independence and autonomy in childhood and adolescence.

• Taking a lifespan approach to services for individuals with CP could improve social participation and better prepare them for the challenges of adulthood.

Keywords:

• Cerebral palsy
• social participation
• ageing
• adults
• WHO ICF
• systematic review

Introduction

Cerebral palsy (CP) is a complex condition, which affects 1.5–3.4 per 1000 live births [1] and encompasses a range of possible comorbidities such as movement disorders, sensory problems, speech impairment, seizures, and behavioural differences [2]. While much research, support and healthcare are focused on children with CP, most individuals with CP have similar life expectancies to that of the general population [3]. Although medically described as non-progressive, the experience of living with CP changes as an individual ages [4]: alongside the challenges that all individuals experience during adolescence and the transition to adulthood, adults with CP face additional difficulties in fully participating in society in later life due to pain, transportation needs and regular medical appointments [5,6]. Previous research has examined the transition from paediatric to adult healthcare [4,7–10], while others have focused on access to healthcare in adulthood [11] and the effects of exercise on quality of life of adults with CP [12], but few have explored the interrelationship of ageing with CP and social participation.

There are clear difficulties involved in defining “social participation,” as it is a term often used interchangeably with “participation.” Since the publication of the International Classification of Functioning, Disability and Health (ICF) in 2001 [13], the concept of participation has been a growing theme in publications from health and social care fields [14]. However, many studies fail to provide a clear definition of “participation”, and commonly blur the lines between it and social participation [15], as well as social/community integration, involvement, and engagement. In a systematic review of definitions of social participation, Levasseur et al. [16,p.2145] found 43 definitions across seven disciplines, the content of which varied, but were summarised as “the person’s involvement in activities that provided interactions with others in society or the community.”

The World Health Organisation [13,p.10] defines participation as “involvement in a life situation.” Levasseur et al. [17] note that difficulties in application of definitions that align with the ICF model could be related to the model’s attempt to integrate the opposing positions of medical and social model beliefs about disability. The medical model places health conditions of the individual as the main contributor to disability, whereas the social model considers the impact of environmental, social, and attitudinal barriers to be the predominant issue [18,19]. Combining the medical and social models has resulted in the individual’s environment becoming a sub-factor of participation in the ICF, rather than a key and defining component of participation.

The Institute for Social Participation [20] states that the term social participation shares close links with social inclusion and social capital, whereby individuals achieve social participation through social inclusion. This position is shared by Hyyppa et al. [22,p.390] who define social participation as “how actively the person participates in the activities of formal and informal groups.” Gao et al. [22] highlight that many studies measure social participation by counting the number of social activities an individual attends, whereas Imms et al. [23] argue that such counts effectively measure attendance, which does not necessarily constitute participation. For the purposes of this review, and due to the prevalence of the ICF model in the literature, the stance taken by Levasseur et al. [17] will be followed, in that the ICF can be interpreted as constituting social participation when “participation” and “activities” are considered together as involvement and engagement with community activities.

While there have been some improvements in the social participation of disabled adults in recent years [24,25], there remains a stark contrast in access to opportunities for individuals with CP in comparison to the general population [26,27]. The United Nations Convention on the Rights of People with Disabilities (UN CRPD) [28] highlights the right of all disabled individuals to have access to the environment and participation in political, public, and cultural life on an equal basis with others. Currently, levels of social participation and opportunities for disabled people to enhance their social participation are still considered to be limited [29], and so this systematic review sought to address the question, “what is the existing evidence base for social participation in adults with cerebral palsy?”

Methods

Information sources

Databases that included medical and healthcare publications, in addition to those with social sciences material, were used as information sources for a systematic search. These included PubMed, CINAHL Plus, PsycINFO and Web of Science. Searches were conducted between December 2021 and February 2022. Reference lists of included articles were hand-searched for additional relevant papers.

Search strategy

Similar search strategies were used for all databases, with slight editing of truncation symbols and proximity searching to allow use in different databases. A combination of Medical Subject Headings (MeSH) and key words were used to provide a comprehensive search, and these were integrated using Boolean operators (AND/OR). Key search terms “adult,” “cerebral palsy” and “social participation” were included in the search, with variations of the terms used to expand the search. Previously published reviews relating to participants with CP were examined for additional search terms. Advice was sought from an expert subject librarian before carrying out the search. All search terms used are summarised in Table 1.

Table 1. Search terms.

Social participation	Cerebral palsy	Adult
Social/community engagement Social/community activities Social/community involvement Social/community participation Participation	Cerebral pals* CP ((Hemipleg* or diplegi* or quadriplegi* or unilateral*) ataxi*) ((Hemipleg* or diplegi* or quadriplegi* or unilateral*) spastic*) Little* disease	Adult* Grow* up Young adult Aged Middle-aged Older* Elder* Later in life Aging

Eligibility criteria

Studies published between 2011 and 2021 were considered eligible for this review to reflect the most recent research findings (i.e., within the last 10 years). Inclusion criteria were: adult participants who had CP (at least 50% of the sample), studies focused on or which included measures of social participation, studies that included empirical data, and were published in the English language. Studies which provided no data on people with CP, had a majority of participants aged under 18 years old, were discussion or review articles, or did not discuss social participation were excluded from the review.

Selection process

Database searches were conducted and results exported to reference management software, EndNote version 20 [30]. Following de-duplication, all remaining articles were imported to Rayyan, an online systematic review tool [31]. Hand-searching of reference lists was also conducted to identify additional articles. Two reviewers independently screened the title and abstract of each record according to the inclusion/exclusion criteria and met to resolve any conflicts. A third reviewer was available for advice if conflicts could not be resolved. After title and abstract screening, two reviewers also completed full text screening for the retained articles.

Data collection process

A standardised data extraction tool was created following guidance from the Joanna Briggs Institute Manual for Evidence Synthesis [32]. Data from the remaining 16 articles were extracted by two independent reviewers. Reviewers extracted data on author/s name and year of publication, country, aim(s), design, participants, methods/measures, findings, and definition of social participation. The final item, “definition of social participation” was not always made explicit by authors, in which case a decision was made based on the domains the research focussed on and measures used.

Quality assessment

The Mixed Methods Appraisal Tool [33] was used for quality appraisal of included studies. Two reviewers completed quality appraisal independently, one on all the included studies and the second reviewer on half of the included studies, selected at random. Following current guidance on the use of the MMAT [33], a brief description of the assessment of quality decision was included alongside appraisal questions.

Analysis

Due to the heterogeneity of included studies a meta-analytic approach was not feasible. A narrative synthesis was employed to answer the review’s research question. Popay et al. [34] describe narrative synthesis as a textual approach to summarising the findings of a systematic review, grouping together commonalities between papers to present the evidence as a narrative.

Results

The following section details the findings of the systematic search, characteristics of the included studies and quality appraisal of the studies. Findings in relation to the definitions of social participation employed and the three main themes identified in the narrative synthesis are then presented.

Systematic search results

A total of 345 records were identified from the systematic search: CINAHL (n = 77), PsycINFO (n = 45), PubMed (n = 61) and Web of Science (n = 155). Following de-duplication, all remaining articles (n = 247) were screened by title and abstract: 225 records were excluded, with 22 articles retained for full text review. A further six articles were excluded following full text review as there was either no clear discussion of social participation (n = 3) or the articles had inadequate numbers of adult participants with CP (n = 3). Of the eight articles identified from hand-searching reference lists, only one was included following full text screening. The selection process is outlined in Figure 1, PRISMA flowchart [35].

Figure 1. PRISMA flowchart.

t shows the PRISMA flowchart for the review. Title: Identification of studies via databases and registers. Section 1: Identification. Records identified from: Databases (CINAHL (77), PsycINFO (45), PubMed (61), Web of Science (155)) Combined (n = 338) Hand-searching reference lists (n = 8). Records removed before screening: duplicate records removed by endnote (n = 120), duplicate records removed by researcher (n = 24). Section 2: Screening. Records screened (n = 247), records excluded (n = 225). Reports sought for retrieval (n = 22), reports not retrieved (n = 0). Reports assessed for eligibility (n = 22), reports excluded: no discussion of social participation (n = 3), Inadequate number of adults with CP (n = 3). Section 3: Included. Studies included in review (n = 16).

Study characteristics

The characteristics of the 16 studies included in this review are presented in Table 2. Included studies followed both qualitative (n = 5) and quantitative (n = 10) study designs, with one [38] adopting a mixed methods approach. Numbers of participants in each study ranged from 7 to 335 individuals. One study [36] included individuals from various diagnostic groups, but was included as 50% of participants had CP, in line with the inclusion criteria of the review. Studies were conducted in eight countries with The Netherlands being the most common place of origin (n = 5, of which n = 4 related to one longitudinal study). The remaining studies were conducted in the USA (n = 2), Canada (n = 2), Australia (n = 3), Croatia (n = 1), Sweden (n = 1), Latvia (n = 1) and South Africa (n = 1).

Table 2. Characteristics of included studies.

Author(s) and country of origin	Aim(s)	Design	Participants	Methods	Findings	Definition of social participation
Bartolac, & Jokic [36] Croatia	To explore the everyday experience of social and occupational participation of persons with physical disabilities living in Croatia.	Qualitative	15 Adults (CP n = 7, SCI n = 3, MD n = 5), ages 20–65 years, 9 women, GMFCS not recorded.	Six semi-structured interviews on sources of stress, two focus groups.	Three themes emerged. First, occupational and social participation are described as a means for inclusion. Work and completing tasks/chores provides a sense of achievement, belonging, pride and purpose. Secondly, exclusion is seen as a consequence of occupational deprivation and social marginalization. Finally, enablers and barriers to meaningful participation were highlighted. Enablers: positive attitude and empowerment. Barriers: illness, low confidence, physical limitations, loss of spontaneity.	Social model definition.
Carroll et al. [37] United States	To explore how adults with CP experience aging and the impact of growing older on participation.	Qualitative	38 Adults with spastic CP, ages 18–65 (average 31.42 years), 63.2% female, GMFCS I–III	Semi-structured interviews	Themes included individual agency, family and social context, and sociocultural contexts in a life-course development context. The impact of parental expectations, apprehension of aging, inaccessible environments and lack of social supports were cited as barriers to social participation.	ICF definition.
Gannotti et al. [38] United States	To evaluate changes in gait, descriptive analysis of characteristics associated with gait and participation outcomes, and to highlight characteristics of paediatric rehab interventions aimed at improving gait in adults with CP.	Mixed methods	26 Adults with CP, ages 20–36 (average 25 years), 11 male, GMFCS I–IV.	Case studies, motion analysis, semi-structured interviews. Measures: Short Form Health Survey (SF-36) [39], Canadian Occupational Performance Measure [40]	No direct relationship found between gait and participation outcomes (work activities, social functioning (SF-36), role-emotional (SF-36), living arrangements.) Highlights importance of self-efficacy and determination in achieving success in participation domains.	ICF definition.
Gaskin et al. [24] Australia	To investigate how people with CP have successfully negotiated major challenges of adulthood, and to identify what factors may have contributed to successful outcomes and what supports were needed in achieving these outcomes.	Qualitative	23 Adults with CP, ages 18–47, 13 female, GMFCS I–IV	Semi-structured interviews	Four main themes emerged, with several subcategories, relating to positive and negative experiences in participants’ lives: Formative Experiences – determination; pathways for developing competence; parental expectations; sibling support; involvement in school life; skills for making friends; coping with bullying; experiences with healthcare. Present functioning – positive self-concept; social skills; physical fitness; ability to find a way. Present contexts – availability of support; expectations of involvement; accessible environments. Finding a way – drawing on knowledge and skills; gravitating towards those who are comfortable with difference; maintaining function; using natural and paid supports.	ICF definition.
Hanes et al. [41] Canada	Explore the lived experience of young people with CP with their physical, mental and emotional health in the context of a larger longitudinal study on brain function, physical and mental health, and well-being.	Qualitative	16 Adults with CP, ages 17–29 (average 26 years, SD = 3), 7 female, GMFCS I–V	Three focus groups	Four main themes emerged from focus groups: Health – physical and mental health and illness experience. Meaningful participation in work (major subject) and recreation, benefits of goal setting. Impact of the environment – family, intimate and peer relationships, accessibility. Identity for young people with CP.	Not defined, however, Aligns with ICF definition.
Imms et al. [42] Australia	Investigate the social outcomes of school leavers with CP and explore the determinants of desirable outcomes.	Quantitative, cross-sectional	90 Adults with CP aged 18–25, (average 22.4 years, SD = 2.2), 57% male, GMFCS I–V	Online/paper-based surveys. Measures of variables that may influence participation in life situations and life satisfaction.	Overall, young adults with CP had different social outcomes to the general population in participation domains of education, employment (32.6% CP employed, 75.8% general population), financial status, relationships, living arrangements (12.5% CP living away from parental home, 51.8% general population), community participation and life satisfaction. Most participants with CP (72.7%) reported that their health affects participation in work and were receiving disability support pensions. Young adults with CP reported high levels of emotional/informational support, tangible support, affectionate support and presence of positive social interactions, and a higher proportion were active members of a club or association compared with the general population (41.4% CP, 30% general population).	Not defined, however, aligns with ICF definition.
Jacobson et al. [27] Sweden	To describe social outcomes for young adults with CP and to explore associations of social outcomes with their classification levels within GMFCS, MACS and CFCS and intellectual disability.	Quantitative, cross-sectional	61 Adults with CP, ages 20–22 (average 21 years 2 months, SD = 8 months), 27 female, GMFCS I–V	Social outcomes examined through questions on living arrangements, occupation, personal finances, personal care and relationships. GMFCS [43], MACS [44], CFCS [45].	Participation largely defined by communication levels and ID. Fine motor function had some associations with participation in employment (p < 0.01–0.06) and finance (p < 0.001) while gross motor function (GMFCS) showed no significant associations with social outcomes. 20% of young adults had moved out of the parental home, 43% dependent on family support. Majority without ID (79%) were employed. All associations negative: increased disability, lower probability of achieving participation goals.	Not defined, however, aligns with ICF definition.
Jiang et al. [46] Australia	Compares the QOL of young adults with CP in Australia with able-bodied peers to explore the relationship between QOL and impairments, functioning, and social participation.	Quantitative, cross-sectional	335 Adults with CP (or proxies, 62% self-report), average 24.7 years (SD = 2.8), 51% male, GMFCS I–V	Social participation data obtained from questions on education, employment, living situation and marital status.	General population and self-report group had similar scores for QOL in social relationship and environment domains (p < 0.05), but lower scores in physical health, psychological well-being and role function (p < 0.001) Mobility/functional independence strongly associated with QOL related to physical health and employment but not to satisfaction with social relationships. Barthel index scores and social participation variables significantly linked to the social relationship domain score in simple linear regressions but not in the multivariate analysis. Employment status associated with each QOL domain except psychological wellbeing. Highlights the benefit of employment and employment-related support services on social participation.	Not defined, however, aligns with ICF definition.
Palisano et al. [43] Canada	To describe how young adults with CP experience life-course health development. To create key messages for paediatric health service providers to promote children’s capacities for future adult roles and healthy adult living.	Qualitative	23 Adults with CP, ages 25–33 years (average 28.2 years, SD = 2.4), 13 male, GMFCS I–V	Semi-structured interviews lasting 50–60min.	Four themes were identified from interviews. Personal life-course – unique nature of each developmental journey and constant change of healthy living potential for people with CP. Contexts of healthy living – relationships between person and circumstances of their lives (family, health care, community etc.), transactional as the individual can influence the context as much as the context influences the individual. Health development through everyday experiences – these shape the life-course and develop the individual capacity for healthy living and social participation. Healthy living as an adaptive process – adjusting to changing circumstances and learning from experience.	Not defined, however, takes a social model stance.
Rozkalne et al. [47] Latvia	To identify the level of participation in the context of the developmental transition from adolescence to adult life for young adults with CP and the factors that had an influence on participation.	Quantitative, cross-sectional	81 Adults with CP and normal/slightly decreased cognitive function, ages 16–21 years (average 18 years [IQR 20–17]), 41 male, GMFCS I–V	Rotterdam Transition Profile [48]	Overall, low levels of autonomy in all domains of participation. Participants were in phase 0 (no experience) in the domains of education and employment (21%), intimate relationships (56%) and sexuality (59%). Higher disability levels (measured by WHODAS) were associated with decreased participation. Age positively associated with participation in education and employment, finance, housing and sexuality. Low GMFCS negative associations with autonomy in social activities, sexuality and transportation. Only 9% of adults were autonomous in education and employment domains, 44% depending on other adults for money/allowance.	Not defined, however, aligns with ICF definition.
Schmidt et al. [49] The Netherlands	1 Describe the long-term development of autonomy in individuals with CP in several domains of participation and examine differences in development between individuals with high versus low levels of GMF. 2 Compare the proportions of individuals with CP who are autonomous in participation to typically developing peers.	Quantitative, cohort study, PERRIN combination of two 13-year follow up studies.	189 Individuals with CP without intellectual disability, ages 12–34 (average 21 years 11 months, SD = 4 years 11 months), 117 male, 72 female, GMFCS I–V.	Rotterdam Transition Profile [48]	90% of Individuals with CP reached autonomy in participation in adulthood (27 years). Transport – most autonomous from 13+. Leisure, finance, intimate relationships, employment – autonomous from 18 to 22+, Housing – autonomous from 27+. Those with lower gross motor function had lower odds of organising own transport and less experienced in sexual relationships. Social participation reliant on provision of transport from family members. GMFCS 3–5 autonomy higher in finances regardless of age (related to disability benefits in Netherlands).	Not defined, however, aligns with ICF definition.
Slaman et al. [50] The Netherlands	To evaluate the effect of a lifestyle intervention on fatigue, participation, QOL, GMF, motivation, self-efficacy and social support, and to explore mediating effects of physical behaviour and physical fitness.	Quantitative randomised controlled trial with intention to treat analysis	41 Adults with CP, ages 16–24 (average 20 years, SD = 3), 27 male, 30 female, GMFCS I–IV.	6-Month intervention of physical fitness training with counselling sessions. LIFE-H Questionnaire [51].	While the intervention saw an increase in family support for participation during intervention (difference = 5.38, p = 0.04), participants still experienced no change to physical limitations in achieving social participation.	ICF definition.
Van Gorp et al. [52] The Netherlands	To determine childhood predictors of participation in domestic life and interpersonal relationships of young adults with CP.	Quantitative, prospective cohort study, 13 year follow up of 9–16 cohort (PERRIN)	67 Adults with CP, ages 21–27 (average 24.6 years, SD = 1.6), 44 m 22,GMFCS I–V	Life-H Questionnaire [51], Vineland Adaptive Behaviour Scale (Vine-II) [53]	Childhood factors predict up to 90% of variation in participation in adulthood. Environmental and personal factors in childhood did not influence future participation. Participation in domestic life − 73% performed below adequate level for age, 66% experienced difficulty. Participation in interpersonal relationships − 64% performed below adequate for age, 33% experienced difficulty. Having ID or epilepsy influenced lower attendance and difficulty in participation in interpersonal relationships. Individuals with lower motor capacity, low manual ability and ID were at increased risk for low participation in domestic life and interpersonal relations in the future.	ICF definition.
Van Gorp et al. [54] The Netherlands	To determine the long-term course of difficulty in participation of individuals with CP without ID between ages 16–34 years of age.	Quantitative, prospective cohort study, 13 year follow up of two age cohorts (PERRIN 9–13 + 16–24)	151 Individuals with CP aged 16–34 (no mean stated), 63% male, 71% GMFCS I–IV	Life-H Questionnaire [51].	Difficulty in participation increased for individuals with CP in mid-late 20s. High average levels of participation but large proportions experience difficulty (41–95% young adults) and require assistance, particularly in domestic life, education and employment and recreation. Those with lower motor function experience more difficulty with 95% of individuals in GMFCS IV experiencing difficulty in one or more participation domains. However, this group continued to develop participation into early 20s, whereas those with higher function reached peak participation levels at younger age. Increasing difficulty for all in older age in 5/6 domains (not responsibilities). Highest levels of difficulty in GMFCS 4, oldest age bracket (23–34) and in housing, education and employment and recreation.	ICF definition.
Van Wely et al. [55] The Netherlands	To examine which teenage factors predict participation attendance, as well as difficulty and need for assistance with participation in domestic life and interpersonal relationships among adults with CP without ID.	Quantitative, prospective cohort study, 13 year follow up of PERRIN 16–24 cohort.	53 Adults with CP without intellectual disability, ages 24–29, (average 31.7 years, SD = 1.4), 28 m 25 f, GMFCS I–V	Vineland Adaptive Behaviour Scales [53], Life-H Questionnaire [51].	High average scores in participation in domestic life [Vine-II: 42.6 (SD 10.3) out of 48 points; Life-H: 8.3 (SD 1.4) out of 10 points] and interpersonal relationships [Vine-II: 73.3 (SD 4.0) out of 76 points; Life-H: 9.2 (SD 1.2) out of 10 points], but high number scored below reference norms for difficulty and need for assistance. Domestic life – low attendance in adulthood predicted by lower GMF and protective parenting at teenage (R^2 = 60.9%). More difficulty/need for assistance predicted by lower GMF, rigid personality and special education (R^2= 64.6%). Interpersonal relationships – low attendance predicted by rejective parenting and less daily support in teenage (R^2= 40.6%). More difficulty/need for assistance predicted by being male, socially avoidant and rejective parenting (R^2 = 49.6%). Environmental and personal factors + GMF (domestic only) + teenage participation in communication and daily living skills predict participation in domestic life and/or interpersonal relationships in adulthood. Parenting style and personality type significant predictors of participation in adulthood.	ICF definition.
Veerbeek et al. [56] South Africa	To determine changes in level of accomplishment and satisfaction in daily activities and social participation, and functional mobility in adults with CP more than 25 years after selective dorsal rhizotomy (SDR). To compare these outcomes of adults with CP to a matched group of typically developing adults.	Quantitative, cross-sectional, 9 year follow up.	26 Adults with CP and spastic diplegia, ages 34–41 (median age 35.8 years), 16 male, GMFCS I–III	Life-H Questionnaire [51], Functional Mobility Scale (walking performance) [57], GMFCS [43]	Overall, most adults independent and satisfied with their participation in daily life. Strong correlations found between accomplishment and functional mobility (0.85–0.89), and satisfaction and functional mobility (0.73–0.79). Slight change in housing dependency in follow up but all satisfied – happy to have/pay someone else to assist with domestic tasks. Some small differences in daily activities and social roles to typically developing peers, adults with CP more dependent in mobility and recreation. Interestingly, higher levels of employment in CP than in developing countries (70% of CP cohort employed cf 29–54% in Western countries) – perhaps highlighting impact of disability benefits. No change in functional mobility in 9 years is remarkable as most studies report decline in early-mid 20s.	Not defined but aligns with ICF model.

Abbreviations: CP: cerebral palsy; SCI: spinal cord injury; MD: muscular dystrophy; GMFCS: Gross Motor Function Classification System; ICF: International Classification of Functioning; SD: standard deviation; MACS: Manual Ability Classification System; CFCS: Communication Function Classification System; ID: intellectual disability; QOL: quality of life; IQR: interquartile range; WHODAS: World Health Organisation Disability Assessment Scale; PERRIN: Pediatric Rehabilitation Research in the Netherlands; GMF: gross motor function.

Quality appraisal

Quality assessment using the MMAT [33] showed that 11 studies addressed all criteria [24,27,36,37,41,42,46,50,52,54,58], whilst four studies failed to adequately address non-response bias [47,49,55,56]. The mixed methods study [38] failed to provide sufficient detail to assess quality and did not address inconsistencies between qualitative and quantitative findings. Responses to MMAT questions are summarised in Table 3, with a brief explanation of quality decisions.

Table 3. Quality assessment (MMAT).

First author & year	Q1	Q2	Q3	Q4	Q5	Explanation
Bartolac (2019) [36]	Y	Y	Y	Y	Y	All criteria clearly addressed
Carroll (2021) [37]	Y	Y	Y	Y	Y	All criteria clearly addressed
Gannotti (2013) [38]	Y	Y	Y	N	CT	Divergences and inconsistencies of quantitative and qualitative results were not addressed; insufficient detail was provided to assess if each component of the study adhered to quality criteria.
Gaskin (2021) [24]	Y	Y	Y	Y	Y	All criteria clearly addressed
Hanes (2019) [41]	Y	Y	Y	Y	Y	All criteria clearly addressed
Imms (2021) [42]	Y	Y	Y	Y	Y	All criteria clearly addressed
Jacobson (2019) [27]	Y	Y	Y	Y	Y	All criteria clearly addressed
Jiang (2016) [46]	Y	Y	Y	Y	Y	All criteria clearly addressed
Palisano (2020) [58]	Y	Y	Y	Y	Y	All criteria clearly addressed
Rozkalne (2019) [47]	Y	Y	Y	CT	Y	Details of nonresponse bias are not discussed
Schmidt (2020) [49]	Y	Y	Y	CT	Y	Details of nonresponse bias are not discussed
Slaman (2015) [50]	Y	Y	Y	Y	Y	All criteria clearly addressed
Van Gorp (2018) [54]	Y	Y	Y	Y	Y	All criteria clearly addressed
Van Gorp (2020) [52]	Y	Y	Y	Y	Y	All criteria clearly addressed
Van Wely (2020) [55]	Y	Y	Y	CT	Y	Details of nonresponse bias are not discussed
Veerbeek (2021) [56]	Y	Y	Y	CT	Y	Details of nonresponse bias are not discussed

Y: YES – the paper clearly addresses the question; N: NO – the paper has clearly shown a negative answer to the question; CT: cannot tell – no statement is included which allows an answer to be drawn for the question.

Definitions of social participation

Of the 16 included studies, only seven [36–38,50,52,54,55] provided definitions of social participation. Six of these did so using the ICF model, either considering participation and activities together or reporting on social participation specifically. One study [36] defined social participation through the social model. Thus, nine of the included studies [24,27,41–43,46,47,49,56] did not define social participation but were included in the review as the measures used (quantitative studies) or questions asked (qualitative) aligned with the ICF (n = 8) [24,27,41,42,46,47,49,56] or the social model (n = 1) [43].

Results from the narrative synthesis

Following narrative synthesis, three themes were identified: (1) Impact of Home & Work Life on Social Participation, (2) Considerations Across the Lifespan, and (3) Factors Limiting Autonomy.

Impact of home & work life on social participation

The first theme considered the impact of home and work environments on an individual’s social participation. The impact of the home environment was explored through the concepts of family support and parenting styles. Three of the five included qualitative papers [24,37,41] found that family expectations both enabled and hindered social participation, as some individuals reported their parents encouraged them to be involved, while others were held back from building independence by protective parents. However, van Wely et al. [55] found that the home environment in childhood (including a rejective parenting style) did not impact adult participation, whereas Van Gorp et al. [54] found that environment in the teenage years negatively impacted social participation within the same cohort. Hanes et al. [41] also noted the importance of family support to enable social participation for provision of transportation. Similarly, Jacobson et al. [27] reported that 43% of young adults with CP were dependent on family support, both financially and for transport, with only 20% living independently by the age of 22 years.

Family support and parenting style in childhood was also found to impact individual beliefs about capacity to work. Gaskin et al. [24] found that over-protective parenting in childhood may result in an apprehension towards employment due to a fear of negative employer attitudes, while supportive parenting instilled a firm belief in the individual’s employability. The benefits of employment were framed by Bartolac and Jokic [36] in the context of occupational participation, which provided an avenue to meaningful participation and an improved sense of wellbeing. Jiang et al. [46] reported a link between quality of life and social participation in the domain of employment, while Hanes et al. [41] noted that work was a key element of meaningful social participation discussed in their qualitative interviews. Although a substantial increase in school completion in recent years by individuals with CP in Australia was found by Imms et al. [42], a clear lack of improved participation outcomes in the domain of employment were evident, with employment levels of adults with CP (32.6%) being much lower than those of the general population (75.8%).

Considerations across the lifespan

Four of the five included qualitative papers [24,37,41,58] discussed the importance of considering the provision of appropriate care and support across the lifespan. Three papers [37,41,58] framed this issue in a Life Course analysis perspective [59] and considered the timeliness of interventions for individuals with CP to be crucial for ensuring optimum opportunity for social participation. One of the key findings by Gaskin et al. [24] was the importance of experiences in the formative years, with many successful outcomes in adulthood being attributed to the provision of adequate support and physical interventions in childhood. Palisano et al. [58] considered healthy living, that is physical, mental, and social health, to be a key component of social participation, and noted that the potential for healthy living is constantly changing as individuals with CP age. In addition, Carroll et al. [37] found that social participation was hindered by the physical effects of accelerated ageing, and by the apprehension of ageing, with participants stating that they had limited knowledge on what to expect in later life with CP. Consideration of a lifespan approach was also explored through the individual experience of ageing and the difficulties involved in this process. In their longitudinal study, Van Gorp et al. [54] found that difficulty in social participation increased in domains of education and employment, recreation, community life, housing, and interpersonal relationships for adults with CP in their mid to late 20s, and consequently achievement of desired levels of social participation were low. Both Van Gorp et al. [52] and Van Wely et al. [55] reported that individuals may benefit from early intervention in a rehabilitative setting to develop participation skills, with Van Gorp et al. [52] finding that CP-related childhood factors such as intellectual disability, epilepsy, or lower levels of mobility were shown to predict up to 90% of variation in participation in adulthood.

The impact of physical interventions received during childhood on social participation in adulthood were considered in four papers [24,38,50,56]. This included discussion of medical procedures such as Selective Dorsal Rhizotomy (SDR), orthopaedic surgery and physical therapy, as well as structured exercise programmes. In their longitudinal study, Gannotti et al. [38] found that the majority of participants (n = 22/26, GMFCS I–IV) who had undergone orthopaedic surgery as a child maintained or improved their gross motor function and gait abilities in adulthood and had levels of social participation similar to the general population. Similarly, Veerbeek et al. [56] noted that higher levels of accomplishment and satisfaction in social participation were evident in individuals with higher levels of gross motor function and improved mobility following an early medical intervention in childhood. Notably however, in qualitative interviews, Gaskin et al. [24] found that while participants recognised the positive impact of childhood physical interventions, they also felt that their social participation in childhood had been negatively impacted by the need to take time off to recover, which impacted not only their education but also their personal relationships. The importance of promoting fitness in childhood and adolescence to improve social participation outcomes in adulthood were discussed by both Slaman et al. [50] and Gannotti et al. [38]. Participation in a fitness training intervention by Slaman et al. [50] was shown to improve quality of life and family support, and decrease fatigue and pain in adults with CP, while Gannotti et al. [38] also demonstrated a relationship between increased social participation in adulthood and fitness and physical therapy in childhood/adolescence.

Factors limiting autonomy

Several papers [24,27,36,38,41,46,47,49,52,54,58] included discussion of factors which impact an individual’s social participation by limiting their autonomy. These included physical limitations [27,36,47,52,54], societal expectations [24,27,46,47,49,52], and environmental barriers [24,36,38,41,58]. Five included papers discussed the impact of an individual’s motor skills on their social participation [27,36,47,52,54]. Rozkalne et al. [47] noted that physical limitations may lead to lower levels of autonomy for adults with CP in all participation domains (education and employment, finance, housing, leisure, intimate relationships, sexuality, and transportation). Further to this, Jacobson et al. [27] and Bartolac and Jokic [36], found that increased physical disability and lower levels of gross motor function, were linked to a lower probability of the individual with CP achieving their desired level of social participation. In a qualitative study, participants expressed that feelings of low self-confidence, the additional effort required and the fear of injuring themselves due to decreased physical ability were barriers to meaningful participation [36]. In addition, Van Gorp et al. [52,54] showed that individuals with limited mobility experienced more difficulty in participation than those with greater motor function.

Van Gorp et al. [54] suggested that increasing difficulty in achieving social participation may be explained by an increase in societal expectations as the individual enters adulthood, whereby adults are expected to be more autonomous in how they access society, leisure activities, housing, and employment. These expectations were alluded to in several studies included in this review [24,27,46,47,49,52]. In their quantitative study, Schmidt et al. [49] considered autonomy as a key element of participation as it involves self-determination and independence. Jacobson et al. [27] noted that a lack of autonomy in participation in younger years, particularly in activities of daily living, was an obstacle to successful transition to adulthood, while Rozkalne et al. [47] and Schmidt et al. [49] found that autonomy in participation increased with age. However, as Bartolac and Jokic [36] highlighted through their social model approach to participation, environmental barriers and inaccessible physical spaces impact an individual’s experience of independence and autonomy, and a lack of choice around how they wish to participate in society. These barriers to meaningful social participation for adults with CP were highlighted in several papers [24,36,38,41,58]. Gaskin et al. [24], Palisano et al. [58] and Gannotti et al. [38] echoed these findings highlighting how addressing modifiable environmental barriers could allow individuals to reach their desired level of social participation, in addition to improving individual mental health and employment opportunities.

Discussion

This review has examined the current evidence base for social participation in adults with CP. Findings from the 16 studies included in the review demonstrate that social participation is often not a clearly defined concept. Despite this, studies included in this review were found to be of good methodological quality following quality appraisal using the MMAT [33], demonstrating that while the quantity of conducted studies in this field is low, the quality of that research is high. However, the review demonstrated that although social participation is an important aspect of overall health and wellbeing of adults with CP, it is often hindered by reduced opportunities for building independence and social participation in childhood. The results showed that this is due to protective parenting styles, lack of support in finding employment, the consequences of accelerated ageing and a lack of a lifespan approach to care, and a lack of autonomy caused by physical impairments and inaccessible physical environments.

By not providing a clear definition of social participation, it is possible that studies relating to the concept may have been missed when searching the literature. While participation outcomes such as employment, education and mobility have been reported on since the 1970s [60], the measures used to assess these domains have been inconsistent. Participation has also been a key theme in research and legislation relating to disabled people since the publication of the UN CRPD [28], however, the value of research on participation may be lost if the frequency of attendance is measured rather than the quality of involvement. For these reasons, social participation requires a clear definition so that research focuses on “meaningful involvement”, as called for in the UN CRPD as a basic human right for disabled people. In a review on outcomes of adolescents and adults with CP, conducted by Frisch and Msall [61], six commonly used measures of social participation were highlighted, only two of which emerged in the current review (Life-H [51]; Short Form 36 Health Survey [39]). This inconsistent use of measures and the complexity of factors examined may relate to the difficulties involved in defining social participation [16,17]. This lack of a clear, universal definition of social participation, combined with variability in measures used to capture the concept, has resulted in the possibility that papers of relevance may not have been identified in this systematic search.

The results from this review showed how an individual’s childhood environment, and the support they received from family and professionals affects how they experience social participation later in life [6,24,47,49,54,55]. In addition to studies included in this review, evidence shows that parenting styles can have either a positive or negative impact on the individual in adulthood. Wiegerink et al. [62] found that a rejective parenting style resulted in poor participation in relationships in adulthood, while Reedman et al. [63] found that participation in leisure activities in childhood was more successful when parents were supportive of the individual’s autonomy. This heralds a need for support to be provided for parents in early years, to highlight the impact of parenting style on social participation in later life. Also highlighted in this review was the impact that family support may have on future social participation such as fostering self-belief in the individual’s capabilities, as well as a positive attitude towards employment. It is widely known that disabled people may face barriers when seeking employment such as discrimination, lack of access to required training and education, and inaccessible workplaces [64,65]. While the disability employment gap remains a key concern in many countries [66–68], Vornholt et al. [69] suggest that disabled people are increasingly being recognised as an underutilised demographic as the current population of workers age. Employment has been found to have a positive impact on disabled individuals, including opportunities for socialisation, a sense of purpose, and a feeling of participation [70,71], and thus better pathways to employment opportunities are necessary to ensure individuals can participate to their desired extent.

The need for the adoption of a life course perspective to healthcare was described by several papers in the review [37,41,58]. Interventions and support provided at the correct time, when the individual is ready (rather than when a service can provide it), could be more beneficial in the longer term. In a study on adult beliefs about the impact of surgery they received in childhood, Gannotti et al. [72] found that 58% of participants believed that orthopaedic surgery had a positive impact on their functioning as adults, and related this to being happy, independent and being able to build meaningful relationships and participate in activities. However, participants who did not agree with this stated that there is a need for more information to be provided to families on the long-term outcomes and the decline in physical health that comes with ageing, once again drawing attention to the need for a lifespan approach to healthcare and provision of information. Palisano et al. [58] noted that the timing of interventions, provision of assistive technologies and changes to the physical environment should correspond with the individual’s readiness to achieve desired participation outcomes, as everyone develops at a different rate. In addition to this, providing support for parents to ensure a supportive childhood home environment, as well as thinking long-term about development goals, could help the individual to prepare for change as they age. Finally, while a discussion of transition from paediatric to adult healthcare falls outside the remit of this review, many studies have highlighted the negative impact that this process may have on social outcomes and participation in adulthood if it is not planned adequately with sufficient information and support provided for both the individual and their families in childhood [8,73–76]. Transition must therefore be considered from early in the individual’s life for a lifespan approach to healthcare to be adopted.

This review highlighted the lack of autonomy and limited confidence many adults with CP experience due to reduced mobility and how social participation is impacted by apprehension about how physical abilities may change over time. Previous studies have highlighted the importance of autonomy and self-efficacy in the development of independence for disabled individuals, and that independence is a key factor contributing to how, and to what extent, individuals participate in society [77–79]. Bartolac and Jokic [36] discussed the impact of personal attributes as enablers of social participation, and how a positive attitude towards capabilities could improve the frequency and success of participation. Imms et al. [42] reported that 48.7% of participants felt that a lack of confidence impacted their participation in education. Participation, and the promotion of independence to achieve social participation, should be the end goal for rehabilitation of individuals with CP, as well as key considerations for health development [80,81]. This independence has been alluded to through studies on autonomy [47,49,54] as well as its interrelationship with self-efficacy [50,82,83]. Van der Slot et al. [82] suggest that by improving self-efficacy, quality of life and social participation could be improved for individuals with CP. This could in turn assist the individual in navigating expectations of adulthood and improve confidence in attending and participating in social events.

Finally, a critical factor limiting the opportunity for autonomy in social participation for adults with CP is the lack of accessible physical environments. Article 9 of the UN Convention on the Rights of Persons with Disabilities (UN CRPD, [28,p.8]) highlights the right of all persons with disabilities to “live independently and participate fully in all aspects of life,” calling for access to the physical environment, transport, and information to be provided on an equal basis with others. Despite this, accessibility remains a key issue in achieving social participation goals for people with CP. In a review on the role of the physical environment on social participation for people with limited mobility, Bigonnesse et al. [73] found that the environment can be both an enabler and a barrier to social participation. Safe neighbourhoods, open public spaces such as parks, and the proximity of homes to facilities such as shops and banks enabled individuals to participate in society more easily. However, narrow pavements with poorly designed curb cuts [84], lack of accessible parking spaces [85] and insufficient provision of public transport [86] remain significant barriers to social participation. Hanes et al. [56] reported that while participants believed they had the physical ability and the desire to participate in society, they were hindered from doing so due to the inaccessibility of the environment. This was particularly true for wheelchair users who recalled issues accessing events in areas with steep inclines, as well as the unreliability and lack of elevators.

Changes to UK legislation such as the Disability Discrimination Act 1995/2005 (DDA) [87] sought to reduce marginalisation and improve participation in society for all disabled people, through accessible employment, education, and transport. However, Bell and Heitmueller [88] argue that due to a lack of awareness and financial support, no positive impact has been evident on employment rates of disabled people in the UK since the publication of the DDA [87], and Sayce and Boardman [89] highlight that disabled people are still experiencing inequality and discrimination in access to the environment. Similarly, in the USA, Carroll et al. [37] found that while the enactment of the Americans with Disabilities Act (1990/2008) [90] saw the provision of more accessible public transport, this was an unreliable and frustrating service, as the frequency and number of services available were insufficient to meet the needs of disabled people. Without appropriate provision of services and recognition of the accessibility of those services, social participation for disabled people, including adults with CP, cannot be achieved.

Strengths and limitations

This review provides a useful addition to the knowledge-base on adults with CP, and has highlighted a lack of clarity in definition of participation, social or otherwise, in several papers. We contend that social participation is a distinct concept, but acknowledge that term is used interchangeably with participation and thus must often be interpreted, leading to a risk of non-consensus among readers. This review also identifies sparsity in the literature regarding social participation in adults with CP and, given the small number of studies deemed eligible for inclusion and the exclusion of studies that were not published in English, conclusions should be interpreted with caution. Key strengths of this review are that two reviewers completed data extraction for included papers, thus reducing the risk of bias. An expert subject librarian was consulted when developing the search strategy to ensure the most appropriate terms were included. Reviewers also followed guidance from the Joanna Briggs Institute [32] when developing a data extraction table, used the latest version of a standardised tool for quality appraisal [33], and followed PRISMA guidelines when constructing the review [35].

Future research

The review highlights the need for future longitudinal studies: of the ten included quantitative studies, only four were longitudinal [49,52,54,55] and all related to different elements of the same study. Longitudinal data on individuals with CP can provide an insight into factors that contribute to social participation and allows consideration of how these may change over time. With a clear definition of social participation, and the creation of an appropriate measure for the concept, more robust data could be collected on how social participation is impacted by the experience of CP. Previous reviews with this population have addressed the wider context of the lives of individuals with CP, with a focus on the physical impact of CP [91] and exercise interventions [12,92,93], as well as pain [94,95], and access to healthcare [7,11]. Future reviews should consider specific factors that hinder or promote social participation for adults with CP, in order to provide families and healthcare professionals with information on how to best support individuals across the lifespan.

Conclusions

Social participation is important for all individuals as an essential part of healthy living and wellbeing. This review sought to examine the current evidence on social participation for adults with CP and has shown that there is a lack of consensus on a definition. Clarity and consistency on a definition and measurement of the concept would facilitate a deeper understanding of social participation across the lifespan and enable fusrther research into when and how best to support social participation in people with CP. Aside from this, the barriers that still exist in society which hinder individuals from achieving their desired levels of social participation, such as physically inaccessible public spaces, are not new issues. The ongoing effort to increase awareness in public and political domains must be further substantiated with action and financial support, in order to fully implement the legislation that is already in place to improve equality for people with disabilities in access to the environment, public life, work and education. Further research should focus on the longitudinal development of social participation for individuals with CP, aiming to develop autonomy and independence from childhood, as well as developing appropriate family support, to benefit the individual in adulthood.

Disclosure statement

No potential conflict of interest was reported by the author(s).

Additional information

Funding

This review forms part of the research student’s PhD at Queen’s University Belfast and is funded by the Department for the Economy, Northern Ireland.