Exploring participation after paediatric acquired brain injury

This study aimed to explore the levels of participation in a UK sample of children and young people (CYP) with an acquired brain injury (ABI) at home, school and in the community through a cross-sectional study. The Child and Family Follow-Up Survey was distributed to parents/carers of 134 CYP with ABI (CYP-ABI) who received neuro-rehabilitation from 2014 to 2016. Access and recruitment were problematic resulting in a low response rate (4%). Widespread restrictions in participation were reported by four of the five respondents with community-structured events/activities and educational activities being the most restricted. Factors impacting on participation were cognitive-based and movement skills, family stress and lack of support/encouragement in the community. Study results provide information pertaining to the feasibility of undertaking research with this population while also highlighting the restrictions to participation experienced by CYP-ABI more than two years after injury.


Introduction
While global prevalence is unclear, annually in the United Kingdom, an estimated 40,000 children and young people (CYP) sustain an acquired brain injury (ABI) (NHS England, 2013). A childhood ABI can lead to a complex array of physical, cognitive, social, emotional and behavioural

Participants and procedures
A cross-sectional survey was identified as an appropriate method, to access information from a larger target population (Bowling, 2014). The methodological and procedural components; the recruitment processes and procedures and appropriateness of the selected outcome measure (Child and Family Follow-Up Survey (CFFS)) were also evaluated (Arain et al., 2010).
A purposive sampling strategy was used to recruit participants from four centres in England. Local collaborators identified eligible participants from existing service user databases using predetermined eligibility criteria (stated below). Postal invitation letters and participant information sheets, specific for parents/carers and for CYP, were sent, inviting participants to access and complete the online survey. Further telephone reminders were completed by the local collaborators four weeks after the postal invitations were sent.

Inclusion criteria
Parents/carers of CYP-ABI aged 5-18 who received inpatient neuro-rehabilitation at one of the identified four centres between October 2014 and October 2016. Sufficient command of the English language to understand the study literature, consent and complete the survey. CYP aged 11-18 associated with the participating parent/carer who consented to complete the CYP section of the survey.

Exclusion criteria
Parents/carers of CYP under 5 or over 18 (outcome measure requirements). Insufficient command of the English language to complete the survey. Parents/carers and CYP unable to provide assent or consent to participation. CYP under age of 11 years (outcome measure requirement).

Ethics approval
Ethical and governance approvals were gained from the National Research Ethics Committee and four participating National Health Service sites (REF: 17/WM/0163). Vital to any clinical ethical approval is that all principles of ethical research have been met (Emanuel et al., 2000). In this research study, practical examples include compliance with the General Data Protection Regulations 2018, data confidentiality and protection and risk-benefit equipoise. The research team carefully considered the online survey approach's potential to allow frank and honest responses while recognizing the need for parental guidance where a child completed their sections, given the potentially sensitive nature of the issues being explored. Participants were provided with contact details for the research team and their local clinicians should any concerns arise.

Survey
Bristol Online Survey (BOS) was used to create an online survey using demographic questions, consent and the CFFS. Permission was given by the CFFS's author (Professor Gary Bedell, PhD) for this and some minor alterations to the wording (American to English) for local context and UK spelling. Content and face validity were reviewed by the research team.
A review of the literature and the participation outcomes used in previous studies (Chien et al., 2014;De Kloet et al., 2015) identified the CFFS as an appropriate outcome measure for this feasibility work. Originally developed to monitor outcomes and needs of CYP-ABI, the CFFS was developed from the ICF and quantitative and qualitative research regarding the participation of CYP with a range of disabilities (Bedell, 2011). The CFFS is a parent-reported standardized measure, validated in North America and Australia, with good test-retest reliability (0.67-0.94), internal consistency (0.91-0.98) and evidence of criterion validity (Galvin et al., 2010). Normative data are not currently available, but results can be descriptively interpreted to understand patterns of participation (Galvin et al., 2010;McDougall et al., 2013). Included within the CFFS are three quantitative scales; the Child and Adolescent Scale of Participation (CASP), Child and Adolescent Factors Inventory (CAFI) and Child and Adolescent Scale of Environment (CASE), and openended questions giving opportunity for further information to be shared.
CASP: Participation levels of CYP-ABI in activities at home, school and the community are scored for 20 items using a Likert scale (1-4) or not applicable. Item scores are added together and divided by the maximum score for each applicable item giving a total score ranging from 0 to 100. Higher scores indicate CYP are participating at age expected levels. Parent and CYP aged 11þ versions are available and important to ensure both perspectives are captured (McDougall et al., 2013). The CASP-Youth version (CASP-Y) has been validated in Canadian English-speaking 11-17-year-olds (McDougall et al., 2013). It is considered a reliable and valid measure as strong internal structure validity and internal consistency (Cronbach's a ¼ 0.87 (youth) and 0.95 (parent)) is reported for both versions and intra-class correlation (ICC) moderate agreement (ICC ¼ 0.63, 95% confidence interval ¼ 0.41-0.75) between the two versions (Adolfsson et al., 2011;Bedell and Coster, 2008;Chien et al., 2014;McConachie et al., 2006).
CAFI: Parent-reported scale that measures the impact of child-related impairment factors on participation across 15 items.
CASE: Parent-reported scale that assesses environmental factors as facilitating or hindering participation across 18 items.
Both CAFI and CASE are scored on a scale of 1-3 or not applicable, total scores can be calculated for each measure, higher scores indicating significant impairment or environmental problems impacting on participation.
The survey also included 12 open-ended questions which provided the respondent opportunity to offer free text responses and provide more detail. These questions included the impact of their health condition on participation, equipment or strategies used to support participation, current service provision for their CYP and family; satisfaction with provision and suggestions for improving services.

Data analysis
Completed survey results were downloaded from BOS and reviewed in Microsoft Excel format. Participants completed the survey anonymously, however, where names were mentioned in the free text boxes these were anonymized. STATA (Version 15, 2015) was used to analyse quantitative data. Descriptive analysis was conducted for the demographic information, CASP, CASP-Y, CAFI and CASE data.
The responses to open-ended question were analysed using the thematic analytical approach by Braun and Clarke (2006). This data-driven approach allowed for emergent themes to be identified through a process of (1) reading for familiarity; (2) line-by-line coding; and (3) reading, connecting and aggregating the code into themes (emergent) (Maguire and Delahunt, 2017).

Respondents
In total, 134 (centre 1 n ¼ 60, centre 2 n ¼ 17, centre 3 n ¼ 17 and centre 4 n ¼ 40) invitations were sent. Only five responses were received giving a response rate of 4%. All five parent respondents were mothers. Three of the participants were over 11 years old, and all 3 consented to complete the CASP-Y. The characteristics of the participants are displayed in Table 1.

Participation (CASP/CASP-Y)
Parent-reported total scores ranged from 70 to 100 (median ¼ 82.50). Age-appropriate participation in all items was reported for 1 CYP, however, restrictions in 19/20 items were reported for 4 CYP (Table 2). Structured events/activities and educational activities were the most restricted (n ¼ 4 somewhat or very restricted). Further restrictions were reported in communication, social, play and leisure activities with friends, moving around neighbourhood, household activities and using transport.
Three CYP completed the CASP-Y, with one CYP reporting no participation restrictions. Two CYP participants reported restrictions in 18 of 20 items (Table 2). While there was general agreement in the restricted items between parents and CYP, there were differences in terms of the    degree of restriction. One CYP's total score is higher than the parent's score (83.82 vs. 71.05), and the other CYP's score lower (66.25 vs. 70.00).

Child-related difficulties (CAFI)
One CYP had no parent-reported problems in any areas. Significant impairments were reported by four parents with total scores ranging from 44.44 to 71.11. Cognitive-based skills, behaviour management, movement, strength and energy levels, speech and sensory problems were the highest reported impairments, with only hearing reported as unaffected by all.

Environmental difficulties (CASE)
Environmental barriers were reported for four of the five participants. Family stress, lack of support/encouragement and services in the community were reported as barriers to participation.

Open-ended questions findings
Four of five participants responded to the open-ended questions with 14 different codes being identified. Two main themes emerged: cognition and communication difficulties and environmental factors. The latter included barriers and facilitators to participation and CYP, parental and school responsiveness to adaptation. Participation restrictions due to communication difficulties were reported in the measurable and open-ended questions by the CYP respondents and their parents, indicating a significant problem for both. One CYP stated 'My speech gets in the way a bit but I try and talk to my friends and they always listen and now I've started talking again me and my friends are getting along much better', while another stated 'Seizures as they make me tired, fatigue, concentration. I sometimes struggle to communicate what I want to say, no word finding'.
Parents and CYP reported strategies used to support and improve participation, particularly at school. Open-ended responses demonstrated families' reliance upon school responsiveness to adapt for the child's altered needs post-ABI and implement support strategies. These included teaching assistant support, altered timetables and working environments. One parent reported 'We try to keep [her] in a more quiet environment at school, she works in small groups rather than a full class, she has regular ''brain breaks,'' she has a chill-out area to go when feeling frustrated'.
Parents were also asked about other services, assistance and information they felt they needed to support participation. Parents identified a lack of emotional and peer support for the CYP-ABI, siblings and parents as a barrier, three providing detailed responses about how they felt they, their CYP and family needed help to learn to accept, develop coping strategies and discuss problems that arise. One parent stated 'support and coping strategies, support groups. Offer support to families to help them learn to accept and cope including siblings once discharged [home]. We were offered nothing and have struggled daily with the stresses'.

Discussion
Participation is vital in all spheres of development for CYP and is an important rehabilitation outcome (Bedell and Coster, 2008;Chien et al., 2014;Galvin et al., 2010). The introduction of the ICF has led to increased focus on participation as an outcome and the development of several outcome measures (Chien et al., 2014). This study aimed to explore the levels of participation in a sample of UK CYP-ABI at home, school and in the community after discharge from inpatient rehabilitation using a multi-centre cross-sectional survey. While recruitment to this study via postal invitation was low, the online data collection method and survey was effective for those participants who completed it. Important knowledge has been gained with participants' voices being enabled to be heard and similar factors impacting participation identified, consistent with previous studies (Bedell and Dumas, 2004;Galvin et al., 2010;Wells et al., 2009). Participation in structured community activities and social, play and leisure activities rely on a high degree of skill socially, cognitively and physically and the ability to adapt quickly to changing situations and responses from others (Galvin et al, 2010). Social interaction is fundamental to child development and disruption at any stage of the lifespan can have implications for mental health, QoL, academic and career achievement (Anderson et al., 2013). Although relatively invisible, the impact of cognitive and communication impairments are common after ABI, impact heavily on participation and are a major source of stress and burden for CYP and families, as demonstrated in this small sample (Turkstra et al., 2015). Increased awareness of ongoing cognitive and communication difficulties is vital to ensure effective support and assistance for CYP and their families (Galvin et al., 2010;Turkstra et al., 2015).
The environmental factors component of the ICF represents external influences on function, physical, social and attitudinal (Ciccia and Threats, 2015). Family stress, lack of support/encouragement and services in the community were reported by parents as environmental factors impacting on participation. Parental stress and burden are known to be directly related to functional outcome after youth traumatic brain injury with sibling reaction/stress also playing an important role (Aitken et al., 2009;Ciccia and Threats, 2015). While there are positive developments within wider society to address participation of people with disabilities, there are still many barriers to achieving full societal participation following childhood ABI (Brittain and Beacom, 2016). A family-centred ethos to longterm CYP-ABI rehabilitation is critical in recognizing and addressing the impact on the whole family and the influence of family stress on the CYPs recovery (Aitken et al., 2009;Cole et al., 2009).

Limitations
This study has provided novel insights pertaining to participation of CYP-ABI. While we recognize that the findings are not generalizable, they do indicate that there may be outstanding needs and issues for CYP-ABI and their parents that require further exploration, support and intervention. However, this study does have a number of limitations that must be recognized relating to access to participants and recruitment/survey responses.
Firstly, the CYP-ABI population were difficult to identify and access. Currently, in the United Kingdom, there is no established CYP-ABI registry or national patient and public involvement (PPI) forum. Therefore, this study accessed potential participants via the CYP-ABI's local hospital through the clinical ABI services database. However, this approach was limited as it was dependent upon local hospitals maintaining the quality and completeness of these databases (such as up-to-date contact details). As such, some potential participants may not have been invited due to changes in contact details.
Secondly, the postal invitation recruitment method proved unsuccessful as it yielded a very low response rate. Therefore, it must be recognized that the views/experiences identified in the findings of this study, may not transferable to the national heterogeneous cohort. Despite attempts to improve responses, with a protocol amendment for telephone prompts, this did not result in an increase in recruitment. While other recruitment methods were explored when designing this study, such as telephone invitations or recruitment in clinics, postal invitations were deemed the least intrusive for families and feasible in the time and resources available.
Collectively, the design of the study was considered at length and a parent and child were involved in this process alongside feedback from other local service users. However, in relation to addressing these limitations in future research, more extensive and geographically wider PPI is required to establish effective approaches to recruit participants (Hunt et al., 2015) as well as strategies for optimizing responses to surveys. The reality of typical 21st-century family life is busy balancing multiple demands, with this population having the added complexity of supporting a CYP-ABI; this may have affected the response rate (Aitken et al., 2009;Bailey et al., 2014;Thompson et al., 2015;Wharton, 2012). Therefore, a range of methods for reporting should also be offered (such as postal, telephone, face to face or online), as this may accommodate the potentially diverse and changing demands that families have and enhance opportunities for research participation (Pulham et al., 2019).

Conclusion
This study aimed to explore the levels of participation in CYP-ABI. While the response rate was low, these findings, though not generalizable, are important and informative. Particularly the qualitative responses have provided rich and novel insights into the phenomena. Participation is an important goal of long-term rehabilitation/support for CYP-ABI and clinicians should seek to address factors that affect it. To optimize participation for CYP-ABI throughout their lifespan, awareness needs to be raised. The challenges for researchers of engaging with, accessing and recruiting CYP and their parents for research should not be a prohibitive barrier to work in this field. As researchers, we need to seek appropriate funding, increase preparatory dialogue with CYP-ABI and their parents to delimit such potential barriers in order to increase their involvement in research. Establishing a national network for CYP-ABI and their families should be considered to provide family peer support and support future research through PPI engagement. Further research aimed at comprehensively understanding acceptable and feasible research methods with this population is essential to ensure future research is effective in reaching and engaging CYP-ABI and their families. Additionally, international comparison is required to establish the generalizability/transferability of the phenomena. While it is necessary to investigate participation levels with a larger population, it is apparent that engagement of CYP-ABI and their families in research itself must also be addressed.