Child and family health-related quality of life and participation outcomes and goals after acquired brain injury: a cross-sectional survey

ABSTRACT

Objective

To explore longer-term health-related quality of life (HRQoL) and participation outcomes and goals of children and young people (CYP) with acquired brain injuries (ABI) and their families in a region of the UK and the impact of the COVID-19 pandemic.

Methods

Cross-sectional survey of (5-18 CYP) and their parent-carers 12–43 months following ABI. Included measures of HRQoL, participation, family function and parental wellbeing and demographic and free text questions.

Results

Ninety-five responses (30% response) were received. 67% of CYP were at risk of impaired HRQoL, 72% had severely impaired participation. 53% of parent-carers reported reduced HRQoL and family functioning, 37% of parent-carers screened positive for anxiety/depression. Relationships exist between CYP participation and HRQoL and parental HRQoL and family functioning. Goals were overwhelmingly activity and participation focused. Participants described the global impact of an ABI on the CYP and family as well as the additional impact of the COVID-19 pandemic on CYP and family wellbeing.

Conclusion

ABI significantly impacts CYP participation and both CYP and parent-carer wellbeing in the long-term, potentially further impacted by the COVID-19 pandemic. Rehabilitation interventions should address both participation and the psychological wellbeing of CYP with ABI and their parent-carers.

KEYWORDS:

• Acquired brain injury
• children and young people
• goals
• health-related quality of life
• participation
• outcomes
• survey

Introduction

Acquired Brain Injury (ABI) occurs because of traumatic (fall, road-traffic collision) or non-traumatic (stroke, infection, hypoxia, brain tumor) causes. It is the leading cause of morbidity and mortality in children and young people (CYP) globally, with traumatic brain injury incidence rates of 180–825 per 100,00 and 82.5 per 100,000 for non-traumatic causes (1). In the UK, an estimated 40,000 CYP sustain an ABI every year (2). ABI’s can lead to an array of complex physical, cognitive, social, emotional, and behavioral impairments, which can result in lifelong disability. The consequences of an ABI may continue to become evident months or years after the event, with many issues under-recognized or mis-interpreted (3,4,5). As childhood development progresses and educational and social demands increase through adolescence, these impairments can continue to impose a significant impact on a CYP’s physical and psychological development, health-related quality of life (HRQoL), educational achievement and social participation through into adulthood (3,6).

Predicting outcomes and monitoring long-term consequences poses a challenge for clinicians as childhood development and recovery of function is complex and unique to each CYP. Many factors influence recovery such as: injury characteristics, age, premorbid abilities, family functioning, environmental factors, and variability in rehabilitation service provision (3,7). The International Classification of Functioning, Disability and Health (ICF) can be used to describe the impact of a health condition by mapping the needs, goals and influential factors, to the four domains (body functions and structures, activity and participation, personal and environmental factors) and relationships between factors examined (1,7). Participation in life situations is an essential component of child development, building toward increasing independence through adolescence into adulthood. It is seen as the ultimate outcome of healthcare services being associated with increased HRQoL, social and educational competence and long-term wellbeing (8,9).

The impact of a CYP sustaining an ABI can extend to the whole family with substantial caregiver burden, distress, anxiety, stresses and reduced quality of life, wellbeing and family functioning (10–13). Previous outcome and interview studies found that family impact is increased where there are unmet health and social care needs, and that the impact continues in 40–45% of families after the first year (3,14,15). However, further research is required to explore the longer-term impact and influential factors in a UK context.

A scoping review by Keetley et al. identified poor outcomes and extensive needs of CYP-ABI and their families (3). Many had unmet or unrecognized needs up to 12 years post injury, which transcend ICF, health, social care and educational domains across the care pathway. Whilst there is an understandably large focus on return to school, being central to the development and education of CYP, research regarding reintegration and participation in community life, particularly activities and sports, is lacking. Furthermore, the voice of CYP is limited in the literature meaning little is known regarding the CYP’s own opinions on their outcomes and needs (3,16).

The literature recommends long-term specialist follow-up and integrated care pathways; however, this is sparsely provided in the UK, with many CYP not receiving any specialist or community-based therapy or medical review. Access to this population is further complicated by the lack of any national registries, making investigating the long-term outcomes and needs of UK CYP-ABI challenging (3).

Globally, there is a drive to promote brain health and development, improve care pathways and support recovery, wellbeing and participation for people living with neurological disorders and for rehabilitation practice to be participation-focused (5,11). In order to develop UK services to achieve this, further research is required to understand the outcomes and needs of CYP-ABI and their families within the UK service context (17). In this study, part of a larger mixed methods study, we aimed to describe the HRQoL and participation outcomes of CYP-ABI and their families who received acute inpatient care/rehabilitation at one UK regional specialist center. Additionally, we aimed to identify goals important to CYP-ABI and their parents and also explore the impact of the COVID-19 pandemic and national lockdowns on this population. Studies conducted in Australia investigating the impact of the pandemic on vulnerable children, including those with disabilities, and their families found a significant negative impact on health and wellbeing of both CYP and parents as well as reduced access to usual services and other support structures (13,14). Understanding the impact of the pandemic on UK CYP-ABI and their families is essential in supporting families, planning restoration of services and promoting full participation in society.

Methods

Study design

This study consisted of a cross-sectional survey of community dwelling CYP-ABI and their parent-carers who were at least 12 months since discharge from hospital, as part of a mixed methods study.

Participants and setting

CYP-ABI and their parent-carers were recruited through a pediatric neuro-rehabilitation service based in a regional specialist (tertiary) pediatric neurosciences center serving a population of approximately two million CYP. In the UK CYP-ABI remain in the care of children’s services until they are aged 18 and complete their education when they then transition to adult services. Therefore, we included parent-carers of a school-aged (5–18 years) CYP (at the time of the survey) diagnosed with an ABI of any cause and resident in the UK. CYP-ABI were eligible if they received acute inpatient care/rehabilitation at the regional pediatric specialist center within the last five years and were one or more years post injury. The exclusion criteria included CYP deceased, ongoing safeguarding arrangements, insufficient command of English language to complete survey and carers not having parental responsibility.

Due to the exploratory nature of this study, purposive sampling and a pragmatic approach to calculating sample size was employed (19). The eligible population was estimated to be 150 and a sample size of 92 CYP/parent-carers was calculated (95% confidence interval, 5% margin of error).

The study opened during a national COVID-19 pandemic lockdown in March 2021 and recruitment continued until December 2021. Ethical approval was gained from the UK Health Research Authority (REC-20/EM/0258). Initial contact with potential participants was made by a member of the patient’s usual care team face-to-face, by telephone, or letter and invited to participate by completing the survey online, via telephone, or by post.

The survey was administered using JISC ‘Online Surveys’ with separate versions for parent-carers (including proxy-report) and CYP age groups (5–7 years, 8–12 years, 13–18 years). Patient and public involvement (PPI) representatives were consulted on survey design and pre-tested the final version. It included demographic questions and standardized measures of CYP HRQoL and participation and parental wellbeing, HRQoL and family impact.

Both CYP and parent-carers were asked to state their three most important goals and free text boxes were provided to gather any other information. Parent-carers proxy-reported for CYP unable to self-report due to cognitive impairment. Additional demographic, admission, and injury information (including measure of severity) was collected from the service database by a clinical team member following survey completion.

Measures were selected based on validity and reliability in this population, systematic review recommendations, prior clinical and research use, PPI consultation regarding ease of completion and participant burden, and child- and proxy-report version availability (20,21).

The included measures were (see Table 1 for further details):

• Child and Adolescent Scale of Participation (CASP) to measure CYP participation (22)

• Pediatric Quality of Life Inventory (PedsQL™ 4.0 Generic Core Scales SF15) to measure CYP HRQoL (23)

• Pediatric Quality of Life Inventory Family Impact Module (PedsQL™ FIM) to assess family impact – parent-carer HRQoL and family functioning (23)

• Patient Health Questionnaire for Anxiety and Depression (PHQ-4) to screen parent-carers for anxiety and depression (24)

Table 1. Details of selected outcome measures.

Outcome	Tool	Completion	Scoring
CYP Participation	Child and Adolescent Scale of Participation (CASP)	Self-report (11+) Parent-carer-proxy report	20 items in 4 sections (home participation, community participation, school participation and home and community living activities). 4-point Likert scale is used to rate participation (4 = age expected (full participation), 3 = somewhat limited, 2 =very limited and 1 = unable, not applicable items = 0). Total score divided by the applicable number of items and multiplied by 100 to conform to a 100-point scale. Higher scores indicate higher levels of participation. A score of less than 92 is deemed to represent severely impaired participation (22).
CYP HRQoL	Pediatric Quality of Life Inventory (PedsQL™ 4.0 Generic Core Scales SF15)	Self-report (5–7, 8–12, 13–18 versions) Parent-carer-proxy report	15 items in 4 sections (physical functioning, emotional functioning, social functioning and school functioning). 5-point Likert scale is used to score each item (0 = never, 1 = almost never, 2 = sometimes, 3 = often, 4 = almost always). Scores transformed into a 0–100 scale – total score being the mean of all items. Higher scores indicate better HRQoL. A score of 72.05 or less, being greater than 1 standard deviation (SD) from a UK healthy population mean (83.89) is deemed to represent risk of impaired HRQoL for UK CYP (19).
Family Impact	Pediatric Quality of Life Inventory Family Impact Module (PedsQL™ FIM)	Parent-carer report	36 items in 8 sections (physical functioning, emotional functioning, social functioning, cognitive functioning, communication, worry, daily activities, family relationships). Scoring as per PedsQL^TM Higher score indicates higher HRQoL/family functioning. Score of 1 SD or more (59.6) from the healthy population mean (73.2) is considered to represent risk of impaired family HRQoL (25).
Parental wellbeing (anxiety and depression screener)	Patient Health Questionnaire-4 (PHQ-4)	Parent-carer report	4 items: 2 for symptoms of anxiety (Generalized Anxiety Disorder 2-item (GAD-2), 2 for symptoms of depression (Patient Health Questionnaire-2 (PHQ-2). 4-point Likert scale (0 = not at all, 1 = several days, 2 = more than half the days, 3 = nearly every day) with the sum of all scores being the total (0–12). Total score - > 6 (or > 3 on subscales) = ‘yellow flag’ for presence of a mild-moderate anxiety or depression disorder Total score > 9 (or > 5 on subscales) = ‘red flag’ for moderate-severe anxiety or depression disorder (24).

Survey data submitted via the JISC Online Surveys website was collated and analyzed in Stata (Version 14, 2015). Participant responses were anonymized for analysis. Demographic characteristics and outcomes were analyzed descriptively. Wilcoxon Rank Sum and Chi² tests compared responders to non-responders. Relationships between outcome measures and demographic characteristics were analyzed using Spearman’s rho with Bonferroni correction.

Participant goals were mapped to the ICF-Child and Youth version (ICF-CY). Each goal was independently coded and assigned an alphanumeric code using chapter, first and second level descriptor codes. Coding was independently reviewed by a second reviewer and agreement reached where opinion differed. Multiple codes were used where the goal was multifaceted. Each goal was then assigned to the appropriate ICF domain and analyzed descriptively.

Free text boxes were provided for participants to share comments regarding the impact of the ABI on the CYP and family and also the impact of the COVID-19 pandemic. Responses were analyzed thematically using an inductive approach (23).

Results

From a possible 170 families, completed surveys were received from 51 families a response rate of 30% (Figure 1). A total of 95 CYP and/or their parent-carers responded. There was no missing data within the completed surveys, however two parent-carer responses were not completed where the CYP had completed their version and five CYP responses were not completed where parent-carers had responded.

Figure 1. Study recruitment flowchart.

The median age of CYP at the time of survey completion was 13 years (IQR 8–16) with 36 being male. The median time since injury was 2 years 11 months (IQR 1yr11mo-3yr7mo) with 45% having had a brain tumor. The sample included respondents from all five counties within the region, index of multiple deprivation (IMD) areas and a wide range of ethnicities (Table 2).

Table 2. Respondent characteristics.

Table of respondent characteristic			n =
No. of parent respondents		Total	49
		Mothers	42
		Fathers	7
CYP Demographics
Sex, n (%)		Female	15 (29%)
		Male	36 (71%)
Current Age, median (IQR)			13yr (8yr-16yr) (4–12)
Diagnosis, n (%)		Brain Tumour	23 (45%)
		Trauma	12 (24%)
		Stroke/Bleed	6 (11%)
		Infection/Inflammation	5 (10%)
		Other	5 (10%)
Severity (KOSCHI completed by clinical team at initial assessment), median (IQR)			3b (3a-4b)
Time since injury, years, median (IQR)			2yr11mo (1yr11mo-3yr7mo)
Index of Multiple Deprivation (quintiles), n (%)		5 (10/9) (highest)	11 (22%)
		4 (8/7)	9 (18%)
		3 (6/5)	9 (18%)
		2 (4/3)	12 (24%)
		1 (2/1) (lowest)	10 (20%)
Ethnicity	Any other White background		2 (4%)
Asian or any other Asian background			7 (14%)
Black, African, Caribbean or Black British			2 (4%)
White – British-English, Welsh, Scottish, Northern Irish			39 (76%)
Prefer not to say/Not stated			1 (2%)

Outcome measure scores are presented in Table 3. The median PedsQL score was 64.17 (IQR 43.33–78.33, range 18.75–93.33) with 67% of CYP being at risk of impaired HRQoL. CYP also reported impaired social participation with 72% reporting severely impaired social participation (CASP median 80.63, (IQR 61,25–94.44, range 35–100)). More than half of parent-carers (53%) reported reduced parental HRQoL and family functioning (PedsQL-FIM median 56.25, (IQR 39.58–74.31, range 16.67–100)). The PHQ-4 total scores showed 37% of parent-carers screened positive for anxiety, depression or both with total scores of 6 or more). Subscale analysis revealed 29% of parent-carers screened positive for mild-moderate anxiety and 16% for moderate-severe anxiety (GAD-2 scores) and 27% of parent-carers screened positive for mild-moderate depression and 10% for moderate-severe depression (PHQ-2 scores).

Table 3. Outcome measure scores.

Outcome measure		Median (IQR)	Mean (SD)	Freq (%)
CYP, n=46
CASP	Total Score	80.63 (61.25–94.44)	77.45 (22.55)
	Home participation	91.67 (75.00–95.83)
	Neighbourhood/community participation	75.00 (50.00–93.25)
	School participation	85.00 (70.00–100)
	Home and community living activities	75 (41.67–90.00)
PedsQL^TM	Total Score	64.17 (43.33–78.33)	60.9 (21.06)
	Physical health subscale	60.00 (30.00–80.00)
	Psychosocial subscale	65.00 (50–82.50)
Parents/carers, n = 49
PedsQL^TM FIM	Total Score	56.25 (39.58–74.31)	58.75 (22.63)
	Parental HRQoL subscale	55.00 (43.75–75.00)
	Family functioning subscale	65.63 (46.88–90.63)
PHQ-4	Total Score	4 (2–18)
	Yellow Flag			11 (22%)
	Red Flag			7 (15%)
	GAD-2	2 (15–18)
	Yellow Flag			14 (29%)
	Red Flag			8 (16%)
	PHQ-2	1 (0–3)
	Yellow Flag			13 (27%)
	Red Flag			5 (10%)

There was a strong positive relationship between CASP and PedsQL^TM scores (rho 0.7137 p<0.0001) suggesting higher participation was related to higher HRQoL. Higher levels of CYP HRQoL and participation were also related to higher parental HRQoL and family functioning (PedsQL^TM and PedsQL^TM-FIM rho 0.6393 p=0.0001; CASP and PedsQL^TM-FIM rho 0.5981 p=0.0008). Higher levels of parental anxiety and depression were related to lower CYP participation (CASP) and parental HRQoL and family functioning (PedsQL^TM-FIM) (PHQ-4 and CASP rho −0.4962 p=0.0258; PHQ-4 and PedsQL^TM-FIM rho −0.7557 p<0.0001). No statistically significant relationships were found between the demographic variables themselves (diagnosis, age, time since injury, KOSCHI score, IMD area), and only a weak positive relationship between time since injury and PedsQL^TM and PedsQL^TM-FIM scores (see Appendix 1, SupplementaryTable 1).

Goals

A total of 111 CYP goals and 146 parent-carer goals were mapped to the ICF-CY. Goals spanned three of the four ICF domains with the majority mapped to the Activity and Participation domain, 93% CYP, 83% parent-carers (see Table 4). ‘Recreation and leisure’ goals were the top priority for both parent-carers and CYP, with driving (which includes riding a bike), moving around (e.g., running or swimming), being the next highest priority for CYP and school education and informal social relationships for parent-carers (Figure 2). Comparing goals to different CYP age groups, whilst recreation and leisure goals were the top priority for all ages, there was a difference in priorities below this. CYP aged 5–10 years reported driving (riding bike) and moving around (running/swimming) as important goals; 11–15-year-olds reported school education, informal social relationships, driving (bike) and moving around and 16–18 year-olds reported driving (car/bike) and higher education, acquiring a job, and carrying out daily routines.

Figure 2. Top goals for CYP-ABI and parents/carers.

Table 4. Goals mapped to ICF-CY chapters.

	CYP		Parents/carers
Body Functions	freq	%	freq	%
Chapter 1 – Mental functions	4	3.60	12	8.22
Chapter 2 – Sensory functions and pain	1	0.90	1	0.68
Chapter 4 – Functions of the cardiovascular, hematological, immunological and respiratory systems	1	0.90	1	0.68
Chapter 5 – Functions of the digestive, metabolic, endocrine systems			3	2.05
Chapter 7 – Neuromusculoskeletal and movement-related functions			2	1.37
Activity and Participation
Chapter 1 – Learning and applying knowledge	2	1.80	4	2.74
Chapter 2 – General tasks and demands	4	3.60	4	2.74
Chapter 3 - Communication	1	0.90	1	0.68
Chapter 4 - Mobility	29	26.13	11	7.53
Chapter 5 – Self-care			13	8.90
Chapter 6 – Domestic life	2	1.80	6	4.11
Chapter 7 – Interpersonal Interactions and Relationships	5	4.50	14	9.59
Chapter 8 – Major life areas	18	16.22	27	18.49
Chapter 9 – Community, social and civic life	42	37.84	41	28.08
Environmental Factors
Chapter 1 – Products and technology	2	1.80	1	0.68
Chapter 3 – Support and relationships			1	0.68
Chapter 5 – Services, systems and policies			4	2.74
Total number of goals	111		146
Body Functions goals	6	5	19	13
Activity and Participation goals	103	93	121	83
Environmental Factors goals	2	2	6	4

Free text responses

Impact of the ABI on CYP and family

CYP and parents detailed the global impact of the ABI on both the CYP and family. By far the largest concern for parents was the impact on their CYP’s emotional and mental health, with worries about how the ABI had affected confidence, self-esteem, increased feelings of frustration and isolation, with several families struggling to access mental health support. CYP also recognized the impact on their emotions and mental health.

She has felt very isolated and alone going through it. We didn’t manage to get any mental health support for her until this year (Parent)

Sometimes I don’t want to let people know what it’s like living with it (CYP)

I get angry very easily I get scared crossing roads I forget (CYP)

Other concerns were impairment-related and the impact of these on education, getting the right support in place to support learning, and difficulties participating in sports and activities with friends.

Stress of trying to get the right support in school … .he struggles to retain information … He’s very behind with his year group. And it’s affecting his mental health. (Parent)

It’s hard being competitive with my peers. It’s frustrating being last in a lot of activities (CYP)

The impact on the whole family was particularly evident with several parents detailing how their lives had changed forever with theirs, and their other children’s wellbeing negatively affected. Parents also recognized the impact on the whole family’s ability to participate in social activities.

It’s difficult to put into words how our lives have been affected. I stopped living the day it happened. (Parent)

They [siblings] continue to struggle with the situation and have responded by keeping aloof and distant from X and refuse to discuss the difficulties she and we all face as a family. (Parent)

In every single way, we used to do lots together as a family but are no longer able to do this, friends have been lost and social circles disappeared. (Parent)

Additionally, several parents and CYP spoke of the lack of understanding regarding ABI, particularly where the injury is invisible.

Others don’t understand as she looks fine physically but they can’t see her brain and they don’t know the struggles she faces daily. (Parent)

Some teachers really don’t understand my new behaviour and I think I get punished when I don’t mean to behave as I have. I react first but can’t always help it I can’t think quickly (CYP)

Impact of the pandemic

CYP and parents reported positives and negatives of how the pandemic had impacted them. Whilst some participants reported that it had brought their family closer together and there were less social pressures or stress or anxiety related to having to go to school, the majority of families reported negative effects.

Increased isolation, reduced social interaction and lack of activities were major concerns along with difficult accessing home education and health and rehabilitation services, increased stress and worry for parents.

Like most children, X wasn’t exercising enough, which wasn’t great for keeping him strong and coordinated. (Parent)

I had to learn to home school which I found difficult with minimal teacher support. (CYP)

We had a disaster trying to accomplish any home-schooling. The on-line lessons and set work just didn’t work for us at all and in the end I gave up (after lots of trying!), so she had no formal education. (Parent)

The family being separated during hospital stays. Very isolating for the parent who is in hospital. (Parent)

I lost all my NHS treatment and we have only just started it up, just physio now. (CYP)

Discussion

The aim of this study was to describe the long-term HRQoL and participation outcomes of CYP-ABI and their families, their goals, their views on the impact of an ABI and also how the COVID-19 pandemic had impacted them. This cross-sectional survey elicited 95 CYP and parent-carer voices representing 51 families. They report the substantial ongoing impact of an ABI on HRQoL, participation, family functioning and wellbeing one to four years after injury. Additionally, we gained insight into the impact the COVID-19 pandemic has had on these families. HRQoL and participation are important health outcomes, assessing CYP’s wellbeing, mental health, physical and social functioning, however this study highlights the importance of also assessing parental wellbeing and family functioning and wellbeing (20,26).

Impact on CYP

Through self-report, the voices of the CYP have been heard regarding their health and wellbeing, telling us that all is not well for a substantial proportion of this sample. CYP HRQoL was reduced with 67% scoring below the cutoff values of 72.05 which is comparable to or worse than reported in other studies (27–29). CYP scored similarly across physical and psychosocial subscales demonstrating the global impact of an ABI on their HRQoL.

Reduced participation levels were reported across all domains, with 72% of CYP experiencing severe restrictions, in line with the findings of other international studies (27,30–33). The greatest restrictions were in the community participation and home and community activities domains, which include participation in social, play or leisure activities with friends, structured events and activities (clubs/sports), community mobility and communication and daily life activities (managing money, daily routines, work). Participation in these types of activities may be difficult for CYP-ABI due to functional impairments but other barriers may be impacting e.g., environmental factors – physical environment, support and relationships as well as pandemic restrictions.

The significant positive relationship between CYP HRQoL and participation was expected and adds to the evidence base (27). The weak positive relationship between HRQoL and time since injury suggests that there is some improvement over time, but as this was not a longitudinal study this cannot be substantiated.

Through the free text responses, we gain insight into how the CYP feel about their life after ABI and their parents’ concerns. Though only brief, the impact on emotional and mental health, resultant isolation and difficulties accessing support are evident, as is the importance of supporting CYP to participate to maintain their wellbeing (4,14,35–36). These findings warrant further exploration.

Impact on family

The findings show the substantial impact of an ABI on the family, with more than half of parent-carers reporting significantly impaired HRQoL and poor family functioning. In this study, PedsQL-FIM median scores were below those in comparable studies and mean scores were substantially below healthy population norms (12,37,38). Furthermore, PHQ-4 scores revealed over a third of parent-carers screened positive for the presence of an anxiety and/or depression disorder, substantially higher than the general population, further demonstrating the lasting impact of a CYP’s injury on parent-carers (39). These results suggest that the parent-carers and families in this study continue to feel the impact of their CYP’s ABI long after the event, with ongoing disruption to family functioning and parental HRQoL and wellbeing being affected. Furthermore, the significant associations found between parental wellbeing and family functioning and CYP participation and HRQoL outcomes point to the importance of these relationships in CYP long-term recovery. This is supported by evidence from previous research in CYP with ABI in the first-year post injury (12,40). The free text responses contributed greater insight to this issue, particularly parental concerns regarding the impact of the ABI on the wellbeing of the whole family, including siblings.

With parents and families being the main caregivers and source of support for a CYP with ABI, it is imperative to recognize and address the detrimental impact of the ABI on the whole family, given that parental mental health and family functioning are important predictors for the functioning and wellbeing of the CYP (4,9,34,41–43).

This study was unable to determine the factors affecting the outcomes observed or the nature of these causal relationships from these results. Further exploration within the UK context is warranted to investigate influential factors, and barriers and enablers to family-centered care and to inform the development of interventions targeted at addressing the needs of the whole family unit (34,44).

Prioritising goals

Goal-setting is fundamental to rehabilitation. In recent years, there has been increasing emphasis on the prioritization of goals that are meaningful to CYP and their families and participation-focused (21,45,46). Activity and participation goals were clearly prioritized by both CYP and parent-carers in this study, in line with previous studies which also found that families prioritized activity and participation goals during rehabilitation such as recreation and leisure, education, mobility, selfcare and communication (16,47).

Families reported what is important to them in terms of their wellbeing – recreation and leisure (e.g., play, sports, fitness, hobbies, visiting the cinema, sightseeing, tourism).

Other important goals were driving (bike/car), moving around (e.g., running, swimming), school education and informal social relationships, with more focus on independence skills in older CYP. These and recreation and leisure cover a wide range of activities, reflected in the CASP domains where CYP scored worst. This demonstrates their importance to CYP-ABI and their families, and the need for continued monitoring and interventions targeting individual needs and goals. Further exploration of CYP and family goals and needs is required to explore influential factors and how goals change over time, particularly into adolescence.

Impact of pandemic

As this study was conducted during the COVID-19 pandemic, the additional impact of this on the wellbeing of CYP and families cannot be discounted and may account for the lower scores reported here. Certainly, the further social isolation and loss of support services reported by participants in the free text responses only adds to the burden families of a CYP with ABI experience, which supports international evidence regarding the vulnerability of CYP with disabilities during the pandemic (17,18,48,49,50). Ongoing clinical assessment is vital to identify those who are in crisis or at high risk of poor outcomes. Furthermore, clinicians need to be mindful of the impact the pandemic has had on the already complex lives of CYP and families with additional needs and that the real consequences are still being uncovered (50,51).

Strengths and limitations

This study, delivered with methodological rigor, has facilitated the voices of 95 CYP and parent-carers to be heard, allowing them to highlight their considerable ongoing needs. The findings add to the evidence base, particularly regarding CYP self-reported outcomes together with parent-carer reported outcomes and evidence of the relationships between these.

However, accessing and recruiting this population was challenging. We achieved a 30% response as a result of supporting families to respond by offering multiple modes of completion. We are reminded that this is higher than the general survey response rate in pediatric populations (15–25%) and, bearing in mind the study was conducted during the challenges of the COVID-19 pandemic, we are indebted to the families for their participation. We must however recognize there is still a potential risk of bias, limiting generalizability, with many CYP and parent-carer voices from this seldom-heard population remaining unheard (52). Additionally, mainly mothers responded, meaning fathers’ voices are under-represented. Every effort was made to reduce the burden of the survey on families, however, 28 families who initially consented verbally to completing the survey failed to do so, potentially suggesting the burden remained too high. CYP with brain tumors and their families responded in greater numbers than those with TBI or other non-traumatic causes compared to previous population data from the same region, potentially due to the center specializing in childhood brain tumor research and a familiarity with clinical research. In addition, there was a greater (although not statistically significant) representation from more severely affected CYP-ABI, which may be related to differences in engagement with services for those with greater need and may also have contributed to the lower scores reported (see Appendix 1, SupplementaryTable 2).

The challenges in accessing and recruitment led to limitations in design. To reduce participant burden, the survey was cross-sectional, thereby only capturing information at a single time point, with no assessment of pre-existing outcome levels. Parent-carers were the first point of contact via the acute service and therefore contact information may not have been up to date or they may have decided not to participate without consulting their CYP. The heterogeneity of the sample, whilst representative of CYP neurorehabilitation services in the UK and research in this population, meant there were limited numbers in each diagnostic group, which limited sub analyses.

Conclusion

This novel study has explored outcomes for both CYP-ABI and their parent-carers one to four years post ABI during a global pandemic and adds UK findings to the international evidence base, complementing understanding of the evolving burden of this disease. CYP and parent-carers report a substantial impact on their HRQoL, participation, parental HRQoL and wellbeing and family functioning both in their outcome scores and free-text responses. Activity and participation goals were prioritized by both CYP and parent-carers asserting their importance. The potential impact of the pandemic on these findings cannot be ignored and needs further exploration. The relationships between participation, HRQoL, family functioning and parental wellbeing exist and require further investigation. While the nature of these causal relationships remains unclear, the need for family-centered care and multidisciplinary and multimodal interventions targeting outcomes for both the CYP and parent-carer is clear. Holistic long-term care is vital to effectively monitor and address existing and emerging needs as CYP and families navigate their rehabilitation journey and re-build their lives. To do this effectively, further research is required to explore the associations between outcomes and influential factors, including barriers and enablers and develop interventions aimed at addressing family needs across the life course.

Abbreviations

ABI	=	Acquired Brain Injury
CASP	=	Child and Adolescent Scale of Participation
CYP	=	Children and young people
HRQoL	=	Health-related quality of life
IQR	=	Interquartile range
NHS	=	National Health Service
PedsQLTM	=	Pediatric Quality of Life Inventory
PedsQLTM-FIM	=	Pediatric Quality of Life Inventory – Family Impact Module
PHQ-4	=	Patient Health Questionnaire − 4
SD	=	Standard Deviation
UK	=	United Kingdom

Acknowledgments

This study is funded by the NIHR Applied Research Collaboration East Midlands (ARC EM). The views expressed are those of the author(s) and not necessarily those of the NIHR or the Department of Health and Social Care.

Our thanks goes to the children and young people and their parents/carers that took part in this study, our patient and public involvement families, the clinical team for their assistance with recruitment and to Lal Russell for being second reviewer for the goal analysis.

Disclosure statement

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

Supplementary material

Supplemental data for this article can be accessed online at https://doi.org/10.1080/02699052.2024.2309244

Correction Statement

This article has been republished with minor changes. These changes do not impact the academic content of the article.

Additional information

Funding

The work was supported by the National Institute for Health and Care Research Applied Research Collaboration East Midlands.