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Quality of life in young adults with cerebral palsy
Accepted Manuscript Quality of life in young adults with Cerebral Palsy Benran Jiang, PhD, Janet Walstab, BSc, Susan M. Reid, PhD, MClinEpi, BAppSc, Elise Davis, PhD, Dinah Reddihough, MD, BSc, FRACP, FAFRM PII:
S1936-6574(16)30045-0
DOI:
10.1016/j.dhjo.2016.04.006
Reference:
DHJO 487
To appear in:
Disability and Health Journal
Received Date: 21 September 2015 Revised Date:
15 April 2016
Accepted Date: 18 April 2016
Please cite this article as: Jiang B, Walstab J, Reid SM, Davis E, Reddihough D, Quality of life in young adults with Cerebral Palsy, Disability and Health Journal (2016), doi: 10.1016/j.dhjo.2016.04.006. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Title Page Quality of life in young adults with Cerebral Palsy Benran Jiang PhD.a,b, Janet Walstab BSc b,c, Susan M. Reid PhD, MClinEpi, BAppSc b,c, Elise Davis PhD d, Dinah Reddihough MD, BSc, FRACP, FAFRM a,b,c a
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Developmental Medicine, The Royal Children's Hospital, 50 Flemington Road, Parkville, Victoria, 3052, Australia b Department of Paediatrics, University of Melbourne, 50 Flemington Road, Parkville Victoria, 3010, Australia c Developmental Disability & Rehabilitation Research, Murdoch Childrens Research Institute, 50 Flemington Road, Parkville, Victoria, 3052, Australia d The Jack Brockhoff Child Health and Wellbeing Program, Centre for Health Equity, The Melbourne School of Population & Global Health, 207 Bouverie St, The University of Melbourne, Victoria, 3010, Australia. Corresponding author Dr Elise Davis The Jack Brockhoff Child Health and Wellbeing Program, Centre for Health Equity, The Melbourne School of Population & Global Health, Level 5, 207 Bouverie St, The University of Melbourne, VIC 3010, Australia. T: +61 3 8344 0921, E: [email protected]
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Disclosures We acknowledge the Victorian Medical Insurance Agency and the Victorian Department of Health who fund the Victorian Cerebral Palsy Register. Infrastructure support was provided by the Victorian Government's Operational Infrastructure Support Program. There are no conflicts of interest to declare.
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This work has been presented at the American Academy of Cerebral Palsy and Developmental Medicine, Phoenix, USA, September, 2009.
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Acknowledgements We would like to thank the young adults with CP and their families for participating in this study. We would also like to acknowledge Anna Lanigan for her support in finding research participants.
Points of Interest 1) Young adults with CP reported lower QOL than a normative general population sample on all domains except social relationships and environmental context. 2) Mobility and functional independence were strongly associated with QOL related to physical health and employment status, but not to satisfaction with social relationships. 3) Young adults with severe CP can have good psychosocial well-being regardless of their disability.
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Abstract word count: 250 Manuscript word count: 4000 Number of references: 46 Number of figures/tables: 4
ACCEPTED MANUSCRIPT Quality of life in young adults with Cerebral Palsy Abstract
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Background: Little is known about the quality of life (QOL) of young adults with cerebral palsy. Objective/Hypothesis: This cross-sectional analysis compares the QOL of a cohort of young Australian adults with CP with a cohort of able-bodied peers to explore the relationship between QOL and impairments, functioning, and social participation. Methods: Young adults identified from the Victorian Cerebral Palsy Register were invited to complete a survey about QOL, gross motor function, independence in self-care, and social participation. QOL was assessed with the Quality of Life Instrument for Young Adults (YAQOL). A general population sample of young North American adults, who had completed the YAQOL was selected for comparison. Results: Surveys and consent forms were completed by 335 young adults or their proxies, an overall participation rate of 63% of those located. The mean age of the study participants was 24.7 [s.d=2.8] years; 51% were male and 49% female. Two hundred and seven (62%) of the 335 participants self-reported their QOL. When compared with the general population sample, self-reporting participants had similar QOL scores for the social relationship and environmental context domains (p>0.05), while QOL scores were lower for the physical health, psychological well-being, and role function domains (p<0.001). There was no association between psychological well-being and variables related to body structure and gross motor function in young adults with CP. Conclusions: Contrary to the assumption that young adults with severe CP have low psychosocial well-being, it is apparent that these individuals can have good psychosocial well-being regardless of their disability.
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Keywords: Quality of Life, Young Adults, Cerebral Palsy, Well-being
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Introduction Cerebral palsy (CP) is the most common physical disability in childhood.1 Overall, 90% of individuals with CP are expected to grow into adulthood.2 In 2007, it was estimated that 74% of Australians with CP were aged 20 years and over.3 Much is known about outcomes in childhood but far less has been reported about outcomes in young adults, information that would be useful in helping families to plan their child’s future needs and would also assist health providers in formulating short and long term goals. An outcome that is important to consider is quality of life (QOL). Quality of life (QOL), a subjective and multidimensional concept, is defined by the World Health Organisation as ‘an individual's perception of their position in life in the context of the culture and value systems in which they live and in relation to their goals, expectations, standards and concerns’.4
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The level of QOL reported by children with CP varies depending on the QOL instrument used. Studies using instruments that focus on health and function tend to find that the QOL of children with CP is lower than the QOL of their typically developing peers.5-7 By contrast, studies that have used instruments focusing on subjective perceptions of life have demonstrated that, with the exception of physical well-being, QOL of children with CP is similar to their typically developing peers.8, 9 Although gross motor function has been associated with physical well-being,8-10 it has not been shown to be associated with social or psychological well-being.9, 10
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Little is known about the QOL of adults with CP. Although a few studies have now been conducted in adults, only one study was population-based and thus representative of the entire CP population. Other studies included adults across a wide age range or a mix of different disabilities. For example, a large North American study of 3638 young people (16-27 years) with CP and other developmental disabilities demonstrated that 80% of the sample was satisfied with life in general.11 Furthermore, a Japanese study on the subjective well-being of 81 adults with CP (26-51 years) found that adults whose physical health was poor, and those who were dependent in activities of daily living, reported lower well-being scores.12 An additional finding was that well-being scores were lower for individuals aged in their early 40s than for those in their early 30s. A study of 199 adolescents and young adults with CP, which measured QOL using a utility QOL measure demonstrated that QOL scores for adolescents and young adults were similar.13 To our knowledge, the only population-based study of adults with CP was conducted in Norway.14 Using the Life Satisfaction Scale,15 results from this study demonstrated that more than half the participants were satisfied with their life as a whole, their ability to perform activities of daily living, their family life, and their contact with friends. One third felt satisfied with their work situation and less than one third was satisfied with their sexual life and partner relationships. Compared to the general
ACCEPTED MANUSCRIPT population, adults with CP reported significantly lower satisfaction scores for life as a whole, sexual life, family life, ability to perform daily activities, leisure, and work.14
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There have been no population-based studies that have focused specifically on the QOL of young adults with CP aged 20-30 years. Individuals in this age group are at a particularly important time of life when there may be significant issues with respect to social integration following departure from school and pediatric medical services.
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Exploration of the determinants of QOL is important for future program planning and service delivery. To date, there have been only a couple of studies that have examined the determinants of QOL for adults,12, 16, 17 and, as mentioned previously, these studies have had limitations in terms of the type of QOL instrument used and the generalisability of the findings to young adults with CP. Despite their limitations, previous studies have provided some evidence to suggest that QOL is related to the severity of impairment. For example, a Japanese study of adults with CP showed that CP subtype was related to level of subjective well-being.16 Inability to perform daily activities was also related to lower subjective feelings of well-being. A study of adults with developmental disabilities living in the United States of America demonstrated that those who engaged in paid employment, schooling, and/or volunteer work were more likely to report higher levels of life satisfaction.12 Furthermore, in a study of 790 adults with a physical disability, levels of life satisfaction were influenced by employment status and marital status.17 This empirical evidence is inconsistent with a wealth of ongoing research challenging the theory that disability leads to poor QOL.7, 11, 18-20 Whilst is it clear that for children with CP functional status is only weakly related with QOL,18-22 for adults, issues related to a lack of independence may be associated with lower QOL.
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Aims of Study The primary aim of this study was to compare the QOL of a cohort of young Australian adults with CP with a cohort of able-bodied peers, using the same multidimensional instrument. To gain insight into the determinants of QOL, a further aim was to explore the relationship between QOL and impairments, functioning, and social participation.
ACCEPTED MANUSCRIPT Methods This cross-sectional study was conducted at the Royal Children’s Hospital in Melbourne, Australia, and ethics approval was granted from the Hospital’s Human Research Ethics Committee (EHRC 26117).
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Identification of potential CP participants Young adults (n=1007) born between 1976 and 1985 were identified from the Victorian Cerebral Palsy Register which holds data pertaining to people with CP born in Victoria from 1970 onwards. Those who were known to be deceased (n=163) and those for whom no contact address could be found (n=26) were excluded.
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Selection of a general population comparison group A general population sample of 751 young North American adults, aged 18 to 25 years, who had completed The Quality of Life Instrument for Young adults (YAQOL),23 was selected for comparison with the CP sample. Sample size calculation Using a hypothesised mean of 85 and a standard deviation of 20, as published by Chen et al,23 we calculated that a sample of 237 self-reporting CP participants would enable us to find a 10% difference in mean standardised YAQOL scores between the CP and general population groups with 80% power and alpha of 0.05.
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Study procedures Using hospital medical records, an online telephone directory and public access electoral roll, tracing letters were sent to young adults and their families to ascertain whether they would be interested in receiving information about the study and could be contacted at their previously recorded address. Once confirmed, a second more detailed letter was dispatched together with the individually coded survey booklet and the consent form. Individuals who failed to return the survey within one month were given a reminder telephone call. If participants were physically unable to write their responses, they were invited to request a family member or carer to act as their scribe. If there was a significant intellectual disability, a parent or carer was asked to complete the survey as a proxy for the young adult. Outcome measures A survey booklet was compiled to gather information about QOL, gross motor function, independence in self-care, and social participation. QOL was assessed with the Quality of Life Instrument for Young Adults (YAQOL) across five domains: physical health, psychological well-being, social relationships, role function, and environmental context (refer to Table 1). The YAQOL was selected because it allows comparison with a normative group, and it is argued that the
ACCEPTED MANUSCRIPT domains of QOL that are important to people without disabilities would also be important to people with a disability. Domain and total scores range from 0-100, with higher scores indicating better QOL. Testing of the instrument on a general population sample of 751 young adults provided evidence for its construct validity and internal consistency (Cronbach’s α = 0.63-0.88).23
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Gross motor function was classified by the participants or their carer using the Gross Motor Function Classification System (GMFCS).24 The GMFCS is widely used for children with CP but has also been shown to be reliable and valid for classifying the gross motor function of adults.25, 26 The family report version27 of the GMFCS was used for this study. Results from this version are highly consistent with those of professionals..28 Using the GMFCS, gross motor function is categorised into five levels, with level I representing the most independent gross motor function and level V the least.
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Independence in self-care activities was evaluated by the CP participants or their carers using the Barthel Index.29 The Index has ten domains, comprising feeding, bathing, grooming, dressing, bladder management, bowel management, toilet use, transfers, mobility, and ability to use stairs. The maximum total score is 100, with higher scores representing greater self-care independence. Evidence has been presented in support of its construct validity and reliability.30, 31 The self-report version has been found to be closely correlated with the professional report.32
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Data on social participation were obtained from questions about four life areas: education, employment, living situation and marital status. Yearly income described personal income from private sources, salary/wages, pensions and government allowances. Information on CP subtype and comorbidities such as epilepsy and intellectual, speech, hearing, and vision impairments was collected from the Victorian Cerebral Palsy Register.
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Statistical analysis Young adults with CP were described in terms of their level of independence in self-care activities, gross motor function, comorbidities, social participation, and QOL. QOL scores were compared between self-reporting and proxy-reported participants using independent sample t-tests and Mann-Whitney U tests for domain scores with distributions that were normal and non-normal respectively. Self-reported QOL scores for the young adults with CP were also compared with self-reported QOL scores from the general population sample using independent sample t-tests. To reduce the risk of type 1 errors, the more stringent alpha level of 0.01 was used to denote a statistically significant difference between groups.
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To evaluate associations between QOL for self-reporting participants and level of impairment, functional ability, social participation, and personal and environmental factors, univariate and multivariate linear regression analyses were performed. Using a stepwise forward approach, variables were entered into the multivariate regression models at p<0.05 and removed at p>0.10. The best predictive models were presented with p-values, standardized regression co-efficients (β), and the amount of variance explained by the regression model (R2). All analyses were performed in SPSS 13.0 for Windows.33
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Results Of the 818 potential CP participants, 534 responded to the tracing letter and verified their address. The remaining 284 were unable to be traced despite 141 tracing letters being re-sent to new addresses. Surveys and consent forms were completed by 335 young adults or their proxies, an overall participation rate of 63% of those located. Of the remaining 199 individuals (non-participants), 119 declined to participate after receiving the tracing letter or survey, and 80 agreed to participate but did not return the survey or the consent form. Participants and non-participants were similar with respect to age, gender, motor topography, and frequency of comorbidities (all p>0.05). There were relatively fewer participants than non-participants with mild motor impairment (p=0.007).
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Characteristics of the CP group The mean age of the study participants was 24.7 [s.d=2.8] years; 51% were male and 49% female. Two hundred and seven (62%) of the 335 participants self-reported their QOL. Table 2 summarises the type and severity of the impairments, levels of gross motor function, independence in self-care activities, and socio-demographic characteristics of the study participants. Half the CP participants were able to walk independently; the remainder required mobility devices such as walkers, scooters, manual or powered wheelchairs. With respect to self-care, 65% of the young adults were dependent on others for assistance, whereas 35% were classified as independent.
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Compared to the proxy group, self-reporting participants were less likely to have severe motor, intellectual, speech and visual impairments (Table 2). Self-reporting participants also had a lower frequency of epilepsy, higher levels of gross motor function, and a greater likelihood of being independent in activities of daily living (52% versus 9%). Where help was needed, self-reporting participants most often needed assistance with dressing (42%) and stairs (41%). On the other hand, independence was most commonly reported for bladder (79%) and bowel (83%) continence. For the self-report group, 61% were independent in eating and 75% were independent for transfers. For the proxy report group, 11% were independent in eating and 34% were independent in transfers. Levels of QOL As shown in Table 3, self-reporting participants had higher QOL scores than participants in the proxy group for each domain (p<0.0001). The greatest difference was in the role function domain and the least difference was in the environmental context domain. When compared with the general population sample, self-reporting participants had similar QOL scores for the social relationship and environmental context domains (p>0.05), while QOL scores were lower for the physical health, psychological well-being, and role function domains (p<0.001).
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Determinants of QOL Physical Health Domain Using multivariate modelling, the best independent predictors of QOL scores for the physical health domain in self-reporting participants were GMFCS level (p<0.0001), Barthel Index score (p<0.0001), gender (p=0.006), and epilepsy status (p=0.020). Together these accounted for 34% of the variation. Females with relatively milder impairment in gross motor function, greater independence in self-care activities, and no epilepsy were more likely to have better physical health domain scores. Motor topography, intellectual impairment, and socioeconomic status were significantly associated with physical health domain scores in the simple linear regressions, but not in the multivariate analysis.
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Psychological Domain Scores on the psychological well-being domain in self-reporting participants varied according to the level of independence in self-care activities (p=0.003). The best independent predictor identified for this domain was Barthel Index score, but only 5% of the variance in domain scores was explained by this multivariate model. Employment status and educational achievement were significantly associated with psychological well-being domain scores in the simple linear regressions, but not in the multivariate analysis.
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Social Relationship Domain Educational achievement (p=0.003), yearly income (p=0.014), and vision (p=0.034) were significant predictors of independent living on multivariate analysis. These factors together accounted for 16% of the variance in social relationship domain scores. Intellectual impairment, speech impairment, GMFCS level, Barthel Index score and social participation variables were significantly related to the social relationship domain score in14 the simple linear regressions, but not in the multivariate analysis.
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Role Function Domain Lower QOL scores in relation to performance in productive roles such as employment, education, or home duties were associated with higher GMFCS levels (p=0.005), unemployment, (p<0.0001), and intellectual impairment (p<0.0001) on multivariate modelling. These variables together explained 48% of the variance in role function domain scores. Speech and vision impairment, Barthel Index score, living situation, educational achievement and yearly income were significantly related to role function domain score in the simple linear regressions, but not in the multivariate analysis. Environmental Context Domain Using multivariate analysis, QOL in relation to the home environment, community services, and access varied according to gross motor function (p=0.042), Barthel
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Index score (p<0.0001), motor topography (p=0.034), and yearly income (p=0.041). In contrast to young adults with mildly impaired gross motor function and greater independence in self-care activities, those with higher GMFCS levels and lower levels of independence were more likely to report poorer QOL in the environmental context domain. Intellectual impairment, vision impairment, employment status, and educational achievement were also significantly associated with environmental context domain score in the simple linear regressions, but not in the multivariate analysis.
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Discussion This is the first study to document QOL in a population-based sample of young Australian adults with CP. The results indicated that 1) self-reporting participants reported higher QOL than was reported for participants by proxy; 2) young adults with CP reported lower QOL than a normative general population sample on all domains except social relationships and environmental context; and 3) mobility and functional independence were strongly associated with QOL related to physical health and employment status, but they were not related to satisfaction with social relationships.
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Proxy and Self-Reported QOL Compared to self-reporting young adults with CP, proxies reported a lower level of QOL within each domain for participants who were unable to self-report. The biggest difference was in the role function domain where proxies reported markedly lower scores than young adults who self-reported. As most of the participants by proxy (85%) did not engage in any work, education, or home duties, it would be unlikely that their proxies would report a sense of well-being from participation in these roles. There are no comparable studies of adults in the literature, but similar results have been found in QOL studies of children where self and proxy reported data were collected from the same group.34-40 Given the dynamic and subjective nature of QOL, there is often lack of agreement between self and proxy reports. Parents frequently report lower QOL than children themselves, highlighting the complexities involved in reporting someone else’s QOL.
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Levels of QOL of adults with CP Our results indicate that the QOL of self-reporting young adults with CP, although variable, was perceived as being lower than the QOL of their typically developed peers in some, but not all, domains. Self-reporting CP participants reported lower levels of QOL than their peers in the domains of physical health, psychological well-being, and role function, but similar levels in the social relationship and environmental context domains. A Norwegian population-based study of adults with CP aged 18 to 72 years produced somewhat similar findings.14 Using the Life Satisfaction Scale,15 the authors of the Norwegian study found that, compared to levels of satisfaction in a reference group, adults with CP had lower satisfaction in six of nine domains, including family life, sexual life, activities of daily living, leisure, work situation, and life as a whole, but similar levels of satisfaction with respect to economic status, contact with friends, and marital situation. Studies of QOL in children have also shown comparable levels of QOL between CP and normative samples in some domains.8, 9 For example, one study found that self-reported QOL of children with CP aged 8 to 12 years was similar to their peers without CP except for QOL relating to physical well-being and the school environment.8
ACCEPTED MANUSCRIPT Determinants of QOL This study found no association between psychological well-being and variables related to body structure and function in young adults with CP. At the activity level, functional independence was only weakly associated with psychosocial well-being in comparison to physical- and role function well-being, and there was little evidence of an association between gross motor function and psychological well-being.
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Our finding that gross motor function was only associated with the YAQOL domains of physical health, role function, and environmental context, but not to psychological well-being or social relationship, further supported findings from thirteen previous studies of children and adolescents with CP in the United States,6, 9, 18-20, 41 Canada,21, 42, 43 Europe,8 and Australia.7, 10, 44 Although nine of these studies,6, 7, 18-21, 41, 42, 44 used instruments that focus on health status or functioning when describing QOL, such as the Pediatric Quality of Life Inventory, the Health Utilities Index and the Pediatric Evaluation of Disability Inventory, their results also indicated that gross motor function was associated with physical function and functional well-being, but not psychosocial well-being. This finding was also compatible with the findings of other recent studies that used subjective QOL instruments to assess well-being. Dickinson et al8 reported that walking ability was only associated with physical well-being, and was not associated with QOL in six of the ten KIDSCREEN domains, including psychological well-being, self-perception, social support, school environment, financial resources, and social acceptance. Rosenbaum et al43 using the Quality of Life Instrument for people with developmental disabilities, found that none of the QOL domain scores (being, belonging, and becoming) varied with GMFCS level.
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Little attention has been given to the associations between functional independence and QOL for individuals with CP. The current study finding is partly supported by Majnemer’s study of children with CP.42 This study used the CHQ PF-50 and PedsQL 4.0 and demonstrated an association between functional limitation in daily living skills and physical well-being, but not psychosocial well-being. Furukawa et al16 also described lower subjective well-being in adults with CP who were dependent on others for activities of daily living compared to those who were independent. They included a wide age range (26-51y) and used the Philadelphia Geriatric Centre Morale Scale to evaluate subjective well-being rather than a multidimensional QOL instrument. The finding of strong associations between gross motor function, functional independence, and QOL in the physical- and functional-related domains highlights the importance of efforts to improve independence in self-care with ongoing rehabilitation, specially designed equipment, and personal assistance in this population.
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Contrary to the assumption that young adults with severe CP have low psychosocial well-being, it is apparent that these individuals can have good psychosocial well-being regardless of their disability. According to the World Health Organisation definition,4 QOL is a broad construct that includes perceptions of health and function rather than health or functional status per se. Furthermore, individuals who grow up with CP may not have the same life experiences as their peers, and may accept disability and functional limitation as a part of their life. Positive attitudes towards life may enable individuals with disabilities to adapt well to their limitations.11, 45 Personal attitudes, for example, establishing a balance between body, mind and spirit, were identified as one of the coping strategies used to separate disability from life quality.11 Values, goals, and expectations may be adjusted to comply with their abilities. In a study of adolescents with CP, being believed in, believing in yourself, and being accepted by others were key factors relating to happiness and success in life.45 Alternatively, perceptions may be influenced by their environment. For example, individuals attending mainstream education or in employment may establish the same standards for life quality as their able-bodied peers, whereas those in special schools or day centres, having peers with physical or learning disabilities, might set different norms.
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We found no previous studies that explored the relationship between social participation and QOL in adults with CP. In this study of 20-30 year olds, employment status was associated with each QOL domain except psychological well-being. This finding is consistent with previous research showing that life satisfaction was influenced by employment status in young adults with developmental and physical disabilities.12, 17 These findings highlight the benefit of employment and employment-related support services along with occupational training and opportunities for volunteer work as an alternative. Community networks, government policies, and assistive technologies are important means of increasing employment opportunities.46
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Limitations of this study include the fact that the study sample consisted of slightly fewer young adults at the mild end of the severity spectrum than in the CP population and this may have affected the comparison between CP participants and the comparison sample. A further limitation was that proxy-report data were not collected for self-reporting participants and we were unable to fully investigate differences between self and proxy reported QOL. Also, due to the lack of Australian normative QOL data, comparison QOL data were taken from a general population sample of young adults aged 18 to 25 years old in the United States. It is possible that cultural differences contribute to the differences observed between the study and comparison groups. The YAQOL has not been previously used in Australia, and time and cost constraints prevented the recruitment of a comparison sample from the Australian general population.
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Conclusion Previous studies exploring QOL in CP have focused on children and adolescents. This is the first study to examine young adults’ QOL and the relationships between QOL, level of impairment, function, social participation, and personal and environmental factors. The results suggest that self-reported QOL in young adults with CP is lower than QOL reported by able-bodied young adults, except in the areas of social relationships and environmental context. Consistent with this finding, mobility appears to contribute to some aspects of self-reported QOL in young adults with CP but not to QOL scores for social relationships and psychological well-being.
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14. Jahnsen R. Being adult with a "childhood disease" - a survey on adults with cerebral palsy in Norway. Thesis. Oslo: Faculty of Medicine, University of Oslo; 2004. 15. Fugl-Meyer A, Branholm I, Fugl-Meyer K. Happiness and domain-specific life satisfaction in adult northern Swedes. Clinical Rehabilitation. 1991;5(1):25-33. doi: 10.1177/026921559100500105 16. Furukawa A, Iwatsuki H, Nishiyama M, Nii E, Uchida A. A study on the subjective well-being of adult patients with cerebral palsy. Journal of Physical Therapy Science. 2001;13(1):31-5. http://doi.org/10.1589/jpts.13.31 17. Kinney WB, Coyle CP. Predicting life satisfaction among adults with physical disabilities. Archives of Physical Medicine & Rehabilitation. 1992;73(9):863-9. 18. McCarthy ML, Silberstein CE, Atkins EA, Harryman SE, Sponseller PD, Hadley-Miller NA. Comparing reliability and validity of pediatric instruments for measuring health and well-being of children with spastic cerebral palsy. Developmental Medicine & Child Neurology. 2002;44(7):468. 19. Pirpiris M, Gates PE, McCarthy JJ, D'Astous J, Tylkowksi C, Sanders JO, Dorey FJ, Ostendorff S, Robles G, Caron C, Otsuka NY. Function and well-being in ambulatory children with cerebral palsy. Journal of Pediatric Orthopedics. 2006;26(1):119-24. doi: 10.1097/01.bpo.0000191553.26574.27 20. Vargus-Adams J. Health-related quality of life in childhood cerebral palsy. Archives of Physical Medicine & Rehabilitation. 2005;86(5):940-5. 21. Kennes J, Rosenbaum P, Hanna SE, Walter S, Russell D, Raina P, Bartlett D, Galuppi B. Health status of school-aged children with cerebral palsy: information from a population-based sample. Developmental Medicine & Child Neurology. 2002;44(4):240. 22. Leger R, Spencer T. Functional independence, health-related quality of life, parental stress in adolescents with disabilities. (Scientific Posters). Developmental Medicine & Child Neurology. 2008;50(suppl. 4):46-7. 23. Chen H, Cohen P, Kasen S, Gordan K, Dufur R, Smailes E. Construction and validation of a quality of life instrument for young adults. Quality of Life Research. 2004;13(4):747-59. 24. Palisano R, Rosenbaum P, Walters S, Russell D, Wood E, Galuppi B. Developmental and reliability of a system to classify gross motor function in children with cerebral palsy. Developmental Medicine & Child Neurology. 1997;39:214-23. 25. Jahnsen R, Aamodt G, Rosenbaum P. Gross Motor Function Classification System used in adults with cerebral palsy: agreement of self-reported versus professional rating. Developmental Medicine & Child Neurology. 2006;48(9):734-8. doi: 10.1017/S0012162206001575 26. Sandstrom K, Alinder J, Oberg B. Descriptions of functioning and health and relations to a gross motor classification in adults with cerebral palsy. Disability & Rehabilitation. 2004;26(17):1023-31. doi: 10.1080/09638280410001703503
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27. Morris C, Galuppi BE, Rosenbaum PL. Reliability of family report for the Gross Motor Function Classification System. Developmental Medicine & Child Neurology. 2004;46(7):455. doi: 10.1017/S0012162204000751 28. Morris C, Kurinczuk JJ, Fitzpatrick R, Rosenbaum PL. Who best to make the assessment? Professionals' and families' classifications of gross motor function in cerebral palsy are highly consistent. Archives of Disease in Childhood. 2006;91(8):675-9. doi:10.1136/adc.2005.090597 29. Mahoney FI, Barthel D. Functional evaluation: the Barthel Index. Maryland State Medical Journal. 1965;14:61-5. Used with permission. 30. Wyller TB, Sveen U, BautzHolter E. The Barthel ADL Index one-year after stroke – comparison between relatives and occupational therapists scores. Age and Ageing. 1995;24:398–401. doi: 10.1093/ageing/24.5.398 31. Finch E, Brooks D, Stratford PW, Mayo NE. MAS, Berg Balance Scale, Barthel ADL Index. Physical Rehabilitation Outcome Measures: Lippincott Williams & Wilkins; 2002. p. 87–90, 3–4, 169–72. 32. Collin C, Wade DT, Davies S, Horne V. The Barthel ADL Index: a reliability study. International Disability Studies. 1988;10(2):61-3. 33. SPSS 13.0 for Windows. Chicago: SPSS Inc; 2004. 34. Eiser C, Morse R. Can parents rate their child's health-related quality of life? Results of a systematic review. Quality of Life Research. 2001;10(4):347-57. 35. Jokovic A, Locker D, Guyatt G. How well do parents know their children? implications for proxy reporting of child health-related quality of life. Quality of Life Research. 2004;13(7):1297-307. 36. Armstrong RW, Rosenbaum PL, King S. Self-perceived social function among disabled children in regular classrooms. Journal of Developmental & Behavioral Pediatrics. 1992;13(1):11-6. 37. Ellerton ML, Stewart MJ, Ritchie JA, Hirth AM. Social support in children with a chronic condition. Canadian Journal of Nursing Research. 1996;28(4):15-36. 38. Skär LRN. Peer and adult relationships of adolescents with disabilities. Journal of adolescence. 2003;26(6):635-49. doi:10.1016/S0140-1971(03)00061-7 39. Waters E, Davis E, Mackinnon A, Boyd R, Graham HK, Kai Lo S, Wolfe R, Stevenson R, Bjornson K, Blair E, Hoare P, Ravens-Sieberer U, Reddihough D. Psychometric properties of the quality of life questionnaire for children with CP. Developmental Medicine & Child Neurology. 2007;49(1):49-55. 10.1017/S0012162207000126 40. Davis E, Nicolas C, Waters E, Cook K, Gibbs L, Gosch A, Ravens-Sieberer U. Parent-proxy and child self-reported health-related quality of life: using qualitative methods to explain the discordance. Quality of Life Research. 2007;16(5):863-71. 10.1007/s11136-007-9187-3 41. Schneider JW, Gurucharri LM, Gutierrez AL, Gaebler-Spira DJ. Health-related quality of life and functional outcome measures for children with cerebral palsy. Developmental Medicine & Child Neurology. 2001;43(9):601-8.
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42. Majnemer A, Shevell M, Rosenbaum P, Law M, Poulin C. Determinants of life quality in school-age children with cerebral palsy. Journal of Pediatrics. 2007;151(5):470-5. 10.1016/j.jpeds.2007.04.014 43. Rosenbaum PL, Livingston MH, Palisano RJ, Galuppi BE, Russell DJ. Quality of life and health-related quality of life of adolescents with cerebral palsy. Developmental Medicine & Child Neurology. 2007;49(7):516-21. doi:10.1111/j.1469-8749.2007.00516.x 44. Maher CA, Olds T, Williams MT, Lane AE. Self-reported quality of life in adolescents with cerebral palsy. Physical & Occupational Therapy in Pediatrics. 2008;28(1):41-57. doi:10.1300/J006v28n01_04 45. King GA, Cathers T, Polgar JM, MacKinnon E, Havens L. Success in Life for Older Adolescents with Cerebral Palsy. Qualitative Health Research. 2000;10(6):734-49. doi:10.1177/104973200129118796 46. McNaughton D, Light J, Arnold KB. "Getting your wheel in the door": successful full-time employment experiences of individuals with cerebral palsy who use augmentative and alternative communication. Augmentative and Alternative Communication. 2002;18(2):59-76.
ACCEPTED MANUSCRIPT Table 1. Descriptions of domains and scales in the YAQOL Domains
Scales
Descriptions
Physical health
Physical health
Measures overall physical health, limitation due to chronic pain, chronic physical problems, disturbance due to the illness, and energy level in the previous year
Negative Affect Social relationship
Social Support
Role function
Role Stress
Role Autonomy Self-Role Morale
Assesses the extent of stress in the individual’s major productive roles in the settings of employment, education or home duties Assesses the extent of influence over role organization, scheduling, and decisions Assesses the degree of role satisfaction and consistency with long term goals Assesses the degree of role satisfaction and consistency with long term goals in co-workers or other students
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Other’s Role Morale
Environmental context
Measures the extent of contact and quality of relationships with friends Measures the quality of relationships with a partner or with close friends
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Religious Orientation
Examines expectations for the future and recognition of the long-term goals Examines the importance of the religious belief and frequency of service attendance Examines negative, depressed and anxious feelings in life
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Psychological well-being Positive Outlook
Home
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Community
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Mobility
Resources
Evaluates comfort, safety, quietness, and privacy at home Evaluates social and service aspects of the community Evaluates ease of access to work, school, shopping, leisure and social activities Evaluates the ability to access medical care and financial support
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Table 2: Characteristics of the CP study group by reporting status, excluding participants with missing data. Self-report Proxy-report Total CP group group study group n=207 n=128 n=335 n (%) n (%) N (%) GMFCS (n=335) Level I 61 (29.5) 9 (7.0) 70 (20.9) Level II 78 (37.7) 29 (22.7) 107 (31.9) Level III 18 (8.7) 16 (12.5) 34 (10.1) Level IV 31 (15.0) 31 (24.2) 62 (18.5) Level V 19 (9.2) 43 (33.6) 62 (18.5) Cerebral palsy subtype (n=335) Spastic monoplegia/hemiplegia 84 (40.6) 24 (18.7) 108 (32.2) Spastic diplegia 54 (26.1) 22 (17.2) 76 (22.7) Spastic triplegia/quadriplegia 50 (24.1) 65 (50.8)) 115 (34.4) Ataxia/Dyskinesia/Hypotonia 19 (9.2) 17 (13.3) 36 (10.7) Intellectual impairment (n=317) None (IQ≥70) 141 (71.6) 10 (8.3) 151 (47.6) Mild (50≤IQ<70) 35 (17.8) 18 (15.0) 53 (16.7) Moderate (30≤IQ<50) 16 (8.1) 38 (31.7) 54 (17.0) Severe (IQ<30) 5 (2.5) 54 (45.0) 59 (18.7) Speech impairment (n=315) None 129 (66.2) 9 (7.5) 138 (43.8) Some 47 (24.1) 41 (34.2) 88 (27.9) Non-verbal 19 (9.7) 70 (58.3) 89 (28.3) Hearing impairment (n=313) None 178 (91.8) 107 (89.9) 285 (91.1) Some 13 (6.7) 11 (9.2) 24 (7.7) Bilateral deafness 3 (1.5) 1 (0.8) 4 (1.3) Vision impairment (n=313) None 75 (39.1) 24 (19.8) 99 (31.6) Strabismus only 27 (14.1) 11 (9.1) 38 (12.1) Some 90 (46.9) 83 (68.6) 173 (55.3) Functionally blind 0 (0.0) 3 (2.5) 3 (1.0) Epilepsy (n=329) None/resolved 160 (79.6) 42 (32.8) 202 (61.4) Epilepsy 41 (20.4) 86 (67.2) 127 (38.6) Self-care independence (n=335) Independent (Barthel score 95-100) 107 (51.7) 12 (9.4) 119 (35.5) Assistance needed (Barthel score 60-94) 50 (24.2) 24 (18.8) 74 (22.1) Dependent (Barthel score 0-59) 50 (24.2) 92 (71.9) 142 (42.4) Education level achieved (n=334) Below Year 12 51 (24.8) 116 (90.6) 167 (50.5) Completed secondary 40 (19.4) 8 (6.3) 48 (14.4) Tertiary 115 (55.8) 4 (3.1) 119 (35.6) Employment status (n=333) Employed 108 (52.7) 13 10.2 121 (36.3)
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(23.9) (28.8) (47.3) (16.1) (31.2)
6 7 115 1 114
4.7 5.5 89.8 0.8 89.0
(16.5) (19.8) (63.7) (10.2) (53.5)
70 (20.9) 219 (65.4) 46 (13.7)
23 11.1 184 88.9
2 1.6 126 98.4
25 (7.5) 310 (92.5)
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3 2.3 93 72.7 32 25.0
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55 66 212 34 178
67 32.3 126 60.9 14 6.8
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Full-time employment Part-time employment Unemployed Currently job seeking Never sought employment Living situation (n=335) Living independently Living with parents Community residential unit Marital status (n=335) Married or partnered Single
ACCEPTED MANUSCRIPT Table 3: Comparisons between self-report and proxy report CP groups and between the self-report CP group and general population sample for mean YAQOL domain scores
Environmental context, mean (SD)
Proxy-report General group population n=128 sample (n=751) 58.87 (22.0) 46.62 (21.6) 72.05 (16.3) 53.31 (16.9) 43.78 (18.9)
67.79 (12.8)
77.28 (15.8) 67.52 (14.8)
78.61 (10.8)
50.33 (31.5) 9.26 (22.7)
60.83 (14.4)
72.01 (12.7) 64.49 (11.4)
T statistic
P value for group differences
ta=4.95 tb=9.44 ta=4.66 tb=13.43 ta=5.58 tb=1.40
pa<0.0001 pb<0.0001 pa<0.0001 pb<0.0001 pa<0.0001 pb=0.1627 pa<0.0001 * pb<0.0001 pa<0.0001 pb =0.0325
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Physical health, mean (SD) Psychological wellbeing, mean (SD) Social relationship, mean (SD) Role function, mean (SD)
Self-report group n=207
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QOL scores
73.91 (10.8)
tb=6.90 ta=5.46 tb=2.14
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QOL scores range from 0 to 100, higher score indicates better QOL; SD: standard deviation; ta, pa values refer to differences between self and proxy report groups, tb, pb values refer to the differences between the self-report CP group and general population comparison group, calculated using Independent-Sample t tests. *:p value was calculated using Mann-Whitney U test because the role function scores in proxy participants were not normally distributed.
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Table 4: Determinants of YAQOL domain and overall scores for young adults with CP (self-reporting participants, summary of multivariate linear regressions)
Psychological well being
ß (standard coefficients)
p
ß (standard coefficients)
p
Social relationship ß (standard coefficients)
Body structure and function -0.05
0.394
0.04
0.560
-0.10
Motor topography
-0.09
0.104
-0.02
0.673
-0.09
Intellectual impairment
-0.11
0.090
-0.00
0.994
-0.10
Speech impairment
-0.03
0.658
-0.09
0.308
Hearing impairment
-0.08
0.183
-0.01
0.884
Vision impairment
-0.03
0.638
-0.03
0.729
Presence of epilepsy
-0.14
0.020 *
0.00
0.981
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Activity
p
0.146
ß (standard coefficients)
p
0.334
0.295
-0.06
0.542
-0.16
0.034 *
0.225
-0.22
-0.07
0.341
<0.0001 ***
-0.12
0.104
-0.05
0.422
0.04
0.550
-0.10
0.169
-0.05
0.354
0.08
0.159
-0.16
0.034 *
-0.04
0.546
-0.06
0.442
-0.06
0.424
-0.02
0.739
-0.04
0.513
-0.16
0.042 *
0.00
0.962
-0.06
0.475
-0.16
0.005 **
Functional independence (BI)
0.40
<0.0001***
0.23
0.003 **
0.07
0.395
0.06
0.411
Living situation
-0.04
0.562
Marital status
0.00
0.982
Age
0.06
0.280
Gender
-0.17
Educational achievement
-0.05
Personal factors
-0.08
0.360
-0.08
0.344
-0.44
-0.01
0.953
-0.11
0.163
-0.02
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0.907
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<0.0001 ***
-0.01
p
0.07
-0.35
Employment status
ß (standard coefficients)
0.719
Mobility (GMFCS level)
Participation
Environmental context
-0.02
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Role function
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Predictor variables
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Physical health
-0.08
0.284
-0.07
0.353
0.00
0.985
-0.06
0.006 **
-0.07
0.385
0.452
-0.07
0.353
0.36
<0.0001 ***
-0.02
0.834
0.767
0.05
0.439
-0.07
0.241
-0.10
0.111
0.371
-0.13
0.025
-0.05
0.412
0.06
0.401
0.06
0.284
-0.09
0.143
-0.23
0.003 **
-0.03
0.621
-0.03
0.679
<0.0001***
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Yearly income
-0.07
0.302
-0.04
0.666
-0.19
-0.05
0.445
-0.16
0.041 *
0.01
0.887
0.06
0.416
0.04
0.554
0.07
0.260
0.06
0.306
Healthcare needs met
-0.05
0.396
-0.09
0.263
-0.07
0.352
-0.27
-0.07
0.386
Location of residence
0.01
0.780
-0.01
0.949
-0.04
0.579
0.03
0.663
0.014 *
Transferred to adult services
R=0.42, R2=0.18, adjusted R2 =0.16, p <0.0001
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R=0.60, R2=0.36, adjusted R=0.23, R2=0.05, R2 =0.34 adjusted R2 =0.05 p p <0.0001 =0.001
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Environmental factors
-0.05
<0.0001 *** 0.427
R=0.71, R2=0.50, adjusted R=0.44, R2=0.19, adjusted R2 =0.17 R2 =0.48 p <0.0001 p <0.0001
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Analyses were done for 207 self-reporting young adults; *: p < 0.05; **: p < 0.01; ***: p <0.001; R : the coefficient of determination, calculated using the multivariate linear
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regression analysis; GMFCS: Gross Motor Function Classification System; the BI: the Barthel Index; p < 0.05 are presented in bold.
S1936-6574(16)30045-0
DOI:
10.1016/j.dhjo.2016.04.006
Reference:
DHJO 487
To appear in:
Disability and Health Journal
Received Date: 21 September 2015 Revised Date:
15 April 2016
Accepted Date: 18 April 2016
Please cite this article as: Jiang B, Walstab J, Reid SM, Davis E, Reddihough D, Quality of life in young adults with Cerebral Palsy, Disability and Health Journal (2016), doi: 10.1016/j.dhjo.2016.04.006. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Title Page Quality of life in young adults with Cerebral Palsy Benran Jiang PhD.a,b, Janet Walstab BSc b,c, Susan M. Reid PhD, MClinEpi, BAppSc b,c, Elise Davis PhD d, Dinah Reddihough MD, BSc, FRACP, FAFRM a,b,c a
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Developmental Medicine, The Royal Children's Hospital, 50 Flemington Road, Parkville, Victoria, 3052, Australia b Department of Paediatrics, University of Melbourne, 50 Flemington Road, Parkville Victoria, 3010, Australia c Developmental Disability & Rehabilitation Research, Murdoch Childrens Research Institute, 50 Flemington Road, Parkville, Victoria, 3052, Australia d The Jack Brockhoff Child Health and Wellbeing Program, Centre for Health Equity, The Melbourne School of Population & Global Health, 207 Bouverie St, The University of Melbourne, Victoria, 3010, Australia. Corresponding author Dr Elise Davis The Jack Brockhoff Child Health and Wellbeing Program, Centre for Health Equity, The Melbourne School of Population & Global Health, Level 5, 207 Bouverie St, The University of Melbourne, VIC 3010, Australia. T: +61 3 8344 0921, E: [email protected]
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Disclosures We acknowledge the Victorian Medical Insurance Agency and the Victorian Department of Health who fund the Victorian Cerebral Palsy Register. Infrastructure support was provided by the Victorian Government's Operational Infrastructure Support Program. There are no conflicts of interest to declare.
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This work has been presented at the American Academy of Cerebral Palsy and Developmental Medicine, Phoenix, USA, September, 2009.
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Acknowledgements We would like to thank the young adults with CP and their families for participating in this study. We would also like to acknowledge Anna Lanigan for her support in finding research participants.
Points of Interest 1) Young adults with CP reported lower QOL than a normative general population sample on all domains except social relationships and environmental context. 2) Mobility and functional independence were strongly associated with QOL related to physical health and employment status, but not to satisfaction with social relationships. 3) Young adults with severe CP can have good psychosocial well-being regardless of their disability.
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Abstract word count: 250 Manuscript word count: 4000 Number of references: 46 Number of figures/tables: 4
ACCEPTED MANUSCRIPT Quality of life in young adults with Cerebral Palsy Abstract
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Background: Little is known about the quality of life (QOL) of young adults with cerebral palsy. Objective/Hypothesis: This cross-sectional analysis compares the QOL of a cohort of young Australian adults with CP with a cohort of able-bodied peers to explore the relationship between QOL and impairments, functioning, and social participation. Methods: Young adults identified from the Victorian Cerebral Palsy Register were invited to complete a survey about QOL, gross motor function, independence in self-care, and social participation. QOL was assessed with the Quality of Life Instrument for Young Adults (YAQOL). A general population sample of young North American adults, who had completed the YAQOL was selected for comparison. Results: Surveys and consent forms were completed by 335 young adults or their proxies, an overall participation rate of 63% of those located. The mean age of the study participants was 24.7 [s.d=2.8] years; 51% were male and 49% female. Two hundred and seven (62%) of the 335 participants self-reported their QOL. When compared with the general population sample, self-reporting participants had similar QOL scores for the social relationship and environmental context domains (p>0.05), while QOL scores were lower for the physical health, psychological well-being, and role function domains (p<0.001). There was no association between psychological well-being and variables related to body structure and gross motor function in young adults with CP. Conclusions: Contrary to the assumption that young adults with severe CP have low psychosocial well-being, it is apparent that these individuals can have good psychosocial well-being regardless of their disability.
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Keywords: Quality of Life, Young Adults, Cerebral Palsy, Well-being
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Introduction Cerebral palsy (CP) is the most common physical disability in childhood.1 Overall, 90% of individuals with CP are expected to grow into adulthood.2 In 2007, it was estimated that 74% of Australians with CP were aged 20 years and over.3 Much is known about outcomes in childhood but far less has been reported about outcomes in young adults, information that would be useful in helping families to plan their child’s future needs and would also assist health providers in formulating short and long term goals. An outcome that is important to consider is quality of life (QOL). Quality of life (QOL), a subjective and multidimensional concept, is defined by the World Health Organisation as ‘an individual's perception of their position in life in the context of the culture and value systems in which they live and in relation to their goals, expectations, standards and concerns’.4
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The level of QOL reported by children with CP varies depending on the QOL instrument used. Studies using instruments that focus on health and function tend to find that the QOL of children with CP is lower than the QOL of their typically developing peers.5-7 By contrast, studies that have used instruments focusing on subjective perceptions of life have demonstrated that, with the exception of physical well-being, QOL of children with CP is similar to their typically developing peers.8, 9 Although gross motor function has been associated with physical well-being,8-10 it has not been shown to be associated with social or psychological well-being.9, 10
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Little is known about the QOL of adults with CP. Although a few studies have now been conducted in adults, only one study was population-based and thus representative of the entire CP population. Other studies included adults across a wide age range or a mix of different disabilities. For example, a large North American study of 3638 young people (16-27 years) with CP and other developmental disabilities demonstrated that 80% of the sample was satisfied with life in general.11 Furthermore, a Japanese study on the subjective well-being of 81 adults with CP (26-51 years) found that adults whose physical health was poor, and those who were dependent in activities of daily living, reported lower well-being scores.12 An additional finding was that well-being scores were lower for individuals aged in their early 40s than for those in their early 30s. A study of 199 adolescents and young adults with CP, which measured QOL using a utility QOL measure demonstrated that QOL scores for adolescents and young adults were similar.13 To our knowledge, the only population-based study of adults with CP was conducted in Norway.14 Using the Life Satisfaction Scale,15 results from this study demonstrated that more than half the participants were satisfied with their life as a whole, their ability to perform activities of daily living, their family life, and their contact with friends. One third felt satisfied with their work situation and less than one third was satisfied with their sexual life and partner relationships. Compared to the general
ACCEPTED MANUSCRIPT population, adults with CP reported significantly lower satisfaction scores for life as a whole, sexual life, family life, ability to perform daily activities, leisure, and work.14
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There have been no population-based studies that have focused specifically on the QOL of young adults with CP aged 20-30 years. Individuals in this age group are at a particularly important time of life when there may be significant issues with respect to social integration following departure from school and pediatric medical services.
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Exploration of the determinants of QOL is important for future program planning and service delivery. To date, there have been only a couple of studies that have examined the determinants of QOL for adults,12, 16, 17 and, as mentioned previously, these studies have had limitations in terms of the type of QOL instrument used and the generalisability of the findings to young adults with CP. Despite their limitations, previous studies have provided some evidence to suggest that QOL is related to the severity of impairment. For example, a Japanese study of adults with CP showed that CP subtype was related to level of subjective well-being.16 Inability to perform daily activities was also related to lower subjective feelings of well-being. A study of adults with developmental disabilities living in the United States of America demonstrated that those who engaged in paid employment, schooling, and/or volunteer work were more likely to report higher levels of life satisfaction.12 Furthermore, in a study of 790 adults with a physical disability, levels of life satisfaction were influenced by employment status and marital status.17 This empirical evidence is inconsistent with a wealth of ongoing research challenging the theory that disability leads to poor QOL.7, 11, 18-20 Whilst is it clear that for children with CP functional status is only weakly related with QOL,18-22 for adults, issues related to a lack of independence may be associated with lower QOL.
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Aims of Study The primary aim of this study was to compare the QOL of a cohort of young Australian adults with CP with a cohort of able-bodied peers, using the same multidimensional instrument. To gain insight into the determinants of QOL, a further aim was to explore the relationship between QOL and impairments, functioning, and social participation.
ACCEPTED MANUSCRIPT Methods This cross-sectional study was conducted at the Royal Children’s Hospital in Melbourne, Australia, and ethics approval was granted from the Hospital’s Human Research Ethics Committee (EHRC 26117).
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Identification of potential CP participants Young adults (n=1007) born between 1976 and 1985 were identified from the Victorian Cerebral Palsy Register which holds data pertaining to people with CP born in Victoria from 1970 onwards. Those who were known to be deceased (n=163) and those for whom no contact address could be found (n=26) were excluded.
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Selection of a general population comparison group A general population sample of 751 young North American adults, aged 18 to 25 years, who had completed The Quality of Life Instrument for Young adults (YAQOL),23 was selected for comparison with the CP sample. Sample size calculation Using a hypothesised mean of 85 and a standard deviation of 20, as published by Chen et al,23 we calculated that a sample of 237 self-reporting CP participants would enable us to find a 10% difference in mean standardised YAQOL scores between the CP and general population groups with 80% power and alpha of 0.05.
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Study procedures Using hospital medical records, an online telephone directory and public access electoral roll, tracing letters were sent to young adults and their families to ascertain whether they would be interested in receiving information about the study and could be contacted at their previously recorded address. Once confirmed, a second more detailed letter was dispatched together with the individually coded survey booklet and the consent form. Individuals who failed to return the survey within one month were given a reminder telephone call. If participants were physically unable to write their responses, they were invited to request a family member or carer to act as their scribe. If there was a significant intellectual disability, a parent or carer was asked to complete the survey as a proxy for the young adult. Outcome measures A survey booklet was compiled to gather information about QOL, gross motor function, independence in self-care, and social participation. QOL was assessed with the Quality of Life Instrument for Young Adults (YAQOL) across five domains: physical health, psychological well-being, social relationships, role function, and environmental context (refer to Table 1). The YAQOL was selected because it allows comparison with a normative group, and it is argued that the
ACCEPTED MANUSCRIPT domains of QOL that are important to people without disabilities would also be important to people with a disability. Domain and total scores range from 0-100, with higher scores indicating better QOL. Testing of the instrument on a general population sample of 751 young adults provided evidence for its construct validity and internal consistency (Cronbach’s α = 0.63-0.88).23
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Gross motor function was classified by the participants or their carer using the Gross Motor Function Classification System (GMFCS).24 The GMFCS is widely used for children with CP but has also been shown to be reliable and valid for classifying the gross motor function of adults.25, 26 The family report version27 of the GMFCS was used for this study. Results from this version are highly consistent with those of professionals..28 Using the GMFCS, gross motor function is categorised into five levels, with level I representing the most independent gross motor function and level V the least.
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Independence in self-care activities was evaluated by the CP participants or their carers using the Barthel Index.29 The Index has ten domains, comprising feeding, bathing, grooming, dressing, bladder management, bowel management, toilet use, transfers, mobility, and ability to use stairs. The maximum total score is 100, with higher scores representing greater self-care independence. Evidence has been presented in support of its construct validity and reliability.30, 31 The self-report version has been found to be closely correlated with the professional report.32
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Data on social participation were obtained from questions about four life areas: education, employment, living situation and marital status. Yearly income described personal income from private sources, salary/wages, pensions and government allowances. Information on CP subtype and comorbidities such as epilepsy and intellectual, speech, hearing, and vision impairments was collected from the Victorian Cerebral Palsy Register.
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Statistical analysis Young adults with CP were described in terms of their level of independence in self-care activities, gross motor function, comorbidities, social participation, and QOL. QOL scores were compared between self-reporting and proxy-reported participants using independent sample t-tests and Mann-Whitney U tests for domain scores with distributions that were normal and non-normal respectively. Self-reported QOL scores for the young adults with CP were also compared with self-reported QOL scores from the general population sample using independent sample t-tests. To reduce the risk of type 1 errors, the more stringent alpha level of 0.01 was used to denote a statistically significant difference between groups.
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To evaluate associations between QOL for self-reporting participants and level of impairment, functional ability, social participation, and personal and environmental factors, univariate and multivariate linear regression analyses were performed. Using a stepwise forward approach, variables were entered into the multivariate regression models at p<0.05 and removed at p>0.10. The best predictive models were presented with p-values, standardized regression co-efficients (β), and the amount of variance explained by the regression model (R2). All analyses were performed in SPSS 13.0 for Windows.33
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Results Of the 818 potential CP participants, 534 responded to the tracing letter and verified their address. The remaining 284 were unable to be traced despite 141 tracing letters being re-sent to new addresses. Surveys and consent forms were completed by 335 young adults or their proxies, an overall participation rate of 63% of those located. Of the remaining 199 individuals (non-participants), 119 declined to participate after receiving the tracing letter or survey, and 80 agreed to participate but did not return the survey or the consent form. Participants and non-participants were similar with respect to age, gender, motor topography, and frequency of comorbidities (all p>0.05). There were relatively fewer participants than non-participants with mild motor impairment (p=0.007).
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Characteristics of the CP group The mean age of the study participants was 24.7 [s.d=2.8] years; 51% were male and 49% female. Two hundred and seven (62%) of the 335 participants self-reported their QOL. Table 2 summarises the type and severity of the impairments, levels of gross motor function, independence in self-care activities, and socio-demographic characteristics of the study participants. Half the CP participants were able to walk independently; the remainder required mobility devices such as walkers, scooters, manual or powered wheelchairs. With respect to self-care, 65% of the young adults were dependent on others for assistance, whereas 35% were classified as independent.
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Compared to the proxy group, self-reporting participants were less likely to have severe motor, intellectual, speech and visual impairments (Table 2). Self-reporting participants also had a lower frequency of epilepsy, higher levels of gross motor function, and a greater likelihood of being independent in activities of daily living (52% versus 9%). Where help was needed, self-reporting participants most often needed assistance with dressing (42%) and stairs (41%). On the other hand, independence was most commonly reported for bladder (79%) and bowel (83%) continence. For the self-report group, 61% were independent in eating and 75% were independent for transfers. For the proxy report group, 11% were independent in eating and 34% were independent in transfers. Levels of QOL As shown in Table 3, self-reporting participants had higher QOL scores than participants in the proxy group for each domain (p<0.0001). The greatest difference was in the role function domain and the least difference was in the environmental context domain. When compared with the general population sample, self-reporting participants had similar QOL scores for the social relationship and environmental context domains (p>0.05), while QOL scores were lower for the physical health, psychological well-being, and role function domains (p<0.001).
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Determinants of QOL Physical Health Domain Using multivariate modelling, the best independent predictors of QOL scores for the physical health domain in self-reporting participants were GMFCS level (p<0.0001), Barthel Index score (p<0.0001), gender (p=0.006), and epilepsy status (p=0.020). Together these accounted for 34% of the variation. Females with relatively milder impairment in gross motor function, greater independence in self-care activities, and no epilepsy were more likely to have better physical health domain scores. Motor topography, intellectual impairment, and socioeconomic status were significantly associated with physical health domain scores in the simple linear regressions, but not in the multivariate analysis.
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Psychological Domain Scores on the psychological well-being domain in self-reporting participants varied according to the level of independence in self-care activities (p=0.003). The best independent predictor identified for this domain was Barthel Index score, but only 5% of the variance in domain scores was explained by this multivariate model. Employment status and educational achievement were significantly associated with psychological well-being domain scores in the simple linear regressions, but not in the multivariate analysis.
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Social Relationship Domain Educational achievement (p=0.003), yearly income (p=0.014), and vision (p=0.034) were significant predictors of independent living on multivariate analysis. These factors together accounted for 16% of the variance in social relationship domain scores. Intellectual impairment, speech impairment, GMFCS level, Barthel Index score and social participation variables were significantly related to the social relationship domain score in14 the simple linear regressions, but not in the multivariate analysis.
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Role Function Domain Lower QOL scores in relation to performance in productive roles such as employment, education, or home duties were associated with higher GMFCS levels (p=0.005), unemployment, (p<0.0001), and intellectual impairment (p<0.0001) on multivariate modelling. These variables together explained 48% of the variance in role function domain scores. Speech and vision impairment, Barthel Index score, living situation, educational achievement and yearly income were significantly related to role function domain score in the simple linear regressions, but not in the multivariate analysis. Environmental Context Domain Using multivariate analysis, QOL in relation to the home environment, community services, and access varied according to gross motor function (p=0.042), Barthel
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Index score (p<0.0001), motor topography (p=0.034), and yearly income (p=0.041). In contrast to young adults with mildly impaired gross motor function and greater independence in self-care activities, those with higher GMFCS levels and lower levels of independence were more likely to report poorer QOL in the environmental context domain. Intellectual impairment, vision impairment, employment status, and educational achievement were also significantly associated with environmental context domain score in the simple linear regressions, but not in the multivariate analysis.
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Discussion This is the first study to document QOL in a population-based sample of young Australian adults with CP. The results indicated that 1) self-reporting participants reported higher QOL than was reported for participants by proxy; 2) young adults with CP reported lower QOL than a normative general population sample on all domains except social relationships and environmental context; and 3) mobility and functional independence were strongly associated with QOL related to physical health and employment status, but they were not related to satisfaction with social relationships.
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Proxy and Self-Reported QOL Compared to self-reporting young adults with CP, proxies reported a lower level of QOL within each domain for participants who were unable to self-report. The biggest difference was in the role function domain where proxies reported markedly lower scores than young adults who self-reported. As most of the participants by proxy (85%) did not engage in any work, education, or home duties, it would be unlikely that their proxies would report a sense of well-being from participation in these roles. There are no comparable studies of adults in the literature, but similar results have been found in QOL studies of children where self and proxy reported data were collected from the same group.34-40 Given the dynamic and subjective nature of QOL, there is often lack of agreement between self and proxy reports. Parents frequently report lower QOL than children themselves, highlighting the complexities involved in reporting someone else’s QOL.
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Levels of QOL of adults with CP Our results indicate that the QOL of self-reporting young adults with CP, although variable, was perceived as being lower than the QOL of their typically developed peers in some, but not all, domains. Self-reporting CP participants reported lower levels of QOL than their peers in the domains of physical health, psychological well-being, and role function, but similar levels in the social relationship and environmental context domains. A Norwegian population-based study of adults with CP aged 18 to 72 years produced somewhat similar findings.14 Using the Life Satisfaction Scale,15 the authors of the Norwegian study found that, compared to levels of satisfaction in a reference group, adults with CP had lower satisfaction in six of nine domains, including family life, sexual life, activities of daily living, leisure, work situation, and life as a whole, but similar levels of satisfaction with respect to economic status, contact with friends, and marital situation. Studies of QOL in children have also shown comparable levels of QOL between CP and normative samples in some domains.8, 9 For example, one study found that self-reported QOL of children with CP aged 8 to 12 years was similar to their peers without CP except for QOL relating to physical well-being and the school environment.8
ACCEPTED MANUSCRIPT Determinants of QOL This study found no association between psychological well-being and variables related to body structure and function in young adults with CP. At the activity level, functional independence was only weakly associated with psychosocial well-being in comparison to physical- and role function well-being, and there was little evidence of an association between gross motor function and psychological well-being.
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Our finding that gross motor function was only associated with the YAQOL domains of physical health, role function, and environmental context, but not to psychological well-being or social relationship, further supported findings from thirteen previous studies of children and adolescents with CP in the United States,6, 9, 18-20, 41 Canada,21, 42, 43 Europe,8 and Australia.7, 10, 44 Although nine of these studies,6, 7, 18-21, 41, 42, 44 used instruments that focus on health status or functioning when describing QOL, such as the Pediatric Quality of Life Inventory, the Health Utilities Index and the Pediatric Evaluation of Disability Inventory, their results also indicated that gross motor function was associated with physical function and functional well-being, but not psychosocial well-being. This finding was also compatible with the findings of other recent studies that used subjective QOL instruments to assess well-being. Dickinson et al8 reported that walking ability was only associated with physical well-being, and was not associated with QOL in six of the ten KIDSCREEN domains, including psychological well-being, self-perception, social support, school environment, financial resources, and social acceptance. Rosenbaum et al43 using the Quality of Life Instrument for people with developmental disabilities, found that none of the QOL domain scores (being, belonging, and becoming) varied with GMFCS level.
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Little attention has been given to the associations between functional independence and QOL for individuals with CP. The current study finding is partly supported by Majnemer’s study of children with CP.42 This study used the CHQ PF-50 and PedsQL 4.0 and demonstrated an association between functional limitation in daily living skills and physical well-being, but not psychosocial well-being. Furukawa et al16 also described lower subjective well-being in adults with CP who were dependent on others for activities of daily living compared to those who were independent. They included a wide age range (26-51y) and used the Philadelphia Geriatric Centre Morale Scale to evaluate subjective well-being rather than a multidimensional QOL instrument. The finding of strong associations between gross motor function, functional independence, and QOL in the physical- and functional-related domains highlights the importance of efforts to improve independence in self-care with ongoing rehabilitation, specially designed equipment, and personal assistance in this population.
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Contrary to the assumption that young adults with severe CP have low psychosocial well-being, it is apparent that these individuals can have good psychosocial well-being regardless of their disability. According to the World Health Organisation definition,4 QOL is a broad construct that includes perceptions of health and function rather than health or functional status per se. Furthermore, individuals who grow up with CP may not have the same life experiences as their peers, and may accept disability and functional limitation as a part of their life. Positive attitudes towards life may enable individuals with disabilities to adapt well to their limitations.11, 45 Personal attitudes, for example, establishing a balance between body, mind and spirit, were identified as one of the coping strategies used to separate disability from life quality.11 Values, goals, and expectations may be adjusted to comply with their abilities. In a study of adolescents with CP, being believed in, believing in yourself, and being accepted by others were key factors relating to happiness and success in life.45 Alternatively, perceptions may be influenced by their environment. For example, individuals attending mainstream education or in employment may establish the same standards for life quality as their able-bodied peers, whereas those in special schools or day centres, having peers with physical or learning disabilities, might set different norms.
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We found no previous studies that explored the relationship between social participation and QOL in adults with CP. In this study of 20-30 year olds, employment status was associated with each QOL domain except psychological well-being. This finding is consistent with previous research showing that life satisfaction was influenced by employment status in young adults with developmental and physical disabilities.12, 17 These findings highlight the benefit of employment and employment-related support services along with occupational training and opportunities for volunteer work as an alternative. Community networks, government policies, and assistive technologies are important means of increasing employment opportunities.46
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Limitations of this study include the fact that the study sample consisted of slightly fewer young adults at the mild end of the severity spectrum than in the CP population and this may have affected the comparison between CP participants and the comparison sample. A further limitation was that proxy-report data were not collected for self-reporting participants and we were unable to fully investigate differences between self and proxy reported QOL. Also, due to the lack of Australian normative QOL data, comparison QOL data were taken from a general population sample of young adults aged 18 to 25 years old in the United States. It is possible that cultural differences contribute to the differences observed between the study and comparison groups. The YAQOL has not been previously used in Australia, and time and cost constraints prevented the recruitment of a comparison sample from the Australian general population.
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Conclusion Previous studies exploring QOL in CP have focused on children and adolescents. This is the first study to examine young adults’ QOL and the relationships between QOL, level of impairment, function, social participation, and personal and environmental factors. The results suggest that self-reported QOL in young adults with CP is lower than QOL reported by able-bodied young adults, except in the areas of social relationships and environmental context. Consistent with this finding, mobility appears to contribute to some aspects of self-reported QOL in young adults with CP but not to QOL scores for social relationships and psychological well-being.
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ACCEPTED MANUSCRIPT Table 1. Descriptions of domains and scales in the YAQOL Domains
Scales
Descriptions
Physical health
Physical health
Measures overall physical health, limitation due to chronic pain, chronic physical problems, disturbance due to the illness, and energy level in the previous year
Negative Affect Social relationship
Social Support
Role function
Role Stress
Role Autonomy Self-Role Morale
Assesses the extent of stress in the individual’s major productive roles in the settings of employment, education or home duties Assesses the extent of influence over role organization, scheduling, and decisions Assesses the degree of role satisfaction and consistency with long term goals Assesses the degree of role satisfaction and consistency with long term goals in co-workers or other students
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Other’s Role Morale
Environmental context
Measures the extent of contact and quality of relationships with friends Measures the quality of relationships with a partner or with close friends
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Religious Orientation
Examines expectations for the future and recognition of the long-term goals Examines the importance of the religious belief and frequency of service attendance Examines negative, depressed and anxious feelings in life
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Psychological well-being Positive Outlook
Home
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Community
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Mobility
Resources
Evaluates comfort, safety, quietness, and privacy at home Evaluates social and service aspects of the community Evaluates ease of access to work, school, shopping, leisure and social activities Evaluates the ability to access medical care and financial support
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Table 2: Characteristics of the CP study group by reporting status, excluding participants with missing data. Self-report Proxy-report Total CP group group study group n=207 n=128 n=335 n (%) n (%) N (%) GMFCS (n=335) Level I 61 (29.5) 9 (7.0) 70 (20.9) Level II 78 (37.7) 29 (22.7) 107 (31.9) Level III 18 (8.7) 16 (12.5) 34 (10.1) Level IV 31 (15.0) 31 (24.2) 62 (18.5) Level V 19 (9.2) 43 (33.6) 62 (18.5) Cerebral palsy subtype (n=335) Spastic monoplegia/hemiplegia 84 (40.6) 24 (18.7) 108 (32.2) Spastic diplegia 54 (26.1) 22 (17.2) 76 (22.7) Spastic triplegia/quadriplegia 50 (24.1) 65 (50.8)) 115 (34.4) Ataxia/Dyskinesia/Hypotonia 19 (9.2) 17 (13.3) 36 (10.7) Intellectual impairment (n=317) None (IQ≥70) 141 (71.6) 10 (8.3) 151 (47.6) Mild (50≤IQ<70) 35 (17.8) 18 (15.0) 53 (16.7) Moderate (30≤IQ<50) 16 (8.1) 38 (31.7) 54 (17.0) Severe (IQ<30) 5 (2.5) 54 (45.0) 59 (18.7) Speech impairment (n=315) None 129 (66.2) 9 (7.5) 138 (43.8) Some 47 (24.1) 41 (34.2) 88 (27.9) Non-verbal 19 (9.7) 70 (58.3) 89 (28.3) Hearing impairment (n=313) None 178 (91.8) 107 (89.9) 285 (91.1) Some 13 (6.7) 11 (9.2) 24 (7.7) Bilateral deafness 3 (1.5) 1 (0.8) 4 (1.3) Vision impairment (n=313) None 75 (39.1) 24 (19.8) 99 (31.6) Strabismus only 27 (14.1) 11 (9.1) 38 (12.1) Some 90 (46.9) 83 (68.6) 173 (55.3) Functionally blind 0 (0.0) 3 (2.5) 3 (1.0) Epilepsy (n=329) None/resolved 160 (79.6) 42 (32.8) 202 (61.4) Epilepsy 41 (20.4) 86 (67.2) 127 (38.6) Self-care independence (n=335) Independent (Barthel score 95-100) 107 (51.7) 12 (9.4) 119 (35.5) Assistance needed (Barthel score 60-94) 50 (24.2) 24 (18.8) 74 (22.1) Dependent (Barthel score 0-59) 50 (24.2) 92 (71.9) 142 (42.4) Education level achieved (n=334) Below Year 12 51 (24.8) 116 (90.6) 167 (50.5) Completed secondary 40 (19.4) 8 (6.3) 48 (14.4) Tertiary 115 (55.8) 4 (3.1) 119 (35.6) Employment status (n=333) Employed 108 (52.7) 13 10.2 121 (36.3)
ACCEPTED MANUSCRIPT 49 59 97 33 64
(23.9) (28.8) (47.3) (16.1) (31.2)
6 7 115 1 114
4.7 5.5 89.8 0.8 89.0
(16.5) (19.8) (63.7) (10.2) (53.5)
70 (20.9) 219 (65.4) 46 (13.7)
23 11.1 184 88.9
2 1.6 126 98.4
25 (7.5) 310 (92.5)
RI PT
3 2.3 93 72.7 32 25.0
M AN U TE D EP AC C
55 66 212 34 178
67 32.3 126 60.9 14 6.8
SC
Full-time employment Part-time employment Unemployed Currently job seeking Never sought employment Living situation (n=335) Living independently Living with parents Community residential unit Marital status (n=335) Married or partnered Single
ACCEPTED MANUSCRIPT Table 3: Comparisons between self-report and proxy report CP groups and between the self-report CP group and general population sample for mean YAQOL domain scores
Environmental context, mean (SD)
Proxy-report General group population n=128 sample (n=751) 58.87 (22.0) 46.62 (21.6) 72.05 (16.3) 53.31 (16.9) 43.78 (18.9)
67.79 (12.8)
77.28 (15.8) 67.52 (14.8)
78.61 (10.8)
50.33 (31.5) 9.26 (22.7)
60.83 (14.4)
72.01 (12.7) 64.49 (11.4)
T statistic
P value for group differences
ta=4.95 tb=9.44 ta=4.66 tb=13.43 ta=5.58 tb=1.40
pa<0.0001 pb<0.0001 pa<0.0001 pb<0.0001 pa<0.0001 pb=0.1627 pa<0.0001 * pb<0.0001 pa<0.0001 pb =0.0325
RI PT
Physical health, mean (SD) Psychological wellbeing, mean (SD) Social relationship, mean (SD) Role function, mean (SD)
Self-report group n=207
SC
QOL scores
73.91 (10.8)
tb=6.90 ta=5.46 tb=2.14
AC C
EP
TE D
M AN U
QOL scores range from 0 to 100, higher score indicates better QOL; SD: standard deviation; ta, pa values refer to differences between self and proxy report groups, tb, pb values refer to the differences between the self-report CP group and general population comparison group, calculated using Independent-Sample t tests. *:p value was calculated using Mann-Whitney U test because the role function scores in proxy participants were not normally distributed.
1
ACCEPTED MANUSCRIPT
Table 4: Determinants of YAQOL domain and overall scores for young adults with CP (self-reporting participants, summary of multivariate linear regressions)
Psychological well being
ß (standard coefficients)
p
ß (standard coefficients)
p
Social relationship ß (standard coefficients)
Body structure and function -0.05
0.394
0.04
0.560
-0.10
Motor topography
-0.09
0.104
-0.02
0.673
-0.09
Intellectual impairment
-0.11
0.090
-0.00
0.994
-0.10
Speech impairment
-0.03
0.658
-0.09
0.308
Hearing impairment
-0.08
0.183
-0.01
0.884
Vision impairment
-0.03
0.638
-0.03
0.729
Presence of epilepsy
-0.14
0.020 *
0.00
0.981
TE D
Activity
p
0.146
ß (standard coefficients)
p
0.334
0.295
-0.06
0.542
-0.16
0.034 *
0.225
-0.22
-0.07
0.341
<0.0001 ***
-0.12
0.104
-0.05
0.422
0.04
0.550
-0.10
0.169
-0.05
0.354
0.08
0.159
-0.16
0.034 *
-0.04
0.546
-0.06
0.442
-0.06
0.424
-0.02
0.739
-0.04
0.513
-0.16
0.042 *
0.00
0.962
-0.06
0.475
-0.16
0.005 **
Functional independence (BI)
0.40
<0.0001***
0.23
0.003 **
0.07
0.395
0.06
0.411
Living situation
-0.04
0.562
Marital status
0.00
0.982
Age
0.06
0.280
Gender
-0.17
Educational achievement
-0.05
Personal factors
-0.08
0.360
-0.08
0.344
-0.44
-0.01
0.953
-0.11
0.163
-0.02
AC C
0.907
EP
<0.0001 ***
-0.01
p
0.07
-0.35
Employment status
ß (standard coefficients)
0.719
Mobility (GMFCS level)
Participation
Environmental context
-0.02
M AN U
Type of CP
Role function
RI PT
Predictor variables
SC
Physical health
-0.08
0.284
-0.07
0.353
0.00
0.985
-0.06
0.006 **
-0.07
0.385
0.452
-0.07
0.353
0.36
<0.0001 ***
-0.02
0.834
0.767
0.05
0.439
-0.07
0.241
-0.10
0.111
0.371
-0.13
0.025
-0.05
0.412
0.06
0.401
0.06
0.284
-0.09
0.143
-0.23
0.003 **
-0.03
0.621
-0.03
0.679
<0.0001***
ACCEPTED MANUSCRIPT
Yearly income
-0.07
0.302
-0.04
0.666
-0.19
-0.05
0.445
-0.16
0.041 *
0.01
0.887
0.06
0.416
0.04
0.554
0.07
0.260
0.06
0.306
Healthcare needs met
-0.05
0.396
-0.09
0.263
-0.07
0.352
-0.27
-0.07
0.386
Location of residence
0.01
0.780
-0.01
0.949
-0.04
0.579
0.03
0.663
0.014 *
Transferred to adult services
R=0.42, R2=0.18, adjusted R2 =0.16, p <0.0001
SC
R=0.60, R2=0.36, adjusted R=0.23, R2=0.05, R2 =0.34 adjusted R2 =0.05 p p <0.0001 =0.001
RI PT
Environmental factors
-0.05
<0.0001 *** 0.427
R=0.71, R2=0.50, adjusted R=0.44, R2=0.19, adjusted R2 =0.17 R2 =0.48 p <0.0001 p <0.0001
2
Analyses were done for 207 self-reporting young adults; *: p < 0.05; **: p < 0.01; ***: p <0.001; R : the coefficient of determination, calculated using the multivariate linear
AC C
EP
TE D
M AN U
regression analysis; GMFCS: Gross Motor Function Classification System; the BI: the Barthel Index; p < 0.05 are presented in bold.