Journal of Pediatric Rehabilitation Medicine: An Interdisciplinary Approach 6 (2013) 121–127 DOI 10.3233/PRM-130252 IOS Press
121
Measuring quality of life of Finnish children with cerebral palsy Sanna Bölinga,∗ , Varho Tarjaa , Mäenpää Helenab, Forsten Wivib , Autti-Rämö Ilonac and Haataja Leenad a
Neuropediatric Unit of Turku Health Care and Social Services, Turku, Finland University Hospitals for Children and Adolescents, University of Helsinki, Helsinki, Finland c Research Department, Social Insurance Institution of Finland, Helsinki, Finland d University Hospitals for Children and Adolescents, University of Turku, Turku, Finland b
Accepted 29 July 2013
Abstract. PURPOSE: The purpose of the study was to examine the quality of life (QOL) of Finnish children with cerebral palsy (CP), both from the child’s and the caregiver’s point of view, and to analyze the effect of background factors on QOL. METHODS: This study is a part of a national CP research project. The study is based on validated questionnaires measuring QOL (CP QOL-Child). 128 questionnaires were sent to caregivers who had a 4 to 12 year-old child with CP. Children between 9 and 12 years were asked to fill in the child-self-report version. RESULTS: Responses were obtained from 78 guardians and 27 children, with a response rate of 61% in both cases. The overall QOL was reported to be good in Finnish children with CP. The correlation of QOL scores between the caregivers and children was good (n = 25, r = 0.687, p < 0.001), except in the domain of pain and the impact of disability. Parental estimates were consistently lower in all domains. Regarding the background factors, all the functional classification scales were associated inversely with QOL in both groups. CONCLUSION: Despite the good overall QOL, CP is perceived to limit participation. Pain impairs QOL, and pain symptoms should be systematically considered at every follow-up visit. Keywords: Cerebral palsy, child, quality of life, questionnaire
1. Introduction Cerebral palsy (CP) represents a group of permanent disorders in the development of movement and posture that limit activity. It is caused by non-progressive disturbances that occurred in the developing fetal or infant brain. The motor disorders of cerebral palsy are often accompanied by epilepsy and impairments in sensation, perception, cognition, and communication. In addition to fine and gross motor problems, one may have specific deficits in feeding and swallowing, co∗ Corresponding author: Böling Sanna, MA, Pt. Virmuntie 26 as 17, 20540 Turku, Finland. Fax: +358 2 2626899; Email: sanna. [email protected].
ordinated eye movements, articulation of speech, and secondary problems with behavior and musculoskeletal function [1]. CP may affect an individual’s everyday life to the extent that it also has significant impact on their perceived quality of life (QOL). Kiviranta and Jokinen [2] conducted a large study of the services available for disabled adolescents, mainly with CP, in Finland. The study covered some aspects of wellbeing such as school arrangements, child’s health, and the caregivers’ opinions about children’s participation, but it did not focus directly on QOL. The QOL can be assessed by a generic or condition specific, global or health related measure [3]. Whether a structured questionnaire can capture the essentials of quality of life can always be questioned.
c 2013 – IOS Press and the authors. All rights reserved 1874-5393/13/$27.50
122
S. Böling et al. / Measuring quality of life of Finnish children with cerebral palsy
Rosenbaum et al. have assessed QOL and healthrelated QOL (HRQOL) of adolescents with CP. They found that despite being conceptually related, QOL and HRQOL are different constructs, and the results need to be interpreted with caution [1]. The type of QOL instrument that should be used for different purposes has been widely debated [3]. The instrument used in this study (CP QOL-Child questionnaire) was developed in Australia, is based on the International Classification of Functioning, Disability, and Health (ICF) framework, and is condition specific. It has previously been used by Shelly et al. [4] who reported that children with CP may have a high psychosocial QOL score even if they have limited physical functioning. In the study by Dickinson et al. [5] the QOL of children with CP was compared to the QOL of children without CP using the KIDSCREEN questionnaire. They demonstrate that most children aged 8–12 years with CP had similar QOL compared to other children. Waters et al. [6] compared the psychometric properties of tools to measure QOL of children with CP (KIDSCREEN, Child Health Questionnaire, CHQ, and CP QOL child). It was concluded that the CP QOL questionnaire filled in by children has good psychometric properties and is a valid instrument. This is the first time that the QOL of Finnish children with CP has been evaluated using a CP-specific instrument. The aim of this study is to examine the QOL of children with CP in the ICF framework, and to study the associations between background factors and QOL domains.
2. Methods 2.1. Participants This study is a part of a nationwide research project that aims to find reliable, valid, and clinically feasible sets of outcome measures for children and adolescents with CP via a multi professional working model. One hundred twenty eight families who had given their consent and had a 4 to 12 year-old child with CP were sent a Cerebral Palsy Quality of Life Questionnaire for Children (CP QOL-Child; caregiver proxyversion), together with a cover letter. Children between 9 and 12 years were asked to fill in a separate questionnaire themselves (CP QOL-Child; child self-report). Caregivers were asked to consider the child’s ability to fill in the questionnaire. The survey was carried out
during a three-month period from November 2010 to February 2011 in the Turku Hospital District of Southwest Finland (Neuropediatric units of Turku University Hospital and Turku city) and the Helsinki and Uusimaa Hospital District (Neuropediatric Unit of Helsinki University Hospital). 2.2. Questionnaire The CP QOL-Child measures the quality of life of children with CP. Its development has been described elsewhere [7]. The caregiver-proxy version assesses seven domains of QOL including “social wellbeing and acceptance”, “functioning”, “participation and physical health”, “emotional wellbeing and selfesteem”, “access to services”, “pain and impact of disability”, and “family health”. The child self-report version is aimed at children aged 9–12 years and assesses the same domains, except access to services and family health. Reliability tests were performed on the different domains. For the caregiver proxy-report, reliability (α) ranged from 0.72 to 0.89, and for the childrenproxy report reliability (α) ranged from 0.76 to 0.94. The primary caregiver proxy form (caregivers of children aged 4–12 years [7]) comprises 66 items and the child self-report form comprises 52 items. Most of the items begin with the question: ‘How do you think your child feels about . . . ?’ with a nine-point rating scale, where 1 = very unhappy, 3 = unhappy, 5 = neither happy nor unhappy, 7 = happy, and 9 = very happy. The rest of the items begin with a question like ‘How does your child feel about the amount of pain that they have?’, where 1 = not upset at all to 9 = very upset. The questionnaire was translated into Finnish from the original English questionnaire according to the instructions given in the manual [7], and with written consent of the authors. The layout was set according to the original questionnaire by Sanna Böling. 2.3. Classification of functional abilities As a part of the ongoing multi-professional national research project, all of the participating children had been classified by Gross Motor Function Classification system (GMFCS [8]), Manual Ability Classification System (MACS [9]), and Communication Function Classification System (CFCS [10]). 2.4. Statistical analysis Statistical analyzes were conducted with the SPSS (Statistical Package for Social Sciences Program) vers-
S. Böling et al. / Measuring quality of life of Finnish children with cerebral palsy
123
Table 1 Characteristics of the study population Child self-report n = 27 74% 26%
Caregivers proxy-report n = 78 64% 36%
Spastic quadriplegia Spastic diplegia Spastic hemiplegia Dyskinetic CP Other
0% 37% 56% 4% 4%
5% 36% 46% 6% 6%
GMFCS∗)
I II III IV–V
67% 15% 7% 11%
56% 21% 8% 16%
MACS∗∗)
I II III IV–V
44% 44% 4% 7%
44% 31% 17% 8%
CFCS∗∗∗)
I II III IV–V
92% 4% 4% 0%
71% 10% 8% 10%
Type of school/day care
Day care Special education group of day care Not in day care Mainstream school (MS) Special education class in MS School with special needs
0% 0% 0% 59% 19% 22%
21% 10% 3% 40% 13% 13%
Background factor Gender
Value Male Female
Diagnosis
∗ Gross
Motor function Classification System; ∗∗) Manual Ability Classification System; ∗∗∗) Communication Function Classification System.
ion 18. First, the reliability of the sum variables was examined using the Cronbach alpha test, with the lowest acceptable value of 0.6. All sum variables passed this test. Mean values and standard deviations were used as main indicators. The scoring of the QOL questionnaires was performed according the CP-QOL manual [7]. The raw score was converted into a 0–100 scale. The responses were classified into seven sum variables: “Social wellbeing and acceptance”, “Functioning”, “Participation and physical health”, “Emotional wellbeing and self esteem”, “Pain and impact of disability”, “Access to services”, and “Family health”, all measuring different aspects of quality of life. An overall variable denoted “Quality of life” and the sum of all variables were also considered. The following factors were considered in the data analysis: CP diagnosis according to the ICD-10, gender, GMFCS level, MACS level, CFCS level, and school or daycare arrangements (day care, special group of day care, not in daycare, mainstream school, special education class in mainstream school, and school with special needs). The effect of the chosen background factors on different QOL domains was examined by analysis of variance, with the significance
level p < 0.05. The responses of children and caregivers were compared using a paired samples t-test and Pearson‘s correlation coefficient test. Finally, the available background factors of non-responsive families were compared against responsive families using a cross-tabulation test and an independent samples t-test. 2.5. Ethical approval The national research project protocol was approved by the Ethics Review Committees of the Hospital District of the South-West Finland and the Hospital District of Helsinki-Uusimaa in 2008. All the caregivers gave written consent after being given oral and written information concerning the study.
3. Results Seventy-eight (n = 78) caregivers of children aged 4 to 12 years with CP (with median 7 years), completed the Cerebral Palsy Quality of Life Questionnaire for Caregivers. Twenty-seven (n = 27) children aged 9 to 12 years (with median 11 years), completed the Cerebral Palsy Quality of Life Questionnaire for
124
S. Böling et al. / Measuring quality of life of Finnish children with cerebral palsy Table 2 The factors with statistically significant impact on QOL domains in the child self-reports (n = 27)
Diagnosis Spastic diplegia (n = 10) Spastic hemiplegia (n = 15) Dyskinetic (n = 1) Other (n = 1)
Participation and p Emotional wellbeing physical health and self esteem mean std 0.049 78.9 0.029 83.9 10.2 76.1 11.4 85.8 14.1 84.1 13.8 59.6 – 58.8 –‘ 54.8 – 48.6 –
MACS level I (n = 12) II (n = 12) III (n = 1) IV–V (n = 2)
87.6 83.9 68.3 57.2
8.8 15.0 – 3.4
0.017 78.9 83.8 80.1 56.3 53.7
10.2 14.7 – 7.2
School arrangement Mainstream (n = 16) Special education ( n = 5) School with special needs (n = 6)
88.3 80.0 71.3
9.0 14.4 18.9
0.029 78.9 85.3 72.5 62.3
10.4 15.1 17.1
Functioning
p
p
0.013
0.015 85.9 87.2 92.1 77.1
9.3 9.7 11.3
Quality of life
81.9 85.3 82.3 69.1 65.1
7.2 11.7 – 3.6
0.044 81.9 85.5 80.8 73.1
8.3 9.5 13.5
p
0.043
0.044
Table 3 The factors with statistically significant impact on QOL domains in the caregiver proxy-reports (n = 78) p 0.003
GMFCS level I (n = 43) II (n = 16) III (n = 6) IV–V (n = 12)
mean 77.6 71.5 61.9 58.4
std. 10.3 11.0 17.0 11.2
121
Measuring quality of life of Finnish children with cerebral palsy Sanna Bölinga,∗ , Varho Tarjaa , Mäenpää Helenab, Forsten Wivib , Autti-Rämö Ilonac and Haataja Leenad a
Neuropediatric Unit of Turku Health Care and Social Services, Turku, Finland University Hospitals for Children and Adolescents, University of Helsinki, Helsinki, Finland c Research Department, Social Insurance Institution of Finland, Helsinki, Finland d University Hospitals for Children and Adolescents, University of Turku, Turku, Finland b
Accepted 29 July 2013
Abstract. PURPOSE: The purpose of the study was to examine the quality of life (QOL) of Finnish children with cerebral palsy (CP), both from the child’s and the caregiver’s point of view, and to analyze the effect of background factors on QOL. METHODS: This study is a part of a national CP research project. The study is based on validated questionnaires measuring QOL (CP QOL-Child). 128 questionnaires were sent to caregivers who had a 4 to 12 year-old child with CP. Children between 9 and 12 years were asked to fill in the child-self-report version. RESULTS: Responses were obtained from 78 guardians and 27 children, with a response rate of 61% in both cases. The overall QOL was reported to be good in Finnish children with CP. The correlation of QOL scores between the caregivers and children was good (n = 25, r = 0.687, p < 0.001), except in the domain of pain and the impact of disability. Parental estimates were consistently lower in all domains. Regarding the background factors, all the functional classification scales were associated inversely with QOL in both groups. CONCLUSION: Despite the good overall QOL, CP is perceived to limit participation. Pain impairs QOL, and pain symptoms should be systematically considered at every follow-up visit. Keywords: Cerebral palsy, child, quality of life, questionnaire
1. Introduction Cerebral palsy (CP) represents a group of permanent disorders in the development of movement and posture that limit activity. It is caused by non-progressive disturbances that occurred in the developing fetal or infant brain. The motor disorders of cerebral palsy are often accompanied by epilepsy and impairments in sensation, perception, cognition, and communication. In addition to fine and gross motor problems, one may have specific deficits in feeding and swallowing, co∗ Corresponding author: Böling Sanna, MA, Pt. Virmuntie 26 as 17, 20540 Turku, Finland. Fax: +358 2 2626899; Email: sanna. [email protected].
ordinated eye movements, articulation of speech, and secondary problems with behavior and musculoskeletal function [1]. CP may affect an individual’s everyday life to the extent that it also has significant impact on their perceived quality of life (QOL). Kiviranta and Jokinen [2] conducted a large study of the services available for disabled adolescents, mainly with CP, in Finland. The study covered some aspects of wellbeing such as school arrangements, child’s health, and the caregivers’ opinions about children’s participation, but it did not focus directly on QOL. The QOL can be assessed by a generic or condition specific, global or health related measure [3]. Whether a structured questionnaire can capture the essentials of quality of life can always be questioned.
c 2013 – IOS Press and the authors. All rights reserved 1874-5393/13/$27.50
122
S. Böling et al. / Measuring quality of life of Finnish children with cerebral palsy
Rosenbaum et al. have assessed QOL and healthrelated QOL (HRQOL) of adolescents with CP. They found that despite being conceptually related, QOL and HRQOL are different constructs, and the results need to be interpreted with caution [1]. The type of QOL instrument that should be used for different purposes has been widely debated [3]. The instrument used in this study (CP QOL-Child questionnaire) was developed in Australia, is based on the International Classification of Functioning, Disability, and Health (ICF) framework, and is condition specific. It has previously been used by Shelly et al. [4] who reported that children with CP may have a high psychosocial QOL score even if they have limited physical functioning. In the study by Dickinson et al. [5] the QOL of children with CP was compared to the QOL of children without CP using the KIDSCREEN questionnaire. They demonstrate that most children aged 8–12 years with CP had similar QOL compared to other children. Waters et al. [6] compared the psychometric properties of tools to measure QOL of children with CP (KIDSCREEN, Child Health Questionnaire, CHQ, and CP QOL child). It was concluded that the CP QOL questionnaire filled in by children has good psychometric properties and is a valid instrument. This is the first time that the QOL of Finnish children with CP has been evaluated using a CP-specific instrument. The aim of this study is to examine the QOL of children with CP in the ICF framework, and to study the associations between background factors and QOL domains.
2. Methods 2.1. Participants This study is a part of a nationwide research project that aims to find reliable, valid, and clinically feasible sets of outcome measures for children and adolescents with CP via a multi professional working model. One hundred twenty eight families who had given their consent and had a 4 to 12 year-old child with CP were sent a Cerebral Palsy Quality of Life Questionnaire for Children (CP QOL-Child; caregiver proxyversion), together with a cover letter. Children between 9 and 12 years were asked to fill in a separate questionnaire themselves (CP QOL-Child; child self-report). Caregivers were asked to consider the child’s ability to fill in the questionnaire. The survey was carried out
during a three-month period from November 2010 to February 2011 in the Turku Hospital District of Southwest Finland (Neuropediatric units of Turku University Hospital and Turku city) and the Helsinki and Uusimaa Hospital District (Neuropediatric Unit of Helsinki University Hospital). 2.2. Questionnaire The CP QOL-Child measures the quality of life of children with CP. Its development has been described elsewhere [7]. The caregiver-proxy version assesses seven domains of QOL including “social wellbeing and acceptance”, “functioning”, “participation and physical health”, “emotional wellbeing and selfesteem”, “access to services”, “pain and impact of disability”, and “family health”. The child self-report version is aimed at children aged 9–12 years and assesses the same domains, except access to services and family health. Reliability tests were performed on the different domains. For the caregiver proxy-report, reliability (α) ranged from 0.72 to 0.89, and for the childrenproxy report reliability (α) ranged from 0.76 to 0.94. The primary caregiver proxy form (caregivers of children aged 4–12 years [7]) comprises 66 items and the child self-report form comprises 52 items. Most of the items begin with the question: ‘How do you think your child feels about . . . ?’ with a nine-point rating scale, where 1 = very unhappy, 3 = unhappy, 5 = neither happy nor unhappy, 7 = happy, and 9 = very happy. The rest of the items begin with a question like ‘How does your child feel about the amount of pain that they have?’, where 1 = not upset at all to 9 = very upset. The questionnaire was translated into Finnish from the original English questionnaire according to the instructions given in the manual [7], and with written consent of the authors. The layout was set according to the original questionnaire by Sanna Böling. 2.3. Classification of functional abilities As a part of the ongoing multi-professional national research project, all of the participating children had been classified by Gross Motor Function Classification system (GMFCS [8]), Manual Ability Classification System (MACS [9]), and Communication Function Classification System (CFCS [10]). 2.4. Statistical analysis Statistical analyzes were conducted with the SPSS (Statistical Package for Social Sciences Program) vers-
S. Böling et al. / Measuring quality of life of Finnish children with cerebral palsy
123
Table 1 Characteristics of the study population Child self-report n = 27 74% 26%
Caregivers proxy-report n = 78 64% 36%
Spastic quadriplegia Spastic diplegia Spastic hemiplegia Dyskinetic CP Other
0% 37% 56% 4% 4%
5% 36% 46% 6% 6%
GMFCS∗)
I II III IV–V
67% 15% 7% 11%
56% 21% 8% 16%
MACS∗∗)
I II III IV–V
44% 44% 4% 7%
44% 31% 17% 8%
CFCS∗∗∗)
I II III IV–V
92% 4% 4% 0%
71% 10% 8% 10%
Type of school/day care
Day care Special education group of day care Not in day care Mainstream school (MS) Special education class in MS School with special needs
0% 0% 0% 59% 19% 22%
21% 10% 3% 40% 13% 13%
Background factor Gender
Value Male Female
Diagnosis
∗ Gross
Motor function Classification System; ∗∗) Manual Ability Classification System; ∗∗∗) Communication Function Classification System.
ion 18. First, the reliability of the sum variables was examined using the Cronbach alpha test, with the lowest acceptable value of 0.6. All sum variables passed this test. Mean values and standard deviations were used as main indicators. The scoring of the QOL questionnaires was performed according the CP-QOL manual [7]. The raw score was converted into a 0–100 scale. The responses were classified into seven sum variables: “Social wellbeing and acceptance”, “Functioning”, “Participation and physical health”, “Emotional wellbeing and self esteem”, “Pain and impact of disability”, “Access to services”, and “Family health”, all measuring different aspects of quality of life. An overall variable denoted “Quality of life” and the sum of all variables were also considered. The following factors were considered in the data analysis: CP diagnosis according to the ICD-10, gender, GMFCS level, MACS level, CFCS level, and school or daycare arrangements (day care, special group of day care, not in daycare, mainstream school, special education class in mainstream school, and school with special needs). The effect of the chosen background factors on different QOL domains was examined by analysis of variance, with the significance
level p < 0.05. The responses of children and caregivers were compared using a paired samples t-test and Pearson‘s correlation coefficient test. Finally, the available background factors of non-responsive families were compared against responsive families using a cross-tabulation test and an independent samples t-test. 2.5. Ethical approval The national research project protocol was approved by the Ethics Review Committees of the Hospital District of the South-West Finland and the Hospital District of Helsinki-Uusimaa in 2008. All the caregivers gave written consent after being given oral and written information concerning the study.
3. Results Seventy-eight (n = 78) caregivers of children aged 4 to 12 years with CP (with median 7 years), completed the Cerebral Palsy Quality of Life Questionnaire for Caregivers. Twenty-seven (n = 27) children aged 9 to 12 years (with median 11 years), completed the Cerebral Palsy Quality of Life Questionnaire for
124
S. Böling et al. / Measuring quality of life of Finnish children with cerebral palsy Table 2 The factors with statistically significant impact on QOL domains in the child self-reports (n = 27)
Diagnosis Spastic diplegia (n = 10) Spastic hemiplegia (n = 15) Dyskinetic (n = 1) Other (n = 1)
Participation and p Emotional wellbeing physical health and self esteem mean std 0.049 78.9 0.029 83.9 10.2 76.1 11.4 85.8 14.1 84.1 13.8 59.6 – 58.8 –‘ 54.8 – 48.6 –
MACS level I (n = 12) II (n = 12) III (n = 1) IV–V (n = 2)
87.6 83.9 68.3 57.2
8.8 15.0 – 3.4
0.017 78.9 83.8 80.1 56.3 53.7
10.2 14.7 – 7.2
School arrangement Mainstream (n = 16) Special education ( n = 5) School with special needs (n = 6)
88.3 80.0 71.3
9.0 14.4 18.9
0.029 78.9 85.3 72.5 62.3
10.4 15.1 17.1
Functioning
p
p
0.013
0.015 85.9 87.2 92.1 77.1
9.3 9.7 11.3
Quality of life
81.9 85.3 82.3 69.1 65.1
7.2 11.7 – 3.6
0.044 81.9 85.5 80.8 73.1
8.3 9.5 13.5
p
0.043
0.044
Table 3 The factors with statistically significant impact on QOL domains in the caregiver proxy-reports (n = 78) p 0.003
GMFCS level I (n = 43) II (n = 16) III (n = 6) IV–V (n = 12)
mean 77.6 71.5 61.9 58.4
std. 10.3 11.0 17.0 11.2
