Comment
Quality of life in adolescents with cerebral palsy groups of adolescents were generally less than 0·5 SD; impairments were significantly associated with lower adolescent QoL in only three of ten domains (Moods and emotions, Autonomy, and Social support and peers). Adolescents with cerebral palsy had significantly lower QoL than matched controls in the general population in only one domain—Social support and peers (mean difference –2·7 [0·25 SD]; 95% CI –4·3 to –1·4). Similar findings of stability of QoL among school-age children, both with cerebral palsy and typically developing peers, have been reported in several other large studies.9,10 Colver and colleagues’ report adds to the growing recognition that QoL reflects the unique internal milieu and perceived possibilities of each child, with or without cerebral palsy. An important point of Colver and colleagues’ study6 is that there are modifiable factors that can positively change QoL in children with cerebral palsy. These factors include parenting stress, psychological problems, and most notably pain. Pain in childhood or adolescence was strongly associated with low adolescent QoL (up to 6 points lower) in eight domains. Pain in children with cerebral palsy is a
www.thelancet.com Published online October 7, 2014 http://dx.doi.org/10.1016/S0140-6736(14)61599-3
Published Online October 7, 2014 http://dx.doi.org/10.1016/ S0140-6736(14)61599-3 See Online/Articles http://dx.doi.org/10.1016/ S0140-6736(14)61229-0
A Hoon MD
Child development is dynamic and multidimensional, with genetic and epigenetic building blocks upon which the complex effects of environment are overlaid (ie, nurture through nature).1 In children with cerebral palsy and other disabilities, developmental outcome is more than the result of medical treatment, and includes the child’s communication, coping, and problem-solving skills; educational opportunities; family adaptation; and service delivery2—factors all shown by quality of life (QoL). WHO’s definition of QoL as “the individual’s perception of their position in life in the context of the culture and value system in which they live, and in relation to their goals, expectations, standards, and concerns”3 represents the validation of the unique value of every person, typically developing or with a disability. The University of Toronto Centre for Health Promotion defines QoL as “the degree to which a person enjoys the important possibilities of his or her life”.4 This definition emphasises that the quality of a child’s life is interconnected with the nature of caregiver care and the environment in which each child lives. Increasingly, QoL is valued as an important addition to standard biomedical reports, and as a measure of clinical progress and research outcome.5 The large longitudinal cohort study of Allan Colver and colleagues,6 reported in The Lancet, examines how selfreported QoL in adolescents with cerebral palsy varies with impairment, how it compares with the perceived QoL of peers in the general population, and to what extent childhood factors amenable to intervention— pain, psychological problems, and parenting stress— predict adolescent QoL. Young people reported their QoL using KIDSCREEN, a generic QoL instrument with sound psychometric properties designed for children and adolescents.7 This study was done as part of the larger European SPARCLE study whose strengths include randomly selected sampling from population-based cerebral palsy registers in nine European regions.8 Colver and colleagues report outcomes from a 5-year longitudinal follow-up (SPARCLE2) of 355 adolescents aged 13–17 years who were able to self-report their QoL (longitudinal sample) and a cross-sectional sample including 76 additional adolescents (n=431).6 The findings are encouraging in that average differences in QoL between the least-able and most-able
1
Comment
common problem. In a 2012 article,11 75% of children with cerebral palsy were reported to have pain, which was present with all levels of disability, increased with age, and was linked to higher rates of behavioural problems and lower participation. Given the effects on adolescent QoL, pain assessment and management should be a priority for all children with cerebral palsy, irrespective of level of motor impairment. One caveat, which Colver and colleagues recognise, is that the study excluded children and adolescents with cerebral palsy who could not self-report their QoL because of severe learning difficulties. This group often has more severe and multiple impairments, and a parent or caregiver proxy report might yield different QoL results. Thus, no conclusions can be drawn from this study about the QoL of adolescents with severe developmental disabilities, and the challenge is to include their voices in future research. As known by people with cerebral palsy, the medical and rehabilitative community now recognises that affected individuals seek the same QoL and social participation as others, rather than just improved physical function for its own sake.12 Cerebral palsy is a lifelong disorder with interventions during childhood that might have long-term individual, family, and societal benefits. Colver and colleagues’ study6 suggests that children and adolescents with cerebral palsy may need particular help maintaining and developing peer relationships. Childhood interventions that optimise social support, health, and wellbeing may affect not only adolescent QoL, but also adult outcome by optimising each individual’s potential to adapt to, and prevent or reduce, the secondary morbidities often present.13
2
Colver and colleagues are to be commended for their work in placing QoL at the centre of the thoughts of all interested in promoting the wellbeing of people with cerebral palsy. *Alexander H Hoon Jr, Elaine E Stashinko Kennedy Krieger Institute, Baltimore, MD 21205, USA [email protected] We declare no competing interests. Copyright © Hoon et al. Open Access article distributed under the terms of CC BY. 1 2
3
4
5
6
7
8
9
10
11 12 13
Bjornsson HT, Fallin MD, Feinberg AP. An integrated epigenetic and genetic approach to common human disease. Trends Genet 2004; 20: 350–58. Ronen GM, Fayed N, Rosenbaum PL. Outcomes in pediatric neurology: a review of conceptual issues and recommendations. The 2010 Ronnie Mac Keith lecture. Dev Med Child Neurol 2011; 53: 305–12. The WHOQOL Group. The World Health Organization quality of life assessment (WHOQOL): position paper from the World Health Organization. Soc Sci Med 1995; 41: 1403–09. University of Toronto Centre for Health Promotion Quality of Life Research Unit. Quality of life model. http://sites.utoronto.ca/qol/ (accessed Aug 22, 2014). Stashinko EE, Harley LA, Steele RA, Clegg NJ. Parental perspectives on living with a child with HoPE. Am J Med Genet C Semin Med Genet 2010; 154C: 197–201. Colver A, Rapp M, Eisemann N, et al. Self-reported quality of life of adolescents with cerebral palsy: a cross-sectional and longitudinal analysis. Lancet 2014; published online Oct 7. http://dx.doi.org/10.1016/S01406736(14)61229-0. Ravens-Sieberer U, Gosch A, Rajmil L, et al. KIDSCREEN-52 quality-of-life measure for children and adolescents. Expert Rev Pharmacoecon Outcomes Res 2005; 5: 353–64. Colver A; the SPARCLE group. Study protocol: SPARCLE—a multi-centre European study of the relationship of environment to participation and quality of life of children with cerebral palsy. BMC Public Health 2006; 6: 105. Dickinson HO, Parkinson KN, Ravens-Sieberer U, et al. Self-reported quality of life of 8–12 year old children with cerebral palsy: a cross-sectional European study. Lancet 2007; 369: 217–18. Bjornson KF, Belza B, Kartin D, Logsdon RG, McLaughlin J. Self-reported health status and quality of life in youth with cerebral palsy and typically developing youth. Arch Phys Med Rehabil 2008; 89: 121–27. Novak I, Hines M, Goldsmith S, et al. Clinical prognostic messages from a systematic review of cerebral palsy. Pediatrics 2012; 130: e1285–312. Colver A, Fairhurst C, Pharoah POD. Cerebral palsy. Lancet 2014; 383: 1240–49. Peterson MD, Gordon PM, Hurvitz EA. Chronic disease risk among adults with cerebral palsy: the role of premature sarcopoenia, obesity and sedentary behavior. Obes Rev 2013; 14: 171–82.
www.thelancet.com Published online October 7, 2014 http://dx.doi.org/10.1016/S0140-6736(14)61599-3
Quality of life in adolescents with cerebral palsy groups of adolescents were generally less than 0·5 SD; impairments were significantly associated with lower adolescent QoL in only three of ten domains (Moods and emotions, Autonomy, and Social support and peers). Adolescents with cerebral palsy had significantly lower QoL than matched controls in the general population in only one domain—Social support and peers (mean difference –2·7 [0·25 SD]; 95% CI –4·3 to –1·4). Similar findings of stability of QoL among school-age children, both with cerebral palsy and typically developing peers, have been reported in several other large studies.9,10 Colver and colleagues’ report adds to the growing recognition that QoL reflects the unique internal milieu and perceived possibilities of each child, with or without cerebral palsy. An important point of Colver and colleagues’ study6 is that there are modifiable factors that can positively change QoL in children with cerebral palsy. These factors include parenting stress, psychological problems, and most notably pain. Pain in childhood or adolescence was strongly associated with low adolescent QoL (up to 6 points lower) in eight domains. Pain in children with cerebral palsy is a
www.thelancet.com Published online October 7, 2014 http://dx.doi.org/10.1016/S0140-6736(14)61599-3
Published Online October 7, 2014 http://dx.doi.org/10.1016/ S0140-6736(14)61599-3 See Online/Articles http://dx.doi.org/10.1016/ S0140-6736(14)61229-0
A Hoon MD
Child development is dynamic and multidimensional, with genetic and epigenetic building blocks upon which the complex effects of environment are overlaid (ie, nurture through nature).1 In children with cerebral palsy and other disabilities, developmental outcome is more than the result of medical treatment, and includes the child’s communication, coping, and problem-solving skills; educational opportunities; family adaptation; and service delivery2—factors all shown by quality of life (QoL). WHO’s definition of QoL as “the individual’s perception of their position in life in the context of the culture and value system in which they live, and in relation to their goals, expectations, standards, and concerns”3 represents the validation of the unique value of every person, typically developing or with a disability. The University of Toronto Centre for Health Promotion defines QoL as “the degree to which a person enjoys the important possibilities of his or her life”.4 This definition emphasises that the quality of a child’s life is interconnected with the nature of caregiver care and the environment in which each child lives. Increasingly, QoL is valued as an important addition to standard biomedical reports, and as a measure of clinical progress and research outcome.5 The large longitudinal cohort study of Allan Colver and colleagues,6 reported in The Lancet, examines how selfreported QoL in adolescents with cerebral palsy varies with impairment, how it compares with the perceived QoL of peers in the general population, and to what extent childhood factors amenable to intervention— pain, psychological problems, and parenting stress— predict adolescent QoL. Young people reported their QoL using KIDSCREEN, a generic QoL instrument with sound psychometric properties designed for children and adolescents.7 This study was done as part of the larger European SPARCLE study whose strengths include randomly selected sampling from population-based cerebral palsy registers in nine European regions.8 Colver and colleagues report outcomes from a 5-year longitudinal follow-up (SPARCLE2) of 355 adolescents aged 13–17 years who were able to self-report their QoL (longitudinal sample) and a cross-sectional sample including 76 additional adolescents (n=431).6 The findings are encouraging in that average differences in QoL between the least-able and most-able
1
Comment
common problem. In a 2012 article,11 75% of children with cerebral palsy were reported to have pain, which was present with all levels of disability, increased with age, and was linked to higher rates of behavioural problems and lower participation. Given the effects on adolescent QoL, pain assessment and management should be a priority for all children with cerebral palsy, irrespective of level of motor impairment. One caveat, which Colver and colleagues recognise, is that the study excluded children and adolescents with cerebral palsy who could not self-report their QoL because of severe learning difficulties. This group often has more severe and multiple impairments, and a parent or caregiver proxy report might yield different QoL results. Thus, no conclusions can be drawn from this study about the QoL of adolescents with severe developmental disabilities, and the challenge is to include their voices in future research. As known by people with cerebral palsy, the medical and rehabilitative community now recognises that affected individuals seek the same QoL and social participation as others, rather than just improved physical function for its own sake.12 Cerebral palsy is a lifelong disorder with interventions during childhood that might have long-term individual, family, and societal benefits. Colver and colleagues’ study6 suggests that children and adolescents with cerebral palsy may need particular help maintaining and developing peer relationships. Childhood interventions that optimise social support, health, and wellbeing may affect not only adolescent QoL, but also adult outcome by optimising each individual’s potential to adapt to, and prevent or reduce, the secondary morbidities often present.13
2
Colver and colleagues are to be commended for their work in placing QoL at the centre of the thoughts of all interested in promoting the wellbeing of people with cerebral palsy. *Alexander H Hoon Jr, Elaine E Stashinko Kennedy Krieger Institute, Baltimore, MD 21205, USA [email protected] We declare no competing interests. Copyright © Hoon et al. Open Access article distributed under the terms of CC BY. 1 2
3
4
5
6
7
8
9
10
11 12 13
Bjornsson HT, Fallin MD, Feinberg AP. An integrated epigenetic and genetic approach to common human disease. Trends Genet 2004; 20: 350–58. Ronen GM, Fayed N, Rosenbaum PL. Outcomes in pediatric neurology: a review of conceptual issues and recommendations. The 2010 Ronnie Mac Keith lecture. Dev Med Child Neurol 2011; 53: 305–12. The WHOQOL Group. The World Health Organization quality of life assessment (WHOQOL): position paper from the World Health Organization. Soc Sci Med 1995; 41: 1403–09. University of Toronto Centre for Health Promotion Quality of Life Research Unit. Quality of life model. http://sites.utoronto.ca/qol/ (accessed Aug 22, 2014). Stashinko EE, Harley LA, Steele RA, Clegg NJ. Parental perspectives on living with a child with HoPE. Am J Med Genet C Semin Med Genet 2010; 154C: 197–201. Colver A, Rapp M, Eisemann N, et al. Self-reported quality of life of adolescents with cerebral palsy: a cross-sectional and longitudinal analysis. Lancet 2014; published online Oct 7. http://dx.doi.org/10.1016/S01406736(14)61229-0. Ravens-Sieberer U, Gosch A, Rajmil L, et al. KIDSCREEN-52 quality-of-life measure for children and adolescents. Expert Rev Pharmacoecon Outcomes Res 2005; 5: 353–64. Colver A; the SPARCLE group. Study protocol: SPARCLE—a multi-centre European study of the relationship of environment to participation and quality of life of children with cerebral palsy. BMC Public Health 2006; 6: 105. Dickinson HO, Parkinson KN, Ravens-Sieberer U, et al. Self-reported quality of life of 8–12 year old children with cerebral palsy: a cross-sectional European study. Lancet 2007; 369: 217–18. Bjornson KF, Belza B, Kartin D, Logsdon RG, McLaughlin J. Self-reported health status and quality of life in youth with cerebral palsy and typically developing youth. Arch Phys Med Rehabil 2008; 89: 121–27. Novak I, Hines M, Goldsmith S, et al. Clinical prognostic messages from a systematic review of cerebral palsy. Pediatrics 2012; 130: e1285–312. Colver A, Fairhurst C, Pharoah POD. Cerebral palsy. Lancet 2014; 383: 1240–49. Peterson MD, Gordon PM, Hurvitz EA. Chronic disease risk among adults with cerebral palsy: the role of premature sarcopoenia, obesity and sedentary behavior. Obes Rev 2013; 14: 171–82.
www.thelancet.com Published online October 7, 2014 http://dx.doi.org/10.1016/S0140-6736(14)61599-3
