Fertnig / iStockphoto
Mary Ann Cantrell, PhD, RN and Michelle M. Kelly, PhD, CRNP
HEALTH-RELATED QUALITY OF LIFE FOR
Chronically Ill Children
Abstract Approximately 43% of children in the United States (32 million) are currently living with at least 1 of 20 common chronic childhood illnesses. The most common chronic childhood illnesses are asthma, cystic fibrosis, diabetes, obesity, malnutrition, developmental disabilities, cerebral palsy, consequences of low birthweight, and mental illness. For all chronically ill pediatric populations, the outcome of health-related quality of life (HRQOL) is particularly important because many of these children have not and will not be cured, and will continue to manage their chronic illness into adulthood. Advances in biomedical science and technology continue to improve efficacy of treatments and care for chronically ill children, adolescents, and their families, which highlight the importance measurement of HRQOL as a treatment and health status outcome. The construct of HRQOL is subjective, multidimensional, dynamic, and unique to each individual. It includes aspects of physical, psychological, social function, and goal attainment. Outcomes of HRQOL now include the financial implications for these children and their families, as well as financial and organizational consequences for healthcare planning and delivery of services. This article reviews the importance of HRQOL as a health outcome for chronically ill children. A historical overview and synthesis of the conceptualization and measurement of HRQOL for the chronically ill pediatric population is provided. Current research investigations that have measured health outcomes using individual scales tailored to children’s specific symptoms health outcomes, such as PROMIS®—Patient Reported Outcomes Measurement Information System—are reviewed. The clinical applications of HRQOL outcomes research include facilitation of patient-healthcare provider communication, improved patient satisfaction, identification of hidden morbidities, a positive impact on clinical decision making, and improvement of patient outcomes over time. Key words: Chronic health conditions in children; Health-related quality of life in children; Pediatric nursing.
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H
ealthy People 2020 identified quality-of-life improvement as a central public health goal (United States Department of Health and Human Services, 2010). It is a significant aspect of the health status of individuals, families, communities, and the general public. Healthrelated quality of life (HRQOL) is a focus in clinical trials, clinical practice, improvement strategies, and healthcare services research and evaluation (Varni, Burwinkle, & Lane, 2005; Varni, Limbers, Burwinkle, 2007). Improved treatment and survival rates among chronically ill children and adolescents with serious chronic diseases in developed countries have increased during the last decades (Kulpeng et al., 2013). The benefits of measurement of HRQOL in clinical practice include: (1) facilitation of patient-healthcare provider communication, (2) improved patient satisfaction, (3) identification of hidden morbidities, (4) positive impact on clinical decision making, and (5) improvement of patient outcomes over time (Varni et al., 2005). The construct of HRQOL is discussed by those concerned with public health, epidemiology, psychology,
nursing, and medicine. Assessments of HRQOL provide a broad picture of a child’s health, including the subjective facets of health, behaviors, and well-being (Simon, Chan, & Forrest, 2008). Focusing on HRQOL as a national health standard can bridge boundaries between disciplines and between social, mental, and medical services (Centers for Disease Control and Prevention [CDC], 2013). Across disciplines, researchers engaged in defining and identifying the theoretical aspects of HRQOL have developed and tested measures for this construct among pediatric chronically ill populations. Factors that affect the HRQOL of children with health conditions include physical, psychological, and social domains regardless of particular medical diagnosis. These domains, the children’s level of mastery, and degree of adaptation combine to shape their assessment of HRQOL. This paper reviews the importance of HRQOL as a health outcome for chronically ill children and synthesizes the current conceptualization and measurement of HRQOL in the pediatric health literature.
HRQOL Outcomes for Chronically Ill Children and Adolescents National- and state-level estimates derived from the 2007 National Survey of Children’s Health revealed that 43% of U.S. children (32 million) currently have at least 1 of 20 chronic health conditions, and this estimate increases to 54.1% when overweight, obesity, or being at risk for developmental delay are included (Bethell et al., 2011). The most common chronic childhood illnesses include asthma, cystic fibrosis, diabetes, obesity, malnutrition, developmental disabilities, cerebral palsy, consequences of low birthweight, and mental illness. The incidence of some chronic pediatric health conditions, such as asthma and obesity, is rising (van der Lee, Mokkink, Grootenhuis, Heymans, & Offringa, 2007). The prevalence of chronic diseases in children and in young adults, which is a function of incidence and duration, has increased since the 1980s and will likely continue to increase in the future (Enriquez, Hartert, & Persky, 2007; Newacheck, Rising, & Kim, 2006; Reilly & Dorosty, 1999). Likewise, developmental disabilities are common and were reported in one of six children in the United States in 2006– 2008. The number of children with select developmental disabilities (autism, attention deficit hyperactivity disorder, and other developmental delays) has increased (Boyle et al., 2011). For all chronically ill pediatric populations, HRQOL as a healthcare outcome is particularly important because many of these children have not and will not be cured, and sequelae of their disease or its treatment will require management into adulthood (van der Lee et al., 2007). According to the CDC (2013), measurement of HRQOL can achieve the following to improve the overall health of the nation: (1) determination of the burden of preventable disease, injuries, and disabilities; provide valuable new insights into the relationships between January/February 2015
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Chronic health conditions affect 32 million children in the United States.
HRQOL and risk factors; and help monitor progress in achieving the nation’s health objectives, (2) analysis of HRQOL surveillance data can identify subgroups with relatively poor perceived health and help to guide interventions to improve their situations and avert more serious consequences, and (3) interpretation and publication of these data can help identify needs for health policies and legislation, help to allocate resources based on unmet needs, guide the development of strategic plans, and monitor the effectiveness of broad community interventions. For the past decade, research efforts among pediatric healthcare professionals have reflected these stated aims by the CDC (2013). The 2004 report from the Institutes of Medicine/National Research Council, Children’s Health, The Nation’s Wealth: Assessing and Improving Child Health, highlighted that child health is critical to assuring national well-being at present and in the future (McCormick et al., 2011). Estimating the impact of illnesses and morbidities experienced by children and adolescents is now considered to be essential to decision making, policy planning, and intervention research (Waters, Davis, Nicolas, Wake, & Lo, 2008). Waters et al. (2008) conducted a cross-sectional school-based epidemiological study of 5,414 children and adolescents ages 5 to 18 years, and examined parental reports of child health and well-being to: (1) estimate population prevalence of physical and mental health conditions for children, (2) quantify their impact on multiple dimensions of children’s health and well-being, and (3) examine the cumulative effect of concurrent conditions. Findings of this study suggested that children’s health and well-being decrease linearly with increasing presence and frequency of health problems. Three or more concurrent conditions significantly burden children’s health and well-being (Waters et al., 2008). 26
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Determining effect of any condition or disease is complex, but childhood conditions are particularly complicated. Means of quantifying effect of chronic illness apart from diagnosis classification include assessment of functional limitations, health service utilization, or condition severity. McPherson et al. (1998) advocated a noncategorical designation that focused on use or need for special healthcare services, defining children with special healthcare needs as “those who have or are at increased risk for a chronic physical, development, behavioral or emotional condition and who also require health and related services of a type or amount beyond that required by children generally” (p. 138). These are children whose health conditions necessitate special health services to allow them to improve their health, access their environment, or perform the activities of childhood. Identification of risk factors influencing the HRQOL of children with special healthcare needs including general and specific physical, developmental, behavioral, and emotional conditions co-occurring has been the emphasis of recent investigations. Newacheck et al. (2006) conducted a secondary data analysis on the data from the 2003 National Survey of Children’s Health (NSCH) to identify children at risk for special healthcare needs. Newacheck et al. (2006) reported risk factors that decrease or increase the odds of experiencing special healthcare needs were the child’s age and gender, family structure and family conflict, and perception of neighborhood supportiveness. The financial burden of living with a chronic illness has been identified as a dimension of HRQOL for children. As many children and adolescents with chronic illness will continue to manage their conditions into adulthood (van der Lee et al., 2007), quantifying lasting effects of childhood health and economic circumstances on adult health, employment, and socioeconomic status in adults with a chronic childhood illness has become a concern among pediatric health researchers (Case, Fertig, & Paxson, 2005). In addition to the financial burden for families, the increase in prevalence of childhood chronic illness has considerable financial and organizational consequences for healthcare planning and for employment (van der Lee et al., 2007). According to van der Lee et al. (2007), such prevalence data on chronic conditions in children and young adults are not only useful for planning for healthcare and provisions for the welfare of young adults with chronic conditions, they can also be considered, in addition to mortality statistics, as outcome measures in comparisons of determinants of youth health between countries and over time. To enhance societal participation of these children as they approach adulthood and to be able to plan for sufficient and adequate facilities, policy makers and politicians need to rely on valid prevalence data (van der Lee et al., 2007). Measuring HRQOL outcomes can have a meaningful impact in the lives of chronically ill children and their families. As evidence is generated about specific risk factors, targeted interventions can be tested and integrated into the care for these children and families to improve their overall well-being and functioning. Research findings on the January/February 2015
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HRQOL outcomes can help develop evidence-based clinical practice guidelines to address the short- and long-term health and psychosocial issues, behavioral health, and lifestyle management issues. Children born prematurely provide a unique example of how HRQOL findings directly relate to improving the health of children and their families. As they age, the health conditions of children born prematurely persist, but may no longer be attributed to their prematurity (Kelly, 2014). Learning differences and behavioral difficulties affect the social, emotional, and school functioning domains of HRQOL for these children and their families (Kelly, 2014). Assessment of HRQOL, as a patient-reported outcome for children born prematurely, can facilitate child-to-parent and parent-to-healthcare provider discussions to identify services to improve HRQOL.
Historical Overview of HRQOL Research In a critical appraisal of the QOL measurements in the general healthcare literature, Gill and Feinstein (1994) commented, “Since the 1970s, the measurement of quality of life has grown from a small cottage industry to a large academic enterprise” (p. 624). An explosion of theoretical literature, conceptualizing HRQOL is evident in the adult and pediatric literature. The conceptualizations of HRQOL among chronically ill adult and pediatric populations have their theoretical underpinnings rooted in the definition of health from the World Health Organization (WHO). The WHO’s sentinel definition of health identified it as a state of complete physical, mental, and social well-being and not merely the absence of disease or infirmity (WHO, 1948). The WHO’s definition about what constitutes health is still supported by researchers in the present day. Davis et al. (2006) reviewed HRQOL and quality-of-life instruments used in pediatric research of children less than 12 years of age and concluded that HRQOL was defined as functioning, feelings about functioning, health, and value assigned to duration of life. Quality of life was defined as position in life, functioning, feelings about functioning, existence, and discrepancy between actual and ideal self (Davis et al., 2006). Based on their review, Davis et al. (2006) concluded that the absence of ill-being does not equate to a high level of well-being. As noted by Sredl (2004), healthcare providers now acknowledge that all aspects of a person’s life affect the delicate balance of well-being known as quality of life. Costain, Hewison, and Howes (1993) critiqued early theoretical models explaining quality of life among oncology patients as primarily function-based or meaning-based. Similarly, Gill and Feinstein (1994) believed that the early conceptualizations of HRQOL did not incorporate patients’ values and preferences. These are limitations of function-based models to measure HRQOL because they operate from a biomedical model, which emphasizes physical functioning and impact January/February 2015
Health-related quality of life (HRQOL), as a national health standard, can bridge boundaries between disciplines and service providers.
of long-term treatment effects on health status. In contrast, meaning-based models emphasize patterns and treatment experience of cancer from a subjective and holistic perspective (Haase, Heiney, Ruccione, & Stutzer, 1999). In a concept analysis of HRQOL in the overall nursing literature, Sredl (2004) identified six major components of HRQOL that contain subconstructs within them: physical, psychological, personality, environmental, social, and future orientation. These aspects of the phenomenon are understood to be multifactorial and interactive with one another. In children, HRQOL is now understood to be multidimensional and is a component of personal health (Simon et al., 2008). The qualitative work examining HRQOL presents a fairly concise picture of the domains that affect HRQOL in children (Davis et al., 2006; Taylor, Gibson, & Franck, 2008); these consist of physical, psychological, and social domains regardless of a particular diagnosis or functional status. Within the pediatric healthcare literature, there are multiple definitions of HRQOL. In a concept analysis of HRQOL by Taylor et al. (2008) that specifically focused on young people with chronic illness, the authors explored quality-of-life literature and developed the concept and definition of quality of life for young children with chronic illness. In their definition, however, the authors added the qualifier “healthrelated” to specify quality of life “from a health perspective and not involving other wider factors such as environment” (Taylor et al., 2008). Taylor et al. (2008) define HRQOL in young people with chronic illness as: MCN
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subjective, multidimensional, and dynamic. It is unique to each individual young person and includes aspects of physical, psychological and social function. It is dependent upon not only the stage of development but also the illness trajectory. This involves the achievement of goals, and aspirations and the constraints imposed through ill-health and treatment (p. 1831). A related issue in the measurement of HRQOL among chronically ill children and adolescents is usefulness and validity of proxy measures. Historically, researchers relied on proxy respondents, parents, caregivers, or medical records, presuming that children were not able to respond to HRQOL questions. In a review of 14 HRQOL measurements that allowed children and parent proxy measures of HRQOL among children and adolescents, Eiser and Morse (2001) concluded that parents are better able to assess physical domains of health and less able to assess social or emotional domains. In contrast, Seid, Varni, and Jacobs (2000) and Varni, Seid, and Kurtin (2001) reported that proxy assessments are more accurate for objective, functional behaviors, and less congruent with patient report for subjective, perception-based, or internalized components of HRQOL. Discrepancy between parent and child reports may stem from parents’ limited perception or awareness of the child’s life (Jokovic, Locker, & Guyatt, 2004). As a child ages and spends time outside of the direct observation of the parents, it is more likely that parents will have less direct knowledge of the child’s abilities, social functioning, and peer interactions (Jokovic et al., 2004). Eiser and Morse (2001) argued that parents of sick children may be more perceptive of their children’s attitudes and abilities. Parents’ perceptions of a child’s HRQOL may not mirror the child in all aspects, but are valuable to assess as parents determine the utilization of healthcare services (Jokovic et al., 2004; Seid et al., 2000; Varni et al., 2001). Parallel reporting designs that use equivalent tools are recommended for assessing health outcomes of children (Jokovic et al., 2004).
Measurement of HRQOL and Health Outcomes in Chronically Ill Children The pediatric healthcare literature has many instruments to measure HRQOL in healthy and chronically ill pediatric populations. In an early review of the state of the art on HRQOL for children, Eiser and Morse (2001) identified 43 measures, 19 generic and 24 disease-specific, in the published literature from 1980 to 1999. The most frequently used measure has been the PEDSQL™ 4.0 (Pediatric Quality of Life Inventory) (Varni et al., 2001; Varni, Seid, & Rode, 1999). In an investigation to evaluate the quality of established parent-child reporting measures, Upton, Lawford, and Eiser (2008) cited 19 studies in which 16 used either the generic or disease-specific versions of the PedsQL the PEDSQL™ 4.0. The PedsQL 4.0 28
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Generic Core Scales Child version includes 23 items encompassing physical functioning (8 items), emotional functioning (5 items), social functioning (5 items), and school functioning (5 items) (Varni et al., 2001). Separate scores for physical, emotional, social, and school functioning can be obtained as well as the combined psychosocial health score combining the emotional, social, and school functioning subscales. The disease-specific versions of the PEDSQL™ 4.0 include modules specific for pediatric end-stage renal disease, sickle cell disease, cancer, cerebral palsy, diabetes, gastrointestinal conditions, obesity, asthma, cardiac, and rheumatology patients (Varni et al., 2007). These disease-specific scales permit comparisons within and across chronically ill pediatric populations. In contrast to existing measures of HRQOL, individual scales that are tailored to children’s specific symptoms are now being recognized as being more relevant in measurement of HRQOL outcomes among chronically ill children. This approach is relatively new for nursing professionals involved in research and care of chronically ill patients. The body of knowledge reflecting this approach in the nursing literature is evolving. The work of multidisciplinary teams, which have included nurse researchers, who have investigated health outcomes for pediatric populations that have used specific symptom measures such as PROMIS®, is presented as an exemplar of this emerging research approach. The National Institutes of Health (NIH) constructed a patient information system known as PROMIS—Patient Reported Outcomes Measurement Information System. The PROMIS system is comprised of highly reliable, precise measures of patient-reported health status for physical, mental, and social well-being (NIH, n.d.). Health domain outcomes for chronically ill children assessed in PROMIS include sleep/wake function, sexual function, cognitive function, and the psychosocial impacts of the illness experience such as the stress response, coping and shifts in selfconcept, social interactions, and spirituality (NIH, n.d.). A complete list of PROMIS instruments to measure specific domains of HRQOL in chronically ill children is provided in Table 1. This NIH-sponsored initiative has its goal to develop systems to support NIH-funded research supported by all of its institutes and centers (NIH, n.d.). The overall objective is standardizing patient-reported outcomes assessment in research investigations, especially clinical trials, using a patient-reported outcomes measurement information system. These outcome assessments can be used as primary or secondary endpoints in clinical studies of the effectiveness of treatment, and can be used across a wide variety of chronic diseases and conditions and in the general population (NIH, n.d.). The data collected in PROMIS provide clinicians and researchers with important patientreported information about the effect of therapy that cannot be found in traditional clinical measures (NIH, n.d.). When used with traditional clinical measures of health, PROMIS tools allow clinicians to better understand how various treatments might affect what patients are able to do and the symptoms they experience, and can be used by January/February 2015
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TABLE 1. PPROMIS Instruments to Measure Specific Domains of HRQOL in Chronically Ill Children Anger Anxiety Asthma impact on daily life Depressive symptoms Fatigue Cognition (general concerns in applied cognitive functioning) Mobility (lower extremity functioning) Pain (interference of pain in daily functioning) Peer relationships Physical activity Self-concept Sexual functioning Sleep/wake functioning Social relationships (interaction with peers) Spirituality Stress experience Stigma Subjective well-being Upper extremity functioning (fine motor functioning and ability to perform activities of daily living)
patients and physicians to improve communication and manage chronic disease (NIH, n.d.). Among the nearly 290 publications that have used PROMIS, 26 of these investigations have addressed health outcomes for pediatric populations. Between the 2011 and 2012 only six publications were located in the literature for children using PROMIS. In contrast, 20 publications were located for 2013 and 2014 (up to July 2014). Bevans, Riley, Moon, and Forrest (2010) discussed in the use of PROMIS the advancements of conceptual and methodological advances in child-reported health outcomes. Bevans et al. (2010) concluded that rapid advances in building a solid scientific foundation for health-reported outcomes in children are a direct result of methodological standards for the assessment of patient-reported outcomes such as PROMIS. Clinical investigations using PROMIS have addressed a wide variety of health outcomes such as pain in the pediatric patient (Varni et al., 2010), health outcomes for children with asthma (Yeatts et al., 2010), cancerrelated fatigue in children (Barsevick et al., 2013), symptom patterns and functional impairment in children with cancer (Buckner et al., 2014), and biological and behavioral correlates of fatigue in adolescents and young adults with sickle cell anemia (Ameringer, Elswick, & Smith, 2014). In addition to these clinical investigations, methodological research examining psychometric properties of the studies by multidisciplinary teams of pediatric oncology researchers PROMIS scales has been done. January/February 2015
For children with chronic illness, HRQOL is particularly important, as they will live with their condition, or its sequelae into adulthood.
Menard et al. (2014) assessed the feasibility and acceptability of the PROMIS pediatric measures, among a cross-sectional study of 200 children in active cancer treatment and in a longitudinal study of 94 children with cancer ranging in ages of 8 to 18 years. Menard et al. (2014) reported that PROMIS measures demonstrated acceptability and feasibility as defined by enrollment and attrition rates as well as missingness by measure, item, participants, and assessment point in time and at different points in cancer treatment and was feasible to use in inpatient and outpatient pediatric oncology settings. Likewise, Hinds et al. (2013) reported PROMIS pediatric measures in pediatric oncology to be valid and clinically feasible indicators of patient-reported outcomes.
Conclusion Pediatric healthcare researchers have made HRQOL and health outcomes a central focus in the care and research base for chronically ill children and adolescents and their families. Data generated from the investigations of HRQOL among pediatric healthcare researchers make an important contribution to improve the overall health of the nation. With the existing incidence and prevalence of chronic illness in children and the continued rise in the prevalence of specific chronic health conditions that will persist in adulthood, the measurement of HRQOL as a central treatment and outcome variable is unquestionable. The research base in the conceptualization and measurement of HRQOL among chronically ill populations reflects systematic and rigorous science, which continues to evolve. The value of assessing HRQOL as an outcome in children is multidimensional. It is advantageous in MCN
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Clinical Nursing Implications: 1. Nurses of all specialties will benefit from a current understanding of HRQOL outcomes in children with chronic illness. 2. There is strong evidence that child self-report and parent proxy-report of the child’s HRQOL may be different, but both perspectives provide valuable insight into the health experience of the child. 3. Assessment, measurement, and discussion of HRQOL, as a patient-reported outcome, are advocated as important features in improving the health of children with chronic illness. 4. Objective, standardized HRQOL measurement tools, such as PROMIS, are vital to the evaluation of clinical interventions and therapies.
determining the efficacy of an intervention, in identifying progress in treatment, spurring discussions between family and healthcare provider, and capturing population data for policy decision making. Current research using individual scales that are tailored to chronically ill children’s specific symptoms to measure health outcomes, specifically PROMIS, is generating data with significant relevance in improving the care and lives of these children and is capturing population data for policy decision making. ✜ Mary Ann Cantrell is a Professor, College of Nursing, Villanova University, Driscoll Hall 338, 800 East Lancaster Avenue, Villanova, PA. She can be reached via e-mail at [email protected] Michelle M. Kelly is an Assistant Professor, College of Nursing, Villanova University, Driscoll Hall 398, 800 Lancaster Avenue, Villanova, PA. The authors declare no conflict of interest. DOI:10.1097/NMC.0000000000000090 References Ameringer, S., Elswick, R. K., Jr., & Smith, W. (2014). Fatigue in adolescents and young adults with sickle cell disease: Biological and behavioral correlates and health-related quality of life. Journal of Pediatric Oncology Nursing, 31(1), 6-17. doi:10.1177/1043454213514632 Barsevick, A. M., Irwin, M. R., Hinds, P., Miller, A., Berger, A., Jacobsen, P., …, Cella, D. (2013). Recommendations for high-priority research on cancer-related fatigue in children and adults. Journal of the National Cancer Institute, 105(19), 1432-1440. doi:10.1093/jnci/djt242 Bethell, C. D., Kogan, M. D., Strickland, B. B., Schor, E. L., Robertson, J., & Newacheck, P. W. (2011). A national and state profile of leading health problems and health care quality for US children: Key insurance disparities and across-state variations. Academic Pediatrics, 11(Suppl. 3), S22-S33. doi:10.1016/j.acap.2010.08.011 Bevans, K. B., Riley, A. W., Moon, J., & Forrest, C. B. (2010). Conceptual and methodological advances in child-reported outcomes measurement. Expert Review of Pharmacoeconomics & Outcomes Research, 10(4), 385-396. doi:10.1586/erp.10.52 Boyle, C. A., Boulet, S., Schieve, L. A., Cohen, R. A., Blumberg, S. J., Yeargin-Allsopp, M, …, Kogan, M. D. (2011). Trends in the prevalence of developmental disabilities in US children, 1997-2008. Pediatrics, 127(6), 1034-1042. doi:10.1542/peds.2010-2989 Buckner, T. W., Wang, J., DeWalt, D. A., Jacobs, S., Reeve, B. B., & Hinds, P. S. (2014). Patterns of symptoms and functional impairments in
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Retrieved July 19, 2014, from http://healthypeople.gov/2020/ topicsobjectives2020/overview.aspx?topicid=2 Upton, P., Lawford, J. & Eiser, C. (2008). Parent-child agreement across child health- related quality of life instruments: A review of the literature. Quality of Life Research, 17, 895-913. doi:10.1007/s11136-0089350-5 van der Lee, J. H., Mokkink, L. B., Grootenhuis, M. A., Heymans, H. S., & Offringa, M. (2007). Definitions and measurement of chronic health conditions in childhood: A systematic review. JAMA: The Journal of the American Medical Association, 297(24), 2741-2751. doi:10.1001/ jama.297.24.2741 Varni, J.W., Burwinkle, R.M., & Lane, M.M. (2005). Health-related quality of life measurement in pediatric clinical practice: An applictaion and precept for future research and applictaion. Health and Quality of Life Outcomes, 3(34). doi: 10.1186/1477-7525-3-34 Varni, J. W., & Limbers, C. A. (2009). The Pediatric Quality of Life Inventory: Measuring pediatric health-related quality of life from the perspective of children and their parents. Pediatric Clinics of North America, 56(4), 843-863. doi:10.1016/j.pcl.2009.05.016 Varni, J. W., Limbers, C. A., & Burwinkle, T. M. (2007). Impaired healthrelated quality of life in children and adolescents with chronic
conditions: A comparative analysis of 10 disease clusters and 33 disease categories/severities utilizing the PedsQL 4.0 Generic Core Scales. Health and Quality of Life Outcomes, 5, 43. doi:10.1186/14777525-5-43 Varni, J. W., Seid, M., & Kurtin, P. S. (2001). PedsQL 4.0: Reliability and validity of the Pediatric Quality of Life Inventory version 4.0 generic core scales in healthy and patient populations. Medical Care, 39(8), 800-812. Varni, J. W., Seid, M., & Rode, C. A. (1999). The PedsQL: Measurement model for the Pediatric Quality of Life Inventory. Medical Care, 37(2), 126-139. Waters, E., Davis, E., Nicolas, C., Wake, M., & Lo, S. K. (2008). The impact of childhood conditions and concurrent morbidities on child health and well-being. Child: Care, Health and Development, 34(4), 418-429. doi:10.1111/j.1365-2214.2008.00825.x 2214.2008.00825.x World Health Organization. (1948). Constitution of the World Health Organization basic document. Geneva, Switzerland: Author. Yeatts, K. B., Stucky, B., Thissen, D., Irwin, D., Varni, J. W., DeWitt, E. M., …, DeWalt, D. A. (2010). Construction of the Pediatric Asthma Impact Scale (PAIS) for the Patient-Reported Outcomes Measurement Information system (PROMIS). The Journal of Asthma, 47(3), 295-302.
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Mary Ann Cantrell, PhD, RN and Michelle M. Kelly, PhD, CRNP
HEALTH-RELATED QUALITY OF LIFE FOR
Chronically Ill Children
Abstract Approximately 43% of children in the United States (32 million) are currently living with at least 1 of 20 common chronic childhood illnesses. The most common chronic childhood illnesses are asthma, cystic fibrosis, diabetes, obesity, malnutrition, developmental disabilities, cerebral palsy, consequences of low birthweight, and mental illness. For all chronically ill pediatric populations, the outcome of health-related quality of life (HRQOL) is particularly important because many of these children have not and will not be cured, and will continue to manage their chronic illness into adulthood. Advances in biomedical science and technology continue to improve efficacy of treatments and care for chronically ill children, adolescents, and their families, which highlight the importance measurement of HRQOL as a treatment and health status outcome. The construct of HRQOL is subjective, multidimensional, dynamic, and unique to each individual. It includes aspects of physical, psychological, social function, and goal attainment. Outcomes of HRQOL now include the financial implications for these children and their families, as well as financial and organizational consequences for healthcare planning and delivery of services. This article reviews the importance of HRQOL as a health outcome for chronically ill children. A historical overview and synthesis of the conceptualization and measurement of HRQOL for the chronically ill pediatric population is provided. Current research investigations that have measured health outcomes using individual scales tailored to children’s specific symptoms health outcomes, such as PROMIS®—Patient Reported Outcomes Measurement Information System—are reviewed. The clinical applications of HRQOL outcomes research include facilitation of patient-healthcare provider communication, improved patient satisfaction, identification of hidden morbidities, a positive impact on clinical decision making, and improvement of patient outcomes over time. Key words: Chronic health conditions in children; Health-related quality of life in children; Pediatric nursing.
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H
ealthy People 2020 identified quality-of-life improvement as a central public health goal (United States Department of Health and Human Services, 2010). It is a significant aspect of the health status of individuals, families, communities, and the general public. Healthrelated quality of life (HRQOL) is a focus in clinical trials, clinical practice, improvement strategies, and healthcare services research and evaluation (Varni, Burwinkle, & Lane, 2005; Varni, Limbers, Burwinkle, 2007). Improved treatment and survival rates among chronically ill children and adolescents with serious chronic diseases in developed countries have increased during the last decades (Kulpeng et al., 2013). The benefits of measurement of HRQOL in clinical practice include: (1) facilitation of patient-healthcare provider communication, (2) improved patient satisfaction, (3) identification of hidden morbidities, (4) positive impact on clinical decision making, and (5) improvement of patient outcomes over time (Varni et al., 2005). The construct of HRQOL is discussed by those concerned with public health, epidemiology, psychology,
nursing, and medicine. Assessments of HRQOL provide a broad picture of a child’s health, including the subjective facets of health, behaviors, and well-being (Simon, Chan, & Forrest, 2008). Focusing on HRQOL as a national health standard can bridge boundaries between disciplines and between social, mental, and medical services (Centers for Disease Control and Prevention [CDC], 2013). Across disciplines, researchers engaged in defining and identifying the theoretical aspects of HRQOL have developed and tested measures for this construct among pediatric chronically ill populations. Factors that affect the HRQOL of children with health conditions include physical, psychological, and social domains regardless of particular medical diagnosis. These domains, the children’s level of mastery, and degree of adaptation combine to shape their assessment of HRQOL. This paper reviews the importance of HRQOL as a health outcome for chronically ill children and synthesizes the current conceptualization and measurement of HRQOL in the pediatric health literature.
HRQOL Outcomes for Chronically Ill Children and Adolescents National- and state-level estimates derived from the 2007 National Survey of Children’s Health revealed that 43% of U.S. children (32 million) currently have at least 1 of 20 chronic health conditions, and this estimate increases to 54.1% when overweight, obesity, or being at risk for developmental delay are included (Bethell et al., 2011). The most common chronic childhood illnesses include asthma, cystic fibrosis, diabetes, obesity, malnutrition, developmental disabilities, cerebral palsy, consequences of low birthweight, and mental illness. The incidence of some chronic pediatric health conditions, such as asthma and obesity, is rising (van der Lee, Mokkink, Grootenhuis, Heymans, & Offringa, 2007). The prevalence of chronic diseases in children and in young adults, which is a function of incidence and duration, has increased since the 1980s and will likely continue to increase in the future (Enriquez, Hartert, & Persky, 2007; Newacheck, Rising, & Kim, 2006; Reilly & Dorosty, 1999). Likewise, developmental disabilities are common and were reported in one of six children in the United States in 2006– 2008. The number of children with select developmental disabilities (autism, attention deficit hyperactivity disorder, and other developmental delays) has increased (Boyle et al., 2011). For all chronically ill pediatric populations, HRQOL as a healthcare outcome is particularly important because many of these children have not and will not be cured, and sequelae of their disease or its treatment will require management into adulthood (van der Lee et al., 2007). According to the CDC (2013), measurement of HRQOL can achieve the following to improve the overall health of the nation: (1) determination of the burden of preventable disease, injuries, and disabilities; provide valuable new insights into the relationships between January/February 2015
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Chronic health conditions affect 32 million children in the United States.
HRQOL and risk factors; and help monitor progress in achieving the nation’s health objectives, (2) analysis of HRQOL surveillance data can identify subgroups with relatively poor perceived health and help to guide interventions to improve their situations and avert more serious consequences, and (3) interpretation and publication of these data can help identify needs for health policies and legislation, help to allocate resources based on unmet needs, guide the development of strategic plans, and monitor the effectiveness of broad community interventions. For the past decade, research efforts among pediatric healthcare professionals have reflected these stated aims by the CDC (2013). The 2004 report from the Institutes of Medicine/National Research Council, Children’s Health, The Nation’s Wealth: Assessing and Improving Child Health, highlighted that child health is critical to assuring national well-being at present and in the future (McCormick et al., 2011). Estimating the impact of illnesses and morbidities experienced by children and adolescents is now considered to be essential to decision making, policy planning, and intervention research (Waters, Davis, Nicolas, Wake, & Lo, 2008). Waters et al. (2008) conducted a cross-sectional school-based epidemiological study of 5,414 children and adolescents ages 5 to 18 years, and examined parental reports of child health and well-being to: (1) estimate population prevalence of physical and mental health conditions for children, (2) quantify their impact on multiple dimensions of children’s health and well-being, and (3) examine the cumulative effect of concurrent conditions. Findings of this study suggested that children’s health and well-being decrease linearly with increasing presence and frequency of health problems. Three or more concurrent conditions significantly burden children’s health and well-being (Waters et al., 2008). 26
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Determining effect of any condition or disease is complex, but childhood conditions are particularly complicated. Means of quantifying effect of chronic illness apart from diagnosis classification include assessment of functional limitations, health service utilization, or condition severity. McPherson et al. (1998) advocated a noncategorical designation that focused on use or need for special healthcare services, defining children with special healthcare needs as “those who have or are at increased risk for a chronic physical, development, behavioral or emotional condition and who also require health and related services of a type or amount beyond that required by children generally” (p. 138). These are children whose health conditions necessitate special health services to allow them to improve their health, access their environment, or perform the activities of childhood. Identification of risk factors influencing the HRQOL of children with special healthcare needs including general and specific physical, developmental, behavioral, and emotional conditions co-occurring has been the emphasis of recent investigations. Newacheck et al. (2006) conducted a secondary data analysis on the data from the 2003 National Survey of Children’s Health (NSCH) to identify children at risk for special healthcare needs. Newacheck et al. (2006) reported risk factors that decrease or increase the odds of experiencing special healthcare needs were the child’s age and gender, family structure and family conflict, and perception of neighborhood supportiveness. The financial burden of living with a chronic illness has been identified as a dimension of HRQOL for children. As many children and adolescents with chronic illness will continue to manage their conditions into adulthood (van der Lee et al., 2007), quantifying lasting effects of childhood health and economic circumstances on adult health, employment, and socioeconomic status in adults with a chronic childhood illness has become a concern among pediatric health researchers (Case, Fertig, & Paxson, 2005). In addition to the financial burden for families, the increase in prevalence of childhood chronic illness has considerable financial and organizational consequences for healthcare planning and for employment (van der Lee et al., 2007). According to van der Lee et al. (2007), such prevalence data on chronic conditions in children and young adults are not only useful for planning for healthcare and provisions for the welfare of young adults with chronic conditions, they can also be considered, in addition to mortality statistics, as outcome measures in comparisons of determinants of youth health between countries and over time. To enhance societal participation of these children as they approach adulthood and to be able to plan for sufficient and adequate facilities, policy makers and politicians need to rely on valid prevalence data (van der Lee et al., 2007). Measuring HRQOL outcomes can have a meaningful impact in the lives of chronically ill children and their families. As evidence is generated about specific risk factors, targeted interventions can be tested and integrated into the care for these children and families to improve their overall well-being and functioning. Research findings on the January/February 2015
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HRQOL outcomes can help develop evidence-based clinical practice guidelines to address the short- and long-term health and psychosocial issues, behavioral health, and lifestyle management issues. Children born prematurely provide a unique example of how HRQOL findings directly relate to improving the health of children and their families. As they age, the health conditions of children born prematurely persist, but may no longer be attributed to their prematurity (Kelly, 2014). Learning differences and behavioral difficulties affect the social, emotional, and school functioning domains of HRQOL for these children and their families (Kelly, 2014). Assessment of HRQOL, as a patient-reported outcome for children born prematurely, can facilitate child-to-parent and parent-to-healthcare provider discussions to identify services to improve HRQOL.
Historical Overview of HRQOL Research In a critical appraisal of the QOL measurements in the general healthcare literature, Gill and Feinstein (1994) commented, “Since the 1970s, the measurement of quality of life has grown from a small cottage industry to a large academic enterprise” (p. 624). An explosion of theoretical literature, conceptualizing HRQOL is evident in the adult and pediatric literature. The conceptualizations of HRQOL among chronically ill adult and pediatric populations have their theoretical underpinnings rooted in the definition of health from the World Health Organization (WHO). The WHO’s sentinel definition of health identified it as a state of complete physical, mental, and social well-being and not merely the absence of disease or infirmity (WHO, 1948). The WHO’s definition about what constitutes health is still supported by researchers in the present day. Davis et al. (2006) reviewed HRQOL and quality-of-life instruments used in pediatric research of children less than 12 years of age and concluded that HRQOL was defined as functioning, feelings about functioning, health, and value assigned to duration of life. Quality of life was defined as position in life, functioning, feelings about functioning, existence, and discrepancy between actual and ideal self (Davis et al., 2006). Based on their review, Davis et al. (2006) concluded that the absence of ill-being does not equate to a high level of well-being. As noted by Sredl (2004), healthcare providers now acknowledge that all aspects of a person’s life affect the delicate balance of well-being known as quality of life. Costain, Hewison, and Howes (1993) critiqued early theoretical models explaining quality of life among oncology patients as primarily function-based or meaning-based. Similarly, Gill and Feinstein (1994) believed that the early conceptualizations of HRQOL did not incorporate patients’ values and preferences. These are limitations of function-based models to measure HRQOL because they operate from a biomedical model, which emphasizes physical functioning and impact January/February 2015
Health-related quality of life (HRQOL), as a national health standard, can bridge boundaries between disciplines and service providers.
of long-term treatment effects on health status. In contrast, meaning-based models emphasize patterns and treatment experience of cancer from a subjective and holistic perspective (Haase, Heiney, Ruccione, & Stutzer, 1999). In a concept analysis of HRQOL in the overall nursing literature, Sredl (2004) identified six major components of HRQOL that contain subconstructs within them: physical, psychological, personality, environmental, social, and future orientation. These aspects of the phenomenon are understood to be multifactorial and interactive with one another. In children, HRQOL is now understood to be multidimensional and is a component of personal health (Simon et al., 2008). The qualitative work examining HRQOL presents a fairly concise picture of the domains that affect HRQOL in children (Davis et al., 2006; Taylor, Gibson, & Franck, 2008); these consist of physical, psychological, and social domains regardless of a particular diagnosis or functional status. Within the pediatric healthcare literature, there are multiple definitions of HRQOL. In a concept analysis of HRQOL by Taylor et al. (2008) that specifically focused on young people with chronic illness, the authors explored quality-of-life literature and developed the concept and definition of quality of life for young children with chronic illness. In their definition, however, the authors added the qualifier “healthrelated” to specify quality of life “from a health perspective and not involving other wider factors such as environment” (Taylor et al., 2008). Taylor et al. (2008) define HRQOL in young people with chronic illness as: MCN
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subjective, multidimensional, and dynamic. It is unique to each individual young person and includes aspects of physical, psychological and social function. It is dependent upon not only the stage of development but also the illness trajectory. This involves the achievement of goals, and aspirations and the constraints imposed through ill-health and treatment (p. 1831). A related issue in the measurement of HRQOL among chronically ill children and adolescents is usefulness and validity of proxy measures. Historically, researchers relied on proxy respondents, parents, caregivers, or medical records, presuming that children were not able to respond to HRQOL questions. In a review of 14 HRQOL measurements that allowed children and parent proxy measures of HRQOL among children and adolescents, Eiser and Morse (2001) concluded that parents are better able to assess physical domains of health and less able to assess social or emotional domains. In contrast, Seid, Varni, and Jacobs (2000) and Varni, Seid, and Kurtin (2001) reported that proxy assessments are more accurate for objective, functional behaviors, and less congruent with patient report for subjective, perception-based, or internalized components of HRQOL. Discrepancy between parent and child reports may stem from parents’ limited perception or awareness of the child’s life (Jokovic, Locker, & Guyatt, 2004). As a child ages and spends time outside of the direct observation of the parents, it is more likely that parents will have less direct knowledge of the child’s abilities, social functioning, and peer interactions (Jokovic et al., 2004). Eiser and Morse (2001) argued that parents of sick children may be more perceptive of their children’s attitudes and abilities. Parents’ perceptions of a child’s HRQOL may not mirror the child in all aspects, but are valuable to assess as parents determine the utilization of healthcare services (Jokovic et al., 2004; Seid et al., 2000; Varni et al., 2001). Parallel reporting designs that use equivalent tools are recommended for assessing health outcomes of children (Jokovic et al., 2004).
Measurement of HRQOL and Health Outcomes in Chronically Ill Children The pediatric healthcare literature has many instruments to measure HRQOL in healthy and chronically ill pediatric populations. In an early review of the state of the art on HRQOL for children, Eiser and Morse (2001) identified 43 measures, 19 generic and 24 disease-specific, in the published literature from 1980 to 1999. The most frequently used measure has been the PEDSQL™ 4.0 (Pediatric Quality of Life Inventory) (Varni et al., 2001; Varni, Seid, & Rode, 1999). In an investigation to evaluate the quality of established parent-child reporting measures, Upton, Lawford, and Eiser (2008) cited 19 studies in which 16 used either the generic or disease-specific versions of the PedsQL the PEDSQL™ 4.0. The PedsQL 4.0 28
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Generic Core Scales Child version includes 23 items encompassing physical functioning (8 items), emotional functioning (5 items), social functioning (5 items), and school functioning (5 items) (Varni et al., 2001). Separate scores for physical, emotional, social, and school functioning can be obtained as well as the combined psychosocial health score combining the emotional, social, and school functioning subscales. The disease-specific versions of the PEDSQL™ 4.0 include modules specific for pediatric end-stage renal disease, sickle cell disease, cancer, cerebral palsy, diabetes, gastrointestinal conditions, obesity, asthma, cardiac, and rheumatology patients (Varni et al., 2007). These disease-specific scales permit comparisons within and across chronically ill pediatric populations. In contrast to existing measures of HRQOL, individual scales that are tailored to children’s specific symptoms are now being recognized as being more relevant in measurement of HRQOL outcomes among chronically ill children. This approach is relatively new for nursing professionals involved in research and care of chronically ill patients. The body of knowledge reflecting this approach in the nursing literature is evolving. The work of multidisciplinary teams, which have included nurse researchers, who have investigated health outcomes for pediatric populations that have used specific symptom measures such as PROMIS®, is presented as an exemplar of this emerging research approach. The National Institutes of Health (NIH) constructed a patient information system known as PROMIS—Patient Reported Outcomes Measurement Information System. The PROMIS system is comprised of highly reliable, precise measures of patient-reported health status for physical, mental, and social well-being (NIH, n.d.). Health domain outcomes for chronically ill children assessed in PROMIS include sleep/wake function, sexual function, cognitive function, and the psychosocial impacts of the illness experience such as the stress response, coping and shifts in selfconcept, social interactions, and spirituality (NIH, n.d.). A complete list of PROMIS instruments to measure specific domains of HRQOL in chronically ill children is provided in Table 1. This NIH-sponsored initiative has its goal to develop systems to support NIH-funded research supported by all of its institutes and centers (NIH, n.d.). The overall objective is standardizing patient-reported outcomes assessment in research investigations, especially clinical trials, using a patient-reported outcomes measurement information system. These outcome assessments can be used as primary or secondary endpoints in clinical studies of the effectiveness of treatment, and can be used across a wide variety of chronic diseases and conditions and in the general population (NIH, n.d.). The data collected in PROMIS provide clinicians and researchers with important patientreported information about the effect of therapy that cannot be found in traditional clinical measures (NIH, n.d.). When used with traditional clinical measures of health, PROMIS tools allow clinicians to better understand how various treatments might affect what patients are able to do and the symptoms they experience, and can be used by January/February 2015
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TABLE 1. PPROMIS Instruments to Measure Specific Domains of HRQOL in Chronically Ill Children Anger Anxiety Asthma impact on daily life Depressive symptoms Fatigue Cognition (general concerns in applied cognitive functioning) Mobility (lower extremity functioning) Pain (interference of pain in daily functioning) Peer relationships Physical activity Self-concept Sexual functioning Sleep/wake functioning Social relationships (interaction with peers) Spirituality Stress experience Stigma Subjective well-being Upper extremity functioning (fine motor functioning and ability to perform activities of daily living)
patients and physicians to improve communication and manage chronic disease (NIH, n.d.). Among the nearly 290 publications that have used PROMIS, 26 of these investigations have addressed health outcomes for pediatric populations. Between the 2011 and 2012 only six publications were located in the literature for children using PROMIS. In contrast, 20 publications were located for 2013 and 2014 (up to July 2014). Bevans, Riley, Moon, and Forrest (2010) discussed in the use of PROMIS the advancements of conceptual and methodological advances in child-reported health outcomes. Bevans et al. (2010) concluded that rapid advances in building a solid scientific foundation for health-reported outcomes in children are a direct result of methodological standards for the assessment of patient-reported outcomes such as PROMIS. Clinical investigations using PROMIS have addressed a wide variety of health outcomes such as pain in the pediatric patient (Varni et al., 2010), health outcomes for children with asthma (Yeatts et al., 2010), cancerrelated fatigue in children (Barsevick et al., 2013), symptom patterns and functional impairment in children with cancer (Buckner et al., 2014), and biological and behavioral correlates of fatigue in adolescents and young adults with sickle cell anemia (Ameringer, Elswick, & Smith, 2014). In addition to these clinical investigations, methodological research examining psychometric properties of the studies by multidisciplinary teams of pediatric oncology researchers PROMIS scales has been done. January/February 2015
For children with chronic illness, HRQOL is particularly important, as they will live with their condition, or its sequelae into adulthood.
Menard et al. (2014) assessed the feasibility and acceptability of the PROMIS pediatric measures, among a cross-sectional study of 200 children in active cancer treatment and in a longitudinal study of 94 children with cancer ranging in ages of 8 to 18 years. Menard et al. (2014) reported that PROMIS measures demonstrated acceptability and feasibility as defined by enrollment and attrition rates as well as missingness by measure, item, participants, and assessment point in time and at different points in cancer treatment and was feasible to use in inpatient and outpatient pediatric oncology settings. Likewise, Hinds et al. (2013) reported PROMIS pediatric measures in pediatric oncology to be valid and clinically feasible indicators of patient-reported outcomes.
Conclusion Pediatric healthcare researchers have made HRQOL and health outcomes a central focus in the care and research base for chronically ill children and adolescents and their families. Data generated from the investigations of HRQOL among pediatric healthcare researchers make an important contribution to improve the overall health of the nation. With the existing incidence and prevalence of chronic illness in children and the continued rise in the prevalence of specific chronic health conditions that will persist in adulthood, the measurement of HRQOL as a central treatment and outcome variable is unquestionable. The research base in the conceptualization and measurement of HRQOL among chronically ill populations reflects systematic and rigorous science, which continues to evolve. The value of assessing HRQOL as an outcome in children is multidimensional. It is advantageous in MCN
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Clinical Nursing Implications: 1. Nurses of all specialties will benefit from a current understanding of HRQOL outcomes in children with chronic illness. 2. There is strong evidence that child self-report and parent proxy-report of the child’s HRQOL may be different, but both perspectives provide valuable insight into the health experience of the child. 3. Assessment, measurement, and discussion of HRQOL, as a patient-reported outcome, are advocated as important features in improving the health of children with chronic illness. 4. Objective, standardized HRQOL measurement tools, such as PROMIS, are vital to the evaluation of clinical interventions and therapies.
determining the efficacy of an intervention, in identifying progress in treatment, spurring discussions between family and healthcare provider, and capturing population data for policy decision making. Current research using individual scales that are tailored to chronically ill children’s specific symptoms to measure health outcomes, specifically PROMIS, is generating data with significant relevance in improving the care and lives of these children and is capturing population data for policy decision making. ✜ Mary Ann Cantrell is a Professor, College of Nursing, Villanova University, Driscoll Hall 338, 800 East Lancaster Avenue, Villanova, PA. She can be reached via e-mail at [email protected] Michelle M. Kelly is an Assistant Professor, College of Nursing, Villanova University, Driscoll Hall 398, 800 Lancaster Avenue, Villanova, PA. The authors declare no conflict of interest. DOI:10.1097/NMC.0000000000000090 References Ameringer, S., Elswick, R. K., Jr., & Smith, W. (2014). Fatigue in adolescents and young adults with sickle cell disease: Biological and behavioral correlates and health-related quality of life. Journal of Pediatric Oncology Nursing, 31(1), 6-17. doi:10.1177/1043454213514632 Barsevick, A. M., Irwin, M. R., Hinds, P., Miller, A., Berger, A., Jacobsen, P., …, Cella, D. (2013). Recommendations for high-priority research on cancer-related fatigue in children and adults. Journal of the National Cancer Institute, 105(19), 1432-1440. doi:10.1093/jnci/djt242 Bethell, C. D., Kogan, M. D., Strickland, B. B., Schor, E. L., Robertson, J., & Newacheck, P. W. (2011). A national and state profile of leading health problems and health care quality for US children: Key insurance disparities and across-state variations. Academic Pediatrics, 11(Suppl. 3), S22-S33. doi:10.1016/j.acap.2010.08.011 Bevans, K. B., Riley, A. W., Moon, J., & Forrest, C. B. (2010). Conceptual and methodological advances in child-reported outcomes measurement. Expert Review of Pharmacoeconomics & Outcomes Research, 10(4), 385-396. doi:10.1586/erp.10.52 Boyle, C. A., Boulet, S., Schieve, L. A., Cohen, R. A., Blumberg, S. J., Yeargin-Allsopp, M, …, Kogan, M. D. (2011). Trends in the prevalence of developmental disabilities in US children, 1997-2008. Pediatrics, 127(6), 1034-1042. doi:10.1542/peds.2010-2989 Buckner, T. W., Wang, J., DeWalt, D. A., Jacobs, S., Reeve, B. B., & Hinds, P. S. (2014). Patterns of symptoms and functional impairments in
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Retrieved July 19, 2014, from http://healthypeople.gov/2020/ topicsobjectives2020/overview.aspx?topicid=2 Upton, P., Lawford, J. & Eiser, C. (2008). Parent-child agreement across child health- related quality of life instruments: A review of the literature. Quality of Life Research, 17, 895-913. doi:10.1007/s11136-0089350-5 van der Lee, J. H., Mokkink, L. B., Grootenhuis, M. A., Heymans, H. S., & Offringa, M. (2007). Definitions and measurement of chronic health conditions in childhood: A systematic review. JAMA: The Journal of the American Medical Association, 297(24), 2741-2751. doi:10.1001/ jama.297.24.2741 Varni, J.W., Burwinkle, R.M., & Lane, M.M. (2005). Health-related quality of life measurement in pediatric clinical practice: An applictaion and precept for future research and applictaion. Health and Quality of Life Outcomes, 3(34). doi: 10.1186/1477-7525-3-34 Varni, J. W., & Limbers, C. A. (2009). The Pediatric Quality of Life Inventory: Measuring pediatric health-related quality of life from the perspective of children and their parents. Pediatric Clinics of North America, 56(4), 843-863. doi:10.1016/j.pcl.2009.05.016 Varni, J. W., Limbers, C. A., & Burwinkle, T. M. (2007). Impaired healthrelated quality of life in children and adolescents with chronic
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