Disability and Health Journal 7 (2014) 36e41 www.disabilityandhealthjnl.com
Review Article
A systematic framework to classify physical activity research for individuals with spina bifida Kerri A. Vanderbom, Ph.D.a,*, Simon Driver, Ph.D.b, and Mara Nery-Hurwit, M.S.b a
School of Health Professions, UAB/Lakeshore Research Collaborative, UAB & Lakeshore Foundation, Birmingham, AL, USA b Movement Studies in Disability, College of Public Health and Human Sciences, Oregon State University, USA
Abstract Background: Identifying the types of research conducted in the area of physical activity (PA) for the spina bifida (SB) population is important in order to move research forward to increase PA behaviors and improve health. The Behavioral Epidemiological Framework is a one way to systematically classify PA and SB literature by organizing research into one of five phases: phase 1 research links behaviors to health, phase 2 includes research focused on developing methods for measuring behavior, phase 3 research identifies factors that influence behavior, phase 4 research evaluates interventions to change the behavior, and phase 5 studies translates research into practice. Objective: To systematically classify PA research for individuals with SB by using the Behavioral Epidemiologic Framework and to identify where the research has focused. Method: An audit of the literature was conducted using search engines and keywords related to PA and SB. Inter-rater reliability was established between the research team coding articles based on established inclusion criteria. Finally, literature was categorized into one of the five phases. Results: Seventy-seven articles met the inclusion criteria and were categorized. Forty three percent of the articles were categorized in phase 1, 21% in phase 2, 32% in phase 3, 4% in phase 4, and zero in phase 5. Conclusion: The majority of articles are in phase 1, indicating that PA and SB research is still in early stages of development. Future research needs to move beyond phase 1 to examine factors that affect PA behaviors and ways to increase PA behaviors in the SB population. Ó 2014 Elsevier Inc. All rights reserved. Keywords: Spina bifida; Physical activity; Health promotion; Disability
Spina bifida and secondary conditions Spina bifida (SB) is the most common birth defect, affecting approximately 1e2 in 10,000 live births.1 SB occurs when the neural tube fails to fuse in the embryo2 and causes problems in four main areas: the central nervous system, the genitourinary tract, musculoskeletal system, and skin.3 Since the mid-1970s, there have been improvements in medical care, and individuals with SB are living longer. Consequently, the majority of the estimated 166,000 individuals in the US living with SB are adults.4,5 Since individuals with SB are living longer, problems with secondary conditions are becoming more apparent. Secondary conditions are preventable conditions associated with the primary diagnosis that an individual with spina bifida is at higher risk of developing.6 Examples of secondary Disclosures: The authors have nothing to disclose about finances or funding. There are no conflicts of interest. This review has not been presented, submitted or published previously. * Corresponding author. Tel.: þ1 541 250 0139. E-mail address: [email protected] (K.A. Vanderbom). 1936-6574/$ - see front matter Ó 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.dhjo.2013.09.002
conditions for the SB population include: obesity, cardiovascular disease, pressure sores, depression, and chronic pain.7e9 For young adults with SB, secondary conditions account for almost half of the health care problems in young adults with SB and are very expensive to treat.10 Due to the range of secondary conditions that can affect people with spina bifida the resultant health care costs are high, estimated to be approximately $560,000 across the lifetime.11 However, this is only an average and the costs for some are thought to be well over 1 million dollars.12 With the growing population of adults with spina bifida, the increased risk of developing secondary conditions, and escalating health care costs, there is a great need for interventions that promote healthy aging.13 Physical activity and spina bifida One method for decreasing the risk of developing secondary conditions and improving health is through physical activity (PA). The health benefits of PA are well documented,14e17 and PA is a leading indicator of health associated
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with decreased mortality and morbidity. Those who are more active have lower rates of coronary heart disease, depression, breast cancer, type II diabetes, metabolic syndrome, and stroke.18 For individuals with SB, PA is associated with more functional independence, higher quality of life,19 and higher self-worth.20 Further, greater functional independence is associated with better self-management and social competence.21 Despite the known benefits of PA, a sedentary lifestyle is common for individuals with SB,22e25 thus placing individuals at greater risk of experiencing secondary and chronic conditions. Therefore, it is essential for researchers to identify the links between PA behaviors and health, and to find ways to increase PA in the SB population. Behavioral Epidemiologic Framework Due to the recognized need to better understand and improve the health of individuals with a disability, there is a need to understand what types of research have been conducted examining the PA behaviors of individuals with SB, and where there is a need for further investigation. One method to establish a better understanding of the types of research that have been conducted in a specific area is an audit of the literature.26 Unlike a more traditional literature review, the aim of an audit is to use a framework to classify research studies into phases. Specifically, the Behavioral Epidemiological Framework is used to classify healthrelated research (e.g., PA behaviors for individuals with SB) into one of five phases: starting with basic research through to translating research into practice27 (see Table 1). The framework has been used previously to classify PA research for individuals with a spinal cord injury (SCI) and traumatic brain injury (TBI).28,29 Research in phase 1 establishes links between behaviors and health, and typically includes basic research such as doseeresponse relationships.27 An example would be developing guidelines for PA based on the doseeresponse relationship between the amounts of PA individuals with SB perform and the risk of secondary conditions. Research in phase 2 examines methods for measuring the behavior, establishing the reliability and validity of measures, and field-testing new instruments.27 For example, to measure PA behaviors for individuals with
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disabilities, The Physical Activity and Disability Survey (PADS)30 was developed and psychometric properties were assessed. Literature in phase 3 studies identifies correlates (e.g., demographic descriptors), or factors that influence the behavior, moderators and mediators of behavioral, psychological, or social variables of interest. An example of research in phase 3 would include research examining what personal and environmental barriers exist for individuals with SB to participate in PA. A second purpose of phase 3 is to test theoretical models related to behavior. Phase 4 research evaluates the efficacy or effectiveness of interventions that are focused on changing behavior. The interventions may focus on behavioral, psychological, or social variables related to health and the outcome of these studies must be a behavioral or biological marker.27 A good example of phase 4 research aimed at individuals with mobility impairments is Living Well with a Disability.31 This was an intervention that educated individuals with the goal of promoting health and reducing secondary conditions. Finally, studies in phase 5 discuss the dissemination process of the behavioral interventions identified in phase 4. Once an intervention has been shown to be effective, it can be translated into practice in a community setting, school, home, etc. The dissemination studies may include descriptive research, adoption, implementation, or sustainability of interventions in different settings, as well as policies.27 Living Well with a Disability31 was shown to be effective after being evaluated in phase 4, therefore was disseminated and adopted through community-based organizations throughout the United States. Each of the phases builds off the previous phase. For example, once a relationship has been established between the behavior (PA) and health in phase 1, better methods can be explored (in phase 2) in order to refine the results of phase 1. However, the relationships between each phase may be non-linear. For example, having reliable and valid measures (phase 2) can directly impact decisions about intervention methods (phase 4).27 It is important to note that the Behavioral Epidemiology Framework is a way to classify research in a given topic area and assesses the types of studies being conducted, but it does not evaluate the methodological, theoretical, or conceptual quality of the research being examined.27
Table 1 Description of each phase of the Behavioral Epidemiology Framework. Phase 1 Includes basic research that documents associations between behavior and health. Research in this phase may also include dose response relationships between the behavior and health outcomes. Phase 2 Establishes the reliability and validity of extant measures, developing new measures, and field-tests new tools. Phase 3 Describes demographic correlates of the behavior; how behavior varies by sex, age, etc. Research in this phase also includes examining moderators and mediators of behavioral, psychological, or social variables of interest. Phase 4 Evaluates the efficacy or effectiveness of interventions that are focused on changing behavior. The interventions may focus on behavioral, psychological, or social variables related to health and the outcome of these studies must be a behavioral or biological marker. Phase 5 The dissemination process of the behavioral interventions identified in phase 4. Effective interventions are translated into practice in a community setting, school, home, etc. Dissemination studies may include descriptive research, adoption, implementation, or sustainability of interventions in different settings. Studies may also examine policies and how they are implemented or maintained.
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Therefore, the purpose of this descriptive study was to use the Behavioral Epidemiology Framework to systematically classify studies on SB and PA into distinct phases to better understand the current state of research in this area. The two questions this paper will address include: (1) What is the current state of PA research for individuals with SB? and (2) What phases of PA research for individuals with SB warrant further development?
Method A survey of the literature was conducted using PubMed, EBSCOHOST, and Google Scholar. No limits were placed on the timeframe for the search and articles found ranged from 1968 to 2012. Key terms included in the search were ‘‘physical activity,’’ ‘‘exercise,’’ ‘‘spina bifida,’’ ‘‘myelomeningocele,’’ ‘‘health promotion,’’ ‘‘barriers,’’ ‘‘benefits,’’ facilitators,’’ and ‘‘theory.’’ The search included qualitative and quantitative studies, as well as meta-analyses and reviews. All of the articles identified in PubMed and EBSCOhost were reviewed, but articles reviewed in Google Scholar ended when the results page finished including articles related to the topic (approximately page 33). Studies were included if they related to SB and PA and fit into one of the five phases of the behavioral framework. Exclusion criteria included studies that focused on biomechanical outcomes of rehabilitation and physical therapy programs (e.g., wheelchair propulsion or gait analysis) versus PA related outcomes (e.g., behavior, measurement, or health risks). Abstracts of the studies were read to determine if they were SB and PA related. If the study was related, it was then read to determine what phase it should be categorized as. Coding rules used to categorize the studies were as follows: (a) editorials and commentaries without extensive references were not coded; (b) if the paper fit into multiple categories, the highest phase was coded; (c) if the paper dealt with measurement of behavior, phase 2 was coded; (d) if the paper dealt with measurement of potential influences or determinants, phase 3 was coded; and (e) if the paper dealt with measurement of factors related to dissemination, phase 5 was coded.27 Articles that met the inclusion criteria were saved using an online reference program, which had the capacity to check that articles were not duplicated. Further, once the final sample of articles was established, the research team checked for duplicates manually. The research team was trained on the phases of the behavioral framework. The training included reviewing the Sallis et al (2000) Behavioral Epidemiological Framework article, open-ended discussions, and practice coding PA and SB research articles into the different phases before the final coding. Out of the 77 articles that were identified, 18 articles (20%) were randomly selected to code to establish inter-rater reliability between two coders. The coders were required to reach at least 80% reliability classifying
the articles before completing the classification of the remaining articles. For this study the researchers coding reliability was 85%. Once reliability between the research team was established, the remaining articles were divided up and coded into one of the five phases. If a member of the research team was unsure about how to code an article, a consensus was reached to decide what phase it best fit into. The percentages of articles categorized in each of the five phases were computed. Further, the percentage of articles published by decade was calculated. Percentages were also calculated to examine the research articles by age (children and youth versus adults with SB, and research that combined both adults and youth) and the percentage of research articles that included mixed disabilities versus a homogenous group of individuals with SB.
Results One hundred nine articles were initially identified in PubMed, 154 in EBSCOhost, and over 20,000 articles in Google Scholar. After conducting the review, a total of 77 articles were identified that fit the SB and PA criteria and were categorized into one of the five phases of the Behavioral Epidemiological Framework. Forty three percent were categorized in phase 1, 21% in phase 2, 32% in phase 3, 4% in phase 4, and 0% in phase 5. Fig. 1 displays the percentage of articles in each phase. Once the research was broken down by age range, 62% of the research focused on children and youth, 20% focused on adults, and 18% included both youth and adults (Fig. 2). When examining the articles by disability, 64% were SB specific and the remaining 32% of the articles included other disabilities such as cerebral palsy (CP) and SCI. When the number of articles that included mixed disability types was broken down by age, 62% of the articles were child and youth research and 16% were adult focused. The remaining 19% of the articles included both children and adults with a mix of disabilities in their samples. Only three articles examining adults consisted of a sample exclusively of individuals with SB.
Fig. 1. Percent of articles in the 5 phases of the behavioral epidemiologic framework.
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Fig. 2. Percent of articles by age groupings in phases 1e5.
Fig. 3 displays the percentage of articles in each decade from the 1970’s to the present. Over half of the articles were published from 2000 to 2010 (60%) and 13% of the research articles were from 2010 to 2012.
Discussion This audit of literature categorized PA and SB research utilizing the Behavioral Epidemiological Framework.27 The majority of research focusing on PA and SB is in phase 1 (43%), indicating that this is a young area of research.27 This is not surprising, as public health efforts focused for so long on the prevention of SB, not health promotion of individuals living with SB.32 Similar results were found when examining phase 1 research for PA and SCI (55%)28 and for TBI (44%),29 indicating that PA and disability research in general is in the early stages of development. Over 70% of PA and SB research was conducted from 2000 to the present, which further demonstrates that this field is still in its early stages of development. Since a large portion of individuals with SB are living longer and aging, secondary conditions are becoming more evident and the consequences of a sedentary lifestyle are more apparent. Phase 1 research shows links between participating in PA and improved mental and physical
Fig. 3. Percent of spina bifida and physical activity research articles published by decade.
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health outcomes. For example, active individuals with SB, compared to those with SB who are not active, report more functional independence and a higher quality of life.19 Also, those who reported more time spent participating in PA and sports had higher athletic competence, physical appearance, and self-worth.33 While phase 1 research is important, building on this phase and examining methods of measuring PA behavior, and determinants of PA are also critical in order to conduct interventions and put them into practice. As recommended by Sawin et al. (2010), research needs to move away from identifying differences in the health of those with SB and those without, and move on to why those differences occur and how they are prevented. There were only 16 studies (21%) in phase 2, indicating that there is a need to establish valid and reliable methods of measuring behaviors, and other variables of interest. If there are not valid and reliable measures available then there is not a good foundation for research to be conducted in phases 1, 3, 4 and 5. While there are 32% of articles in phase 3, a large majority focused on identifying factors that influence PA behaviors. There were very few that focused on the second purpose of phase 3-validating theoretical models and the use of theorydriven constructs. There needs to be more theory-driven research since theory helps inform the development of interventions. Examining factors that emphasize cognitive and motivational strategies (i.e., using the social cognitive theory or transtheoretical model), is important for longterm behavior changes.34 There were only three articles in phase 4 (two focused on youth and one article focused on adults), demonstrating that there are very few PA or health promotion intervention studies for individuals with SB. This is not unexpected, as health promotion programs for individuals with disabilities in general are still in the early phases of development.35 Nery and colleagues28 reported that only 5% of PA and SCI research was in phase 4, while Pawlowski and colleagues29 reported that 6% of PA and TBI research was in phase 4. The one intervention study that focused on adults only included a variety of disabilities, and it was reported that only 2.8 percent of the participants had SB.36 Implementing more phase 4 research will be important in order to evaluate the effectiveness of PA behavior interventions in the SB population period after ‘‘population’’ before being translated and utilized within a community (phase 5). There were zero studies classified as phase 5, indicating that there are no health promotion programs being practiced regularly for individuals with SB. Research needs to go beyond testing interventions and disseminate interventions in order to prevent adverse health outcomes. The lack of translational research is alarming since we know that individuals with SB are at high risk of developing secondary conditions and health care costs are very high. Additionally, very few of the total articles (n 5 77) focused solely on individuals with SB. Sixty two percent were samples of SB only, while the other 32% were mixed
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samples of SB, SCI, CP, etc. Once the PA research is broken up by age it is very clear that there is a lack of research focusing solely on adults with SB. Eighty percent of all of the research articles focusing on adults and PA included a variety of disabilities. Since the majority of individuals living with SB are adults, it is more important than ever to conduct SB specific research to meet the unique needs of this population. When examining the number of articles by decade, there is a trend that the percentage of articles is increasing with each passing decade. One could hypothesize from this trend that there will be even more research performed and published during this decade than the last one. Based on the current review, it is recommended that future research should focus on phases 2, 3, 4, and 5 since it has been established that there are links between behaviors and health (phase 1). Moving out of phase 1 research and performing research in phases 2e5 is important in order to increase the PA behaviors and health in the SB population. The lack of SB specific research examining benefits of PA and health promotion programs are not surprising as it is recognized in general health research that there are many gaps pertaining to knowledge, programs and services for individuals with SB and their families across the life span in three domains: self-management/health, social and personal relationships, and employment/income support.37 PA fits within the first two domains and may be an important mode to improve both physical and mental health in the SB population. To improve health, there is a great need to translate PA research into practice for the SB population. Specifically, there is a need for PA interventions and programs offered for both children and adults with SB. For adults with SB, there is a great need for research in all phases. This study is not without its limitations. There were specific key terms used to search for articles related to SB and PA. Consequently, articles utilizing other terms related to PA and SB health may have been missed. Also, published research examining interventions and dissemination efforts (phases 4 and 5) may not have specified the disability of the sample population (e.g., used a generic term like ‘‘mobility disability’’ instead), ensuring that articles would not be included. However, even if some research was not included, it can be hypothesized from other SB health topics that the results would remain similar to what was found hereethat there is a lack of translational research in the area of SB and PA. Finally, there were a limited number of search engines used to identify articles and other research may have been identified in other engines. However, with the databases utilized in the current study, saturation was noted with the same articles appearing repeatedly. Conclusions We have described the steps taken to examine SB PA research utilizing the Behavioral Epidemiological Framework. The Framework systematically organizes research
into one of five categories, from linking behaviors to health to the dissemination of interventions.27 From the results, there is a clear need for specific research in the development, implementation and maintenance of PA health promotion programs for the SB population. There is an even greater need for more PA research in phases 2e5 for adults with SB. If research does not progress forward into the other phases (i.e., developing reliable and valid measures, using theory, testing and disseminating interventions) secondary conditions and health care costs will remain high, increasing the likelihood of a poor quality of life for the SB population. References 1. Boulet SL, Yang Q, Mai C, et al. Trends in the postfortification prevalence of spina bifida and anencephaly in the United States. Birth Defects Res A Clin Mol Teratol. 2008;82(7):527e532. 2. Patten BA. Embryological stages in the establishing of myeloschisis with spina bifida. Am J Anat. 1953;93(3):365e395. 3. Harris MB, Banta JV. Cost of skin care in the myelomeningocele population. J Pediatr Orthop. 1990;10:355e361. 4. Ouyang L, Grosse SD, Armour BS, Waitzman NJ. Health care expenditures of children and adults with spina bifida in a privately insured US population. Birth Defects Res A Clin Mol Teratol. 2007;79(7): 552e558. 5. Liptak GS, El Samra A. Optimizing health care for children with spina bifida. Dev Disabil Res Rev. 2010;16(1):66e75. 6. Krahn GL, Hammond L, Turner A. A cascade of disparities: health and health care access for people with intellectual disabilities. Ment Retard Dev Disabil Res Rev. 2006;12(1):70e82. 7. Mahmood D, Dicianno B, Bellin M. Self-management, preventable conditions and assessment of care among young adults with myelomeningocele. Child Care Health Dev. 2011;37(6):861e865. 8. Dopler-Nelson M, Widman LM, Abresch RT, et al. Metabolic syndrome in adolescents with spinal cord dysfunction. J Spinal Cord Med. 2007;30:S127eS139. 9. Toward a state of well-being: promoting healthy behaviors to prevent secondary conditions. Preventing Secondary Conditions Associated with Spina Bifida and Cerebral Palsy: Proceedings and Recommendations of a Symposium. Washington, D.C.: Spina Bifida Association of America; 1994. 10. Kinsman SL, Doehring MC. The cost of preventable conditions in adults with spina bifida. Eur J Pediatr Surg. 1996;6(1):17e20. 11. Grosse SD, Ouyang L, Collins JS, Green D, Dean JH, Stevenson RE. Economic evaluation of a neural tube defect recurrence-prevention program. Am J Prev Med. 2008;35(6):572e577. 12. Spina Bifida Association. FAQ: What are the Costs Associated With Spina Bifida?; 2011. Available at: http://www.spinabifidaassociation. org/site/c.liKWL7PLLrF/b.2700315/k.C2AE/What_Are_The_Costs_ Associated_With_Spina_Bifida.htm. Accessed 20.01.13. 13. Dicianno BE, Kurowski BG, Yang JMJ, et al. Rehabilitation and medical management of the adult with spina bifida. Am J Phys Med Rehabil. 2008;87(12):1027e1050. 14. Haskell WL, Lee IM, Pate RR, et al. Physical activity and public health: updated recommendation for adults from the American College of Sports Medicine and the American Heart Association. Med Sci Sports Exerc. 2007;39(8):1423. 15. Wannamethee SG, Shaper AG, Walker M. Changes in physical activity, mortality, and incidence of coronary heart disease in older men. Lancet. 1998;351(9116):1603e1608. 16. Blair SN, Kohl HW III, Paffenbarger RS Jr, Clark DG, Cooper KH, Gibbons LW. Physical fitness and all-cause mortality. JAMA. 1989;262(17):2395e2401.
K.A. Vanderbom et al. / Disability and Health Journal 7 (2014) 36e41 17. Paffenbarger RS Jr, Hyde R, Wing AL, Hsieh C. Physical activity, allcause mortality, and longevity of college alumni. N Engl J Med. 1986;314(10):605e613. 18. U.S. Department of Health and Human Services. Physical Activity Advisory Committee. Physical Activity Guidelines Advisory Committee Report, 2008. Washington, DC: US Department of Health and Human Services 2008; 2008. 19. Buffart LM, van den Berg-Emons RJ, van Meeteren J, Stam HJ, Roebroeck ME. Lifestyle, participation, and health-related quality of life in adolescents and young adults with myelomeningocele. Dev Med Child Neurol. 2009 Nov;51(11):886e894. 20. Buffart LM, van der Ploeg HP, Bauman AE, et al. Sports participation in adolescents and young adults with myelomeningocele and its role in total physical activity behaviour and fitness. J Rehabil Med. 2008 Oct;40(9):702e708. 21. Sawin KJ, Buran CF, Brei TJ, Fastenau PS. Correlates of functional status, self-management, and developmental competence outcomes in adolescents with spina bifida. SCI Nurs. 2003 Summer;20(2):72e85. 22. Buffart LM, van den Berg-Emons RJ, van Wijlen-Hempel MS, Stam HJ, Roebroeck ME. Health-related physical fitness of adolescents and young adults with myelomeningocele. Eur J Appl Physiol. 2008 May;103(2):181e188. 23. Schoenmakers M, Helders P, Gorter J, Takken T. Exercise tolerance, level of physical activity and muscle strength in independent ambulators with Sacral Myelomeningocele. Cerebrospinal Fluid Res. 2005;2. S49. 24. van den Berg-Emons HJ, Bussmann JB, Meyerink HJ, Roebroeck ME, Stam HJ. Body fat, fitness and level of everyday physical activity in adolescents and young adults with meningomyelocele. J Rehabil Med. 2003;35(6):271e275. 25. van den Berg-Emons HJ, Bussmann JB, Brobbel AS, Roebroeck ME, van Meeteren J, Stam HJ. Everyday physical activity in adolescents and young adults with meningomyelocele as measured with a novel activity monitor. J Pediatr. 2001 Dec;139(6):880e886. 26. Webster J, Watson RT. Analyzing the past to prepare for the future: writing a literature review. MIS Q. 2002;26(2):xiii. 27. Sallis JF, Owen N, Fotheringham MJ. Behavioral epidemiology: a systematic framework to classify phases of research on health promotion and disease prevention. Ann Behav Med. 2000;22(4):294e298.
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28. Nery MB, Driver S, Vanderbom KA. A systematic framework to classify the status of research on spinal cord injury and physical activity. Arch Phys Med Rehabil. 2013;94(10):2027e2031. 29. Pawlowski J, Dixon-Ibarra A, Driver S. Review of the status of physical activity research for individuals with traumatic brain injury. Arch Phys Med Rehabil. 2013;94(6):1184e1189. 30. Rimmer JH, Riley BB, Rubin SS. A new measure for assessing the physical activity behaviors of persons with disabilities and chronic health conditions: the Physical Activity and Disability Survey. Am J Health Promot. 2001 Sep-Oct;16(1):34e42. 31. Ravesloot C, Seekins T, White G. Living Well with a Disability health promotion intervention: improved health status for consumers and lower costs for health care policymakers. Rehabil Psychol. 2005;50(3):239. 32. Patrick DL, Richardson M, Starks HE, Rose MA. A framework for promoting health of people with disabilities. In: Lollar D, ed. Preventing Secondary Conditions Associated with Spina Bifida or Cerebral Palsy: Proceedings and Recommendations of a Symposium. Washington, D.C.: Spina Bifida Association of America; 1994:3e16. 33. Buffart LM, van den Berg-Emons RJ, Burdorf A, Janssen WG, Stam HJ, Roebroeck ME. Cardiovascular disease risk factors and the relationships with physical activity, aerobic fitness, and body fat in adolescents and young adults with myelomeningocele. Arch Phys Med Rehabil. 2008 Nov;89(11):2167e2173. 34. Kosma M, Ellis R, Bauer JJ. Longitudinal changes in psychosocial constructs and physical activity among adults with physical disabilities. Disabil Health J. 2012 Jan;5(1):1e8. 35. Drum CE, Peterson JJ, Culley C, et al. Guidelines and criteria for the implementations of community-based health promotion programs for individuals with disabilities. Am J Health Promot. 2009;24(2): 93e101. 36. Ravesloot C, Seekins T, Cahill T, Lindgren S, Nary D, White G. Health promotion for people with disabilities: development and evaluation of the Living Well with a Disability program. Health Educ Res. 2007;22(4):522e531. 37. Sawin KJ, Betz CL, Linroth R. Gaps and opportunities: an agenda for further research, services, and program development in spina bifida. Pediatr Clin North Am. 2010;57(4).
Review Article
A systematic framework to classify physical activity research for individuals with spina bifida Kerri A. Vanderbom, Ph.D.a,*, Simon Driver, Ph.D.b, and Mara Nery-Hurwit, M.S.b a
School of Health Professions, UAB/Lakeshore Research Collaborative, UAB & Lakeshore Foundation, Birmingham, AL, USA b Movement Studies in Disability, College of Public Health and Human Sciences, Oregon State University, USA
Abstract Background: Identifying the types of research conducted in the area of physical activity (PA) for the spina bifida (SB) population is important in order to move research forward to increase PA behaviors and improve health. The Behavioral Epidemiological Framework is a one way to systematically classify PA and SB literature by organizing research into one of five phases: phase 1 research links behaviors to health, phase 2 includes research focused on developing methods for measuring behavior, phase 3 research identifies factors that influence behavior, phase 4 research evaluates interventions to change the behavior, and phase 5 studies translates research into practice. Objective: To systematically classify PA research for individuals with SB by using the Behavioral Epidemiologic Framework and to identify where the research has focused. Method: An audit of the literature was conducted using search engines and keywords related to PA and SB. Inter-rater reliability was established between the research team coding articles based on established inclusion criteria. Finally, literature was categorized into one of the five phases. Results: Seventy-seven articles met the inclusion criteria and were categorized. Forty three percent of the articles were categorized in phase 1, 21% in phase 2, 32% in phase 3, 4% in phase 4, and zero in phase 5. Conclusion: The majority of articles are in phase 1, indicating that PA and SB research is still in early stages of development. Future research needs to move beyond phase 1 to examine factors that affect PA behaviors and ways to increase PA behaviors in the SB population. Ó 2014 Elsevier Inc. All rights reserved. Keywords: Spina bifida; Physical activity; Health promotion; Disability
Spina bifida and secondary conditions Spina bifida (SB) is the most common birth defect, affecting approximately 1e2 in 10,000 live births.1 SB occurs when the neural tube fails to fuse in the embryo2 and causes problems in four main areas: the central nervous system, the genitourinary tract, musculoskeletal system, and skin.3 Since the mid-1970s, there have been improvements in medical care, and individuals with SB are living longer. Consequently, the majority of the estimated 166,000 individuals in the US living with SB are adults.4,5 Since individuals with SB are living longer, problems with secondary conditions are becoming more apparent. Secondary conditions are preventable conditions associated with the primary diagnosis that an individual with spina bifida is at higher risk of developing.6 Examples of secondary Disclosures: The authors have nothing to disclose about finances or funding. There are no conflicts of interest. This review has not been presented, submitted or published previously. * Corresponding author. Tel.: þ1 541 250 0139. E-mail address: [email protected] (K.A. Vanderbom). 1936-6574/$ - see front matter Ó 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.dhjo.2013.09.002
conditions for the SB population include: obesity, cardiovascular disease, pressure sores, depression, and chronic pain.7e9 For young adults with SB, secondary conditions account for almost half of the health care problems in young adults with SB and are very expensive to treat.10 Due to the range of secondary conditions that can affect people with spina bifida the resultant health care costs are high, estimated to be approximately $560,000 across the lifetime.11 However, this is only an average and the costs for some are thought to be well over 1 million dollars.12 With the growing population of adults with spina bifida, the increased risk of developing secondary conditions, and escalating health care costs, there is a great need for interventions that promote healthy aging.13 Physical activity and spina bifida One method for decreasing the risk of developing secondary conditions and improving health is through physical activity (PA). The health benefits of PA are well documented,14e17 and PA is a leading indicator of health associated
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with decreased mortality and morbidity. Those who are more active have lower rates of coronary heart disease, depression, breast cancer, type II diabetes, metabolic syndrome, and stroke.18 For individuals with SB, PA is associated with more functional independence, higher quality of life,19 and higher self-worth.20 Further, greater functional independence is associated with better self-management and social competence.21 Despite the known benefits of PA, a sedentary lifestyle is common for individuals with SB,22e25 thus placing individuals at greater risk of experiencing secondary and chronic conditions. Therefore, it is essential for researchers to identify the links between PA behaviors and health, and to find ways to increase PA in the SB population. Behavioral Epidemiologic Framework Due to the recognized need to better understand and improve the health of individuals with a disability, there is a need to understand what types of research have been conducted examining the PA behaviors of individuals with SB, and where there is a need for further investigation. One method to establish a better understanding of the types of research that have been conducted in a specific area is an audit of the literature.26 Unlike a more traditional literature review, the aim of an audit is to use a framework to classify research studies into phases. Specifically, the Behavioral Epidemiological Framework is used to classify healthrelated research (e.g., PA behaviors for individuals with SB) into one of five phases: starting with basic research through to translating research into practice27 (see Table 1). The framework has been used previously to classify PA research for individuals with a spinal cord injury (SCI) and traumatic brain injury (TBI).28,29 Research in phase 1 establishes links between behaviors and health, and typically includes basic research such as doseeresponse relationships.27 An example would be developing guidelines for PA based on the doseeresponse relationship between the amounts of PA individuals with SB perform and the risk of secondary conditions. Research in phase 2 examines methods for measuring the behavior, establishing the reliability and validity of measures, and field-testing new instruments.27 For example, to measure PA behaviors for individuals with
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disabilities, The Physical Activity and Disability Survey (PADS)30 was developed and psychometric properties were assessed. Literature in phase 3 studies identifies correlates (e.g., demographic descriptors), or factors that influence the behavior, moderators and mediators of behavioral, psychological, or social variables of interest. An example of research in phase 3 would include research examining what personal and environmental barriers exist for individuals with SB to participate in PA. A second purpose of phase 3 is to test theoretical models related to behavior. Phase 4 research evaluates the efficacy or effectiveness of interventions that are focused on changing behavior. The interventions may focus on behavioral, psychological, or social variables related to health and the outcome of these studies must be a behavioral or biological marker.27 A good example of phase 4 research aimed at individuals with mobility impairments is Living Well with a Disability.31 This was an intervention that educated individuals with the goal of promoting health and reducing secondary conditions. Finally, studies in phase 5 discuss the dissemination process of the behavioral interventions identified in phase 4. Once an intervention has been shown to be effective, it can be translated into practice in a community setting, school, home, etc. The dissemination studies may include descriptive research, adoption, implementation, or sustainability of interventions in different settings, as well as policies.27 Living Well with a Disability31 was shown to be effective after being evaluated in phase 4, therefore was disseminated and adopted through community-based organizations throughout the United States. Each of the phases builds off the previous phase. For example, once a relationship has been established between the behavior (PA) and health in phase 1, better methods can be explored (in phase 2) in order to refine the results of phase 1. However, the relationships between each phase may be non-linear. For example, having reliable and valid measures (phase 2) can directly impact decisions about intervention methods (phase 4).27 It is important to note that the Behavioral Epidemiology Framework is a way to classify research in a given topic area and assesses the types of studies being conducted, but it does not evaluate the methodological, theoretical, or conceptual quality of the research being examined.27
Table 1 Description of each phase of the Behavioral Epidemiology Framework. Phase 1 Includes basic research that documents associations between behavior and health. Research in this phase may also include dose response relationships between the behavior and health outcomes. Phase 2 Establishes the reliability and validity of extant measures, developing new measures, and field-tests new tools. Phase 3 Describes demographic correlates of the behavior; how behavior varies by sex, age, etc. Research in this phase also includes examining moderators and mediators of behavioral, psychological, or social variables of interest. Phase 4 Evaluates the efficacy or effectiveness of interventions that are focused on changing behavior. The interventions may focus on behavioral, psychological, or social variables related to health and the outcome of these studies must be a behavioral or biological marker. Phase 5 The dissemination process of the behavioral interventions identified in phase 4. Effective interventions are translated into practice in a community setting, school, home, etc. Dissemination studies may include descriptive research, adoption, implementation, or sustainability of interventions in different settings. Studies may also examine policies and how they are implemented or maintained.
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Therefore, the purpose of this descriptive study was to use the Behavioral Epidemiology Framework to systematically classify studies on SB and PA into distinct phases to better understand the current state of research in this area. The two questions this paper will address include: (1) What is the current state of PA research for individuals with SB? and (2) What phases of PA research for individuals with SB warrant further development?
Method A survey of the literature was conducted using PubMed, EBSCOHOST, and Google Scholar. No limits were placed on the timeframe for the search and articles found ranged from 1968 to 2012. Key terms included in the search were ‘‘physical activity,’’ ‘‘exercise,’’ ‘‘spina bifida,’’ ‘‘myelomeningocele,’’ ‘‘health promotion,’’ ‘‘barriers,’’ ‘‘benefits,’’ facilitators,’’ and ‘‘theory.’’ The search included qualitative and quantitative studies, as well as meta-analyses and reviews. All of the articles identified in PubMed and EBSCOhost were reviewed, but articles reviewed in Google Scholar ended when the results page finished including articles related to the topic (approximately page 33). Studies were included if they related to SB and PA and fit into one of the five phases of the behavioral framework. Exclusion criteria included studies that focused on biomechanical outcomes of rehabilitation and physical therapy programs (e.g., wheelchair propulsion or gait analysis) versus PA related outcomes (e.g., behavior, measurement, or health risks). Abstracts of the studies were read to determine if they were SB and PA related. If the study was related, it was then read to determine what phase it should be categorized as. Coding rules used to categorize the studies were as follows: (a) editorials and commentaries without extensive references were not coded; (b) if the paper fit into multiple categories, the highest phase was coded; (c) if the paper dealt with measurement of behavior, phase 2 was coded; (d) if the paper dealt with measurement of potential influences or determinants, phase 3 was coded; and (e) if the paper dealt with measurement of factors related to dissemination, phase 5 was coded.27 Articles that met the inclusion criteria were saved using an online reference program, which had the capacity to check that articles were not duplicated. Further, once the final sample of articles was established, the research team checked for duplicates manually. The research team was trained on the phases of the behavioral framework. The training included reviewing the Sallis et al (2000) Behavioral Epidemiological Framework article, open-ended discussions, and practice coding PA and SB research articles into the different phases before the final coding. Out of the 77 articles that were identified, 18 articles (20%) were randomly selected to code to establish inter-rater reliability between two coders. The coders were required to reach at least 80% reliability classifying
the articles before completing the classification of the remaining articles. For this study the researchers coding reliability was 85%. Once reliability between the research team was established, the remaining articles were divided up and coded into one of the five phases. If a member of the research team was unsure about how to code an article, a consensus was reached to decide what phase it best fit into. The percentages of articles categorized in each of the five phases were computed. Further, the percentage of articles published by decade was calculated. Percentages were also calculated to examine the research articles by age (children and youth versus adults with SB, and research that combined both adults and youth) and the percentage of research articles that included mixed disabilities versus a homogenous group of individuals with SB.
Results One hundred nine articles were initially identified in PubMed, 154 in EBSCOhost, and over 20,000 articles in Google Scholar. After conducting the review, a total of 77 articles were identified that fit the SB and PA criteria and were categorized into one of the five phases of the Behavioral Epidemiological Framework. Forty three percent were categorized in phase 1, 21% in phase 2, 32% in phase 3, 4% in phase 4, and 0% in phase 5. Fig. 1 displays the percentage of articles in each phase. Once the research was broken down by age range, 62% of the research focused on children and youth, 20% focused on adults, and 18% included both youth and adults (Fig. 2). When examining the articles by disability, 64% were SB specific and the remaining 32% of the articles included other disabilities such as cerebral palsy (CP) and SCI. When the number of articles that included mixed disability types was broken down by age, 62% of the articles were child and youth research and 16% were adult focused. The remaining 19% of the articles included both children and adults with a mix of disabilities in their samples. Only three articles examining adults consisted of a sample exclusively of individuals with SB.
Fig. 1. Percent of articles in the 5 phases of the behavioral epidemiologic framework.
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Fig. 2. Percent of articles by age groupings in phases 1e5.
Fig. 3 displays the percentage of articles in each decade from the 1970’s to the present. Over half of the articles were published from 2000 to 2010 (60%) and 13% of the research articles were from 2010 to 2012.
Discussion This audit of literature categorized PA and SB research utilizing the Behavioral Epidemiological Framework.27 The majority of research focusing on PA and SB is in phase 1 (43%), indicating that this is a young area of research.27 This is not surprising, as public health efforts focused for so long on the prevention of SB, not health promotion of individuals living with SB.32 Similar results were found when examining phase 1 research for PA and SCI (55%)28 and for TBI (44%),29 indicating that PA and disability research in general is in the early stages of development. Over 70% of PA and SB research was conducted from 2000 to the present, which further demonstrates that this field is still in its early stages of development. Since a large portion of individuals with SB are living longer and aging, secondary conditions are becoming more evident and the consequences of a sedentary lifestyle are more apparent. Phase 1 research shows links between participating in PA and improved mental and physical
Fig. 3. Percent of spina bifida and physical activity research articles published by decade.
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health outcomes. For example, active individuals with SB, compared to those with SB who are not active, report more functional independence and a higher quality of life.19 Also, those who reported more time spent participating in PA and sports had higher athletic competence, physical appearance, and self-worth.33 While phase 1 research is important, building on this phase and examining methods of measuring PA behavior, and determinants of PA are also critical in order to conduct interventions and put them into practice. As recommended by Sawin et al. (2010), research needs to move away from identifying differences in the health of those with SB and those without, and move on to why those differences occur and how they are prevented. There were only 16 studies (21%) in phase 2, indicating that there is a need to establish valid and reliable methods of measuring behaviors, and other variables of interest. If there are not valid and reliable measures available then there is not a good foundation for research to be conducted in phases 1, 3, 4 and 5. While there are 32% of articles in phase 3, a large majority focused on identifying factors that influence PA behaviors. There were very few that focused on the second purpose of phase 3-validating theoretical models and the use of theorydriven constructs. There needs to be more theory-driven research since theory helps inform the development of interventions. Examining factors that emphasize cognitive and motivational strategies (i.e., using the social cognitive theory or transtheoretical model), is important for longterm behavior changes.34 There were only three articles in phase 4 (two focused on youth and one article focused on adults), demonstrating that there are very few PA or health promotion intervention studies for individuals with SB. This is not unexpected, as health promotion programs for individuals with disabilities in general are still in the early phases of development.35 Nery and colleagues28 reported that only 5% of PA and SCI research was in phase 4, while Pawlowski and colleagues29 reported that 6% of PA and TBI research was in phase 4. The one intervention study that focused on adults only included a variety of disabilities, and it was reported that only 2.8 percent of the participants had SB.36 Implementing more phase 4 research will be important in order to evaluate the effectiveness of PA behavior interventions in the SB population period after ‘‘population’’ before being translated and utilized within a community (phase 5). There were zero studies classified as phase 5, indicating that there are no health promotion programs being practiced regularly for individuals with SB. Research needs to go beyond testing interventions and disseminate interventions in order to prevent adverse health outcomes. The lack of translational research is alarming since we know that individuals with SB are at high risk of developing secondary conditions and health care costs are very high. Additionally, very few of the total articles (n 5 77) focused solely on individuals with SB. Sixty two percent were samples of SB only, while the other 32% were mixed
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samples of SB, SCI, CP, etc. Once the PA research is broken up by age it is very clear that there is a lack of research focusing solely on adults with SB. Eighty percent of all of the research articles focusing on adults and PA included a variety of disabilities. Since the majority of individuals living with SB are adults, it is more important than ever to conduct SB specific research to meet the unique needs of this population. When examining the number of articles by decade, there is a trend that the percentage of articles is increasing with each passing decade. One could hypothesize from this trend that there will be even more research performed and published during this decade than the last one. Based on the current review, it is recommended that future research should focus on phases 2, 3, 4, and 5 since it has been established that there are links between behaviors and health (phase 1). Moving out of phase 1 research and performing research in phases 2e5 is important in order to increase the PA behaviors and health in the SB population. The lack of SB specific research examining benefits of PA and health promotion programs are not surprising as it is recognized in general health research that there are many gaps pertaining to knowledge, programs and services for individuals with SB and their families across the life span in three domains: self-management/health, social and personal relationships, and employment/income support.37 PA fits within the first two domains and may be an important mode to improve both physical and mental health in the SB population. To improve health, there is a great need to translate PA research into practice for the SB population. Specifically, there is a need for PA interventions and programs offered for both children and adults with SB. For adults with SB, there is a great need for research in all phases. This study is not without its limitations. There were specific key terms used to search for articles related to SB and PA. Consequently, articles utilizing other terms related to PA and SB health may have been missed. Also, published research examining interventions and dissemination efforts (phases 4 and 5) may not have specified the disability of the sample population (e.g., used a generic term like ‘‘mobility disability’’ instead), ensuring that articles would not be included. However, even if some research was not included, it can be hypothesized from other SB health topics that the results would remain similar to what was found hereethat there is a lack of translational research in the area of SB and PA. Finally, there were a limited number of search engines used to identify articles and other research may have been identified in other engines. However, with the databases utilized in the current study, saturation was noted with the same articles appearing repeatedly. Conclusions We have described the steps taken to examine SB PA research utilizing the Behavioral Epidemiological Framework. The Framework systematically organizes research
into one of five categories, from linking behaviors to health to the dissemination of interventions.27 From the results, there is a clear need for specific research in the development, implementation and maintenance of PA health promotion programs for the SB population. There is an even greater need for more PA research in phases 2e5 for adults with SB. If research does not progress forward into the other phases (i.e., developing reliable and valid measures, using theory, testing and disseminating interventions) secondary conditions and health care costs will remain high, increasing the likelihood of a poor quality of life for the SB population. References 1. Boulet SL, Yang Q, Mai C, et al. Trends in the postfortification prevalence of spina bifida and anencephaly in the United States. Birth Defects Res A Clin Mol Teratol. 2008;82(7):527e532. 2. Patten BA. Embryological stages in the establishing of myeloschisis with spina bifida. Am J Anat. 1953;93(3):365e395. 3. Harris MB, Banta JV. Cost of skin care in the myelomeningocele population. J Pediatr Orthop. 1990;10:355e361. 4. Ouyang L, Grosse SD, Armour BS, Waitzman NJ. Health care expenditures of children and adults with spina bifida in a privately insured US population. Birth Defects Res A Clin Mol Teratol. 2007;79(7): 552e558. 5. Liptak GS, El Samra A. Optimizing health care for children with spina bifida. Dev Disabil Res Rev. 2010;16(1):66e75. 6. Krahn GL, Hammond L, Turner A. A cascade of disparities: health and health care access for people with intellectual disabilities. Ment Retard Dev Disabil Res Rev. 2006;12(1):70e82. 7. Mahmood D, Dicianno B, Bellin M. Self-management, preventable conditions and assessment of care among young adults with myelomeningocele. Child Care Health Dev. 2011;37(6):861e865. 8. Dopler-Nelson M, Widman LM, Abresch RT, et al. Metabolic syndrome in adolescents with spinal cord dysfunction. J Spinal Cord Med. 2007;30:S127eS139. 9. Toward a state of well-being: promoting healthy behaviors to prevent secondary conditions. Preventing Secondary Conditions Associated with Spina Bifida and Cerebral Palsy: Proceedings and Recommendations of a Symposium. Washington, D.C.: Spina Bifida Association of America; 1994. 10. Kinsman SL, Doehring MC. The cost of preventable conditions in adults with spina bifida. Eur J Pediatr Surg. 1996;6(1):17e20. 11. Grosse SD, Ouyang L, Collins JS, Green D, Dean JH, Stevenson RE. Economic evaluation of a neural tube defect recurrence-prevention program. Am J Prev Med. 2008;35(6):572e577. 12. Spina Bifida Association. FAQ: What are the Costs Associated With Spina Bifida?; 2011. Available at: http://www.spinabifidaassociation. org/site/c.liKWL7PLLrF/b.2700315/k.C2AE/What_Are_The_Costs_ Associated_With_Spina_Bifida.htm. Accessed 20.01.13. 13. Dicianno BE, Kurowski BG, Yang JMJ, et al. Rehabilitation and medical management of the adult with spina bifida. Am J Phys Med Rehabil. 2008;87(12):1027e1050. 14. Haskell WL, Lee IM, Pate RR, et al. Physical activity and public health: updated recommendation for adults from the American College of Sports Medicine and the American Heart Association. Med Sci Sports Exerc. 2007;39(8):1423. 15. Wannamethee SG, Shaper AG, Walker M. Changes in physical activity, mortality, and incidence of coronary heart disease in older men. Lancet. 1998;351(9116):1603e1608. 16. Blair SN, Kohl HW III, Paffenbarger RS Jr, Clark DG, Cooper KH, Gibbons LW. Physical fitness and all-cause mortality. JAMA. 1989;262(17):2395e2401.
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