PROTOCOL: Education Support Services for Improving School Engagement and Academic Performance of Children and Adolescents with a Chronic Health Condition: A Systematic Review

Abstract

Chronic health conditions in children and adolescents can have profound impacts on education as well as health and well-being. While there are many different definitions of what constitutes a chronic health condition or chronic disease, the World Health Organization (WHO) described chronic diseases as being of “long duration and generally slow progression” (WHO, 2013). O'Halloran 2004 provided a more detailed definition and suggested that chronic diseases may have a duration that has lasted, or is expected to last, at least six months; have a pattern of recurrence or deterioration; have a poor prognosis and produce consequences or sequelae that impact on the individual's quality of life. The Centers for Disease Control and Prevention (CDC) defined chronic diseases as “noncommunicable illnesses that are prolonged in duration, do not resolve spontaneously, and are rarely cured completely” (CDC, 2009). Chronic diseases are the leading cause of death globally (Yach, 2004). Although chronic health conditions have been more commonly linked to developed countries, chronic health conditions also impact people from developing countries, with death rates from chronic diseases exceeding those of communicable diseases in some low-income countries including India and Pakistan (WHO, 2005). Research that focuses specifically on children and adolescents with chronic health conditions in developing countries is limited; however, there is evidence to suggest that chronic conditions such as obesity and type 2 diabetes are experienced globally (WHO, 2005). Children and adolescents now experience higher levels of chronic health conditions than in the past (Canter, 2012; Newacheck, 1992; Van Cleave, 2010), with increasing rates noted in both developed and developing countries (Michaud, 2007). In part, this reflects technological and medical advances that result in children now living longer with complex health conditions. It also reflects increases in the incidence of certain conditions such as chronic inflammatory bowel disease, allergy, type 1 and 2 diabetes, together with increasing diagnosis of developmental and mental disorders in children (e.g., autism) and adolescents (e.g. eating disorders, depression and anxiety) (Sawyer, 2007). A wide range of different chronic conditions affect children and adolescents, including conditions of congenital onset (e.g. cystic fibrosis), acquired illnesses (e.g., asthma, cancer), mental and developmental disorders, and disabilities (e.g., cerebral palsy). Due to the variety of both different chronic conditions and definitions, it is not easy to gain clear figures for the prevalence of chronic health conditions in children and adolescents. One systematic review by van der Lee (2007) reviewed 32 studies that reported prevalence rates of chronic health conditions in children and adolescents and reported prevalence estimates of chronic health conditions between 0.22% and 44%; the majority of these studies were from the US, one was from a middle-income country (i.e., 7.8% of children aged zero to seven years in Jordan had a chronic disease or disability), and none were from low-income countries. Further, not all chronic conditions have poor sequelae. Sequelae appear to be related to the severity of the chronic health condition, and the impact on cognitive and functional capacities. Of people with a chronic health condition, an estimated 5% are affected by severe conditions characterised by limitations to daily activities and frequent bother (Newacheck, 1992). In one large New Zealand study of over 9000 high-school students (most of whom were aged 13 to 17 years), 18% reported having a chronic health condition (Denny, 2014). As many as 28% of students with a chronic health condition reported that their condition negatively impacted their daily activities and 8% noted difficulties in socialising due to their chronic health condition (Denny, 2014), thus emphasising the importance of considering the impact of chronic health conditions on daily life and functioning in addition to the prevalence of these conditions. The impact of chronic health conditions on children's quality of life is described more specifically by Martinez 2009 who suggested that besides the physiological consequences of chronic health conditions, chronic health conditions for children carry secondary psychological and educational consequences. Existing research identifies a wide range of impacts of chronic health conditions for children and young people including effects on school absenteeism, school engagement, school functioning, academic and social-emotional development, and well-being. For example, rates of school absenteeism appear to be higher for children and adolescents with chronic health conditions compared with non-ill peers. Daraganova (2013) found that 18% of students (aged six to seven years) with a medical condition/disability lasting for at least six months had missed school for three or more days. In comparison, 14% of healthy peers had missed school for three or more days. The same author found that 41% of children aged six to seven years with a medical condition were absent from school for one or two days compared with 33% of healthy peers. McDougall (2004) found that children with a chronic illness were absent from school for a mean of 16 days as compared with three days for their healthy peers. Further, it should be kept in mind that some, but not all, children and adolescents with a chronic health condition miss school due to their illness. Both the presence and the severity of chronic health conditions appear related to the extent of school absenteeism. For instance, Moonie 2006 found that children and adolescents with asthma missed a mean of 9.2 days of school per year compared with children and adolescents without asthma who missed 7.9 days of school per year. However, when severity of asthma was considered, pupils with severe persistent asthma were away from school more frequently (11.6 days) than pupils with moderate persistent asthma (10.3 days), mild persistent asthma (11.3 days), mild intermittent asthma (8.5 days) and without asthma (7.9 days). While these differences for asthma appear to be small, Shaw 2008 noted that some chronic conditions take a profound toll on school attendance. For example, “children being treated for chronic childhood leukemia miss an average of 40 school days during the first stages of treatment and have inconsistent attendance for 3 years that follow” (Shaw, 2008, p. 74). School attendance, which is generally higher for primary school children compared with secondary school students (Hancock, 2013), is important for academic and social skill development. When children and adolescents are absent from school due to a chronic health condition, school engagement can be affected, as can the achievement of important educational milestones and the acquisition of knowledge and skills. Chronic health conditions and severity are also associated with academic performance. For instance, Cadman (1987) showed that the odds of not doing well at school for children and adolescents with a chronic health condition alone were not significant (odds ratio 1.3), but were significant for students with a chronic health condition and a disability (odds ratio 4.7). Some chronic health conditions such as cerebral palsy may have an associated cognitive impairment that can directly affect school functioning. However, children and adolescents with chronic health conditions that are not associated with a cognitive impairment can still experience difficulties at school due to school absenteeism, adjustment and emotional problems, and the impact of the chronic health condition and its treatment on the individual such as fatigue (Madan-Swain, 2008). Subsequently, children with a chronic health condition are at greater risk of a range of negative school-related outcomes such as poorer academic achievement and lower motivation to do well at school (Forrest, 2011). Similar to school absenteeism, the degree of these associations may vary across chronic health conditions and according to severity of these conditions. For example, asthma is a common chronic health condition, affecting approximately 12% of children and adolescents aged zero to 14 years in Australia (Australian Bureau of Statistics, 2006). It is also the most common reason for hospitalisation in this age group (Australian Institute of Health and Welfare, 2005). While children and adolescents with asthma report lower school functioning (e.g., experience greater difficulty in concentrating, forgetting things, keeping up with school work, missing school) than healthy peers (Varni, 2007), it has been found that the odds of graduating from high school or being employed were similar for students with asthma compared with their healthy counterparts (Maslow, 2011). By contrast, students with a non-asthmatic chronic health condition (e.g., cancer) were less likely to graduate or be employed compared with healthy peers or students with asthma (Maslow, 2011). Compared with asthma, cancer is far less common. The incidence of childhood cancer in the U.S. was 14.3 per 100,000 children in 2007, while over 175,000 new cases of childhood cancers were reported worldwide in 2008 (American Cancer Society, 2011). Cancer is a serious chronic disease, contributing to 16% of deaths in children and adolescents aged one to 14 years (Australian Institute of Health and Welfare, 2005). Children and adolescents with cancer report poorer school functioning and lower health-related quality of life compared with their healthy peers (Varni, 2007). Children and adolescents with cancer may miss school because of acute treatment or because of complications of the cancer and its treatment (or both) (Prevatt, 2000). A number of studies reported that children and adolescents with cancer encountered higher rates of school absenteeism compared with healthy students or students with other chronic conditions (refer to Vance, 2002 for a review on this topic). Education support services for children and adolescents with chronic health conditions who miss school due to their illness aim to prevent them from becoming disengaged from school, education and learning. Disengagement from school is associated with poorer academic achievement, social emotional functioning and career choices (Abbott-Chapman, 2013; Bond, 2007; Hancock, 2013). Education support services provide academic support with the goal that students are able to perform either as well as their non-ill counterparts or to their fullest potential. Importantly, all children and adolescents have a right to education, including children and adolescents who require hospitalisation due to a chronic illness (Ratnapalan, 2009). Access to education is a fundamental human right for children and adolescents that is enshrined in international law. According to international law, countries have obligations of making available primary education that is compulsory and free to all, secondary education that is generally available and accessible to all, and access to educational programmes on the basis of non-discrimination and equality of educational opportunity (Hodgson, 2012). As such, governments around the world, most often through their ministries or departments of education, invest significant amounts in education support services for children and adolescents with chronic health conditions, especially in children's hospitals. Delivering education support to children and adolescents with a chronic health condition is, therefore, considered an important step in terms of equity and in assisting school-aged students with chronic health conditions to reach their academic potential and to become successful and engaged learners. However, there is very limited evidence on the effectiveness of educational support interventions in improving school engagement and educational outcomes for children and adolescents with chronic health conditions and a scarcity of research evidence outlining best practice models. Moreover, much of the evidence relates to a particular diagnostic illness category with few studies investigating the common impacts of chronic health conditions and effectiveness of education support interventions more generally. Such a non-categorical approach is useful in public policy decision-making in which interventions may be provided universally with the expectation that they will be tailored at the local level to suit specific needs or conditions. Chronic health conditions in children and adolescents are generally managed throughout the healthcare system, including primary care (e.g., general practice), community-based specialist services (e.g., community health centres, private paediatricians) and hospitals. However, educational support programmes for children and adolescents with chronic health conditions may be provided within either the healthcare or education systems. As such, this review will investigate the effectiveness of educational interventions across the healthcare and education systems at a global scale. This review will include educational support interventions delivered to children and adolescents with a chronic health condition who may miss school due to illness, some of whom may or may not have been hospitalised due to their chronic illness. As such, educational support interventions may be provided in settings external to regular school environments (e.g., children's hospitals, community settings or in the home). We will include educational interventions that target one or more of the four sites of hospital, home, community setting or school in this review. This approach is consistent with Daraganova (2013), who stated that, “to improve school attendance, interventions should be implemented at different levels (i.e., individual, family and community) and targeted toward specific subgroups in the population” (p. 75). Educational support may be delivered by hospital-based school teachers, community-based school teachers or by teachers from a student's regular school. Educational support aims to assist children and adolescents who are unable to attend their regular school because of illness to meet their educational needs and goals despite absence from school, with efforts often focusing on facilitating a smooth re-entry to school (Kaffenberger, 2006; Madan-Swain, 2008). Education support may include one-to-one or group tuition and is often highly personalised. Figure 1 provides a logic model depicting how we hypothesise education support interventions for children and adolescents might work. The logic model includes the inputs, modifiable factors that the interventions may target, and the logical/theorised intermediary and primary outcomes that are aimed to be affected by the intervention. Educational support interventions for children and adolescents with chronic health conditions may be delivered in the hospital, school, home or community setting, and may target the child, family, school or hospital staff (e.g., multidisciplinary teams). They may be provided by a child's parent(s) or carer(s), the child's regular school (sometimes referred to as the ‘home school’), the regional/district school authority, the hospital (in the case of long periods of inpatient stays or frequent outpatient visits), private provider organisations or community-based organisations. This approach is consistent with a socio-ecological model that acknowledges the complex interplay between the systems in which children and adolescents are immersed, and the influence of these systems on their development. This review will focus on education support interventions that at minimum have direct involvement with the child (i.e., the sphere of service delivery is at least at the level of the child). These interventions could take the form of one or more of the following formats in either one-to-one or group sessions: face-to-face interaction, online interactions, synchronised or asynchronised, or virtual classroom interactions. These features of the intervention are captured as inputs in the logic model, information about which we will gather in this review to help inform our investigation of the nature of educational support interventions. Educational support interventions delivered to children and adolescents with a chronic health condition aim to target some of the known modifiable predictors of engagement and academic achievement. These modifiable factors are identified in the logic model as attitudes to school, school connectedness and anxiety, each of which will be described below in the context of how we propose educational support interventions might work. Research has shown that students’ attitudes to school, including motivation and approach to learning, are important predictors of school engagement, including ‘drop-out’, and academic achievement (Froiland, 2014; Hillman, 2010; Li-Grining, 2010). Educational support interventions for children and adolescents with chronic health conditions aim to build a sense of connectedness to school staff and peers. This is because “research has shown that students who feel more accepted, included and involved in their school are more likely to be engaged in classroom learning, in extracurricular activities, in interpersonal relationships, and in the wider school community” (Robinson, 2014, p. 14). School connectedness refers to the quality and number of connections students have with ‘place’ (the school) and ‘people’ (teachers, other school staff and peers) (Bond, 2007; Maslow, 2012; Robinson, 2014). Improving school connectedness is important as research has linked feelings of belonging and connectedness to a range of school outcomes including aspects of school engagement and achievement (see review by Osterman, 2000). Maslow (2012) found that, for students with chronic health conditions, school connectedness was an important predictor of academic achievement (i.e., college graduation). Furthermore, social support (e.g., having close friends) has been identified as an important factor that assists young people to manage the effects of their chronic illness on school functioning (Lightfoot, 1999; Shiu, 2004). A student's connectedness to school and their peers has also been linked to later mental health and academic outcomes (Bond, 2007; Pittman, 2007). Children and adolescents with certain chronic health conditions (e.g., chronic fatigue syndrome, epilepsy and asthma) are known to experience heightened levels of anxiety (Pinquart, 2011). Many children and adolescents with a chronic health condition, their parents and their teachers are also concerned about the effects of having a chronic health condition on school work, on keeping connected to peers and peer relationships, and school attendance (Shiu, 2004; Yates, 2010). High levels of intense stress and anxiety, if not managed well, can affect functioning and impede learning, with the risk of negatively affecting school functioning and academic performance. Educational support interventions may aim to alleviate some of the psychological stress and anxiety associated with the effects of the chronic health condition thereby reducing the ‘impairment’ of the condition and assist in building adaptive functioning (Power, 2006). Furthermore, educational support delivered to children and adolescents with chronic health conditions aims to provide students with ongoing learning opportunities, despite not being able to attend their regular school because of illness. This continuity in learning promotes and supports students to keep up-to-date with their school work or learning goals so that academic performance and progress is not negatively impacted due to school absenteeism. Reducing anxiety through receipt of educational support may, therefore, lead to improved mental health and better academic functioning given that mental health problems and school functioning are clearly related (DeSocio, 2004). However, we also acknowledge that anxiety can of itself be a type of mental illness and research has indicated that anxiety disorders are related to lower levels of educational participation (DeSocio, 2004; Woodward, 2001). Modifying attitudes to school, school connectedness and anxiety through educational support is, therefore, hypothesised to lead to improved mental health, better quality of life and may facilitate a smoother transition to school (i.e., adjusting to school life after having been absent from school due to a chronic health condition). We identify these as intermediary outcomes as they can of themselves have a direct relationship with school engagement and academic achievement. Underpinning this logic model is the context in which the intervention is delivered that can influence not only the intervention itself, but also the modifiable factors, intermediary outcomes and primary outcomes. These contextual factors may include severity of illness and degree of cognitive impairment and physical disability associated with the chronic health condition, as well as if the condition is a mental health or physical health condition. They may include potential features of the population such as age/grade level. We have also selected features from the PROGRESS (Place of residence, Race/ethnicity/culture/language, Occupation, Gender/sex, Religion, Education, Socioeconomic status, and Social capital; O'Neill, 2014) framework, to collect information so to, where possible, perform subgroup analysis including gender, place of residence, parental occupation, parental education and socioeconomic status. The purpose of this review is to examine and describe the current state of the evidence about the effectiveness of education support services for children and adolescents with a chronic health condition. To date, there appears to be little research detailing the types and effectiveness of models of educational support services for children and adolescents with chronic health conditions. From scoping searches, the evidence appears limited to qualitative studies, expert opinions, case studies and literature reviews. The quality of this literature has not been reported. Much of the evidence is limited to specific conditions (e.g., Prevatt (2000) reviewed school reintegration programmes for children with cancer). One exception is a literature review conducted by Canter (2012), which explored the effectiveness of school reintegration interventions for children with chronic health conditions. Canter found that these interventions were more effective in increasing knowledge about the child's illness than changing attitudes about chronic health conditions. Moderate improvements in reports of self worth of the ill/injured child were also noted after interventions in four studies included in the Canter review. The approach used in Canter (2012) differs from the current systematic review in a number of ways. Canter examined school re-entry programmes for ill or injured children, whereas our review will not focus on injured children unless their injury is associated with, or results in, a chronic health condition. Canter examined interventions specific to an injured/ill child's return to school, whereas our review will adopt a broader examination of educational support interventions provided to children and adolescents with a chronic health condition while in hospital, at home or at school re-entry. Finally, Canter examined school reintegration programmes and their effect on the outcomes of changing knowledge about, and attitudes towards, chronic illnesses primarily among the ill-student's teachers and peers. Our review will examine the impact of educational support services on school engagement and academic attainment outcomes as experienced by students with chronic health conditions who have received educational support services. To our knowledge, there are no systematic reviews on this topic. In conducting our review, we are motivated by a desire to build the knowledge base about effective models of education support for children and adolescents with a chronic health condition. Information derived from this review can inform current and future research, practice and policy decisions and is expected to be of interest to those who fund or deliver (or both) educational support programmes to children and adolescents with chronic health conditions. Findings from this review may inform future improved models of educational support interventions. Improving models of educational support not only has potential for enhancing students’ educational trajectories, but also has clinical relevance, as it is well acknowledged that education influences health and well-being (Cohen, 2013). Given the interplay between education and health, it is imperative that education features in the holistic provision of care of a child or adolescent with a chronic health condition. This review is also expected to provide an initial examination of the evidence on this topic, which in addition to highlighting research gaps and future research opportunities, will also provide an opportunity to capture developments over time in future updates of the review. To describe the nature of educational support interventions for children and adolescents with a chronic health condition, and to examine the effectiveness of these educational support interventions on school engagement and academic achievement. We will include randomised controlled trials (RCTs) in our review as they are well understood to be the ideal method for evaluating the efficacy and effectiveness of healthcare interventions (National Forum on Early Childhood, 2007; O'Connor, 2011). Importantly, RCTs are considered less vulnerable to bias compared with other study designs (Lewis, 2004); however, there are sometimes ethical and feasibility issues that limit the utility of RCTs (Roland, 1998). It is unlikely that RCTs will be the sole research study design used to evaluate the effectiveness of educational support interventions for children and adolescents with chronic health conditions. As such, we will also include non-randomised studies, which are studies that use non-random methods to allocate people to treatment arms (Reeves, 2011). It is possible that we will encounter non-randomised trials, as it is often not desirable or ethical (or both) to randomise participants systematically into different conditions in school and hospital settings. We will consider controlled before-and-after studies for inclusion in our review. Controlled before-and-after studies incorporate a control group with similar characteristics to the intervention group; however, the group allocation is based on a non-random method. In addition, the intervention and control groups are assessed before and after the intervention. Finally, our review will include studies using an interrupted time series (ITS) design. This study design involves measuring outcomes in people on multiple occasions before the intervention to identify underlying trends and on multiple occasions after the intervention. The observations collected after the intervention provide information as to whether the observed trend noted post intervention is different to that of the underlying trend. ITS may or may not have a control group. We expect to find other study designs in our search (e.g., cross-sectional studies); however, due to the potential high risk of bias associated with these studies and the questionable reliability of evidence produced from such studies, we will not include them in our review. However, we will record the different types of studies that we find and exclude from our review so that we can comment on the current state of evidence in this field. We understand that both RCTs and non-randomised studies may be susceptible to bias, and as such, we will examine and report risk of bias for all included studies. For studies involving a control group, we will accept studies where group allocation was conducted at the cluster level. We will also include qualitative studies if they were conducted within the context of an included quantitative intervention study. We will report on qualitative data if the qualitative information is about the effectiveness of the intervention or factors influencing intervention effectiveness (e.g., enablers or inhibitors). Qualitative information on factors that may influence intervention effectiveness has the potential to provide information about why an intervention may have been effective or ineffective, which we believe would be of interest to readers. We will exclude standalone qualitative studies from our review. Participants will include children and adolescents aged four to 18 years who have a chronic illness or chronic health condition as defined in Description of the condition. We also note that in the US the term ‘special healthcare needs’ is often used either instead of, or to refer to, people with chronic health conditions. While not widely used outside of the US, we will include studies on children and adolescents with special healthcare needs on the condition that the population in the study is described as having a ‘chronic’ health condition. We justify this as the definition of special healthcare needs is broader than that of chronic illness. Children with special healthcare needs are “those who have or are at increased risk for a chronic physical, developmental, behavioral, or emotional condition and who also require health and related services of a type or amount beyond that required by children generally” (McPherson, 1998, p. 138). In this definition, the presence of a chronic health condition is considered together with the degree of support required to aide healthy functioning. As such, children and adolescents with a chronic health condition, disability, mental disorder (or a combination of these) could be captured under the broad umbrella term of special healthcare