Abstract
Celiac disease (CD) is a chronic autoimmune disease precipitated by exposure to gluten (proteins found in wheat, barley, and rye) and characterized by damage to the small intestine resulting from the ingestion of gluten (Ludvigsson et al., 2013). Prevalence of CD varies across different countries and is estimated to affect approximately 1%–2% of the adult and pediatric population (Fasano et al., 2003; Ludvigsson et al., 2015; Mustalahti et al., 2010). Despite its varying prevalence, CD is one of the most common chronic diseases in children. Maintaining a lifelong gluten-free diet (GFD) is the only treatment available today for those diagnosed with CD (Green & Cellier, 2007). Adaptation to a GFD, involving strict dietary limitations, can be difficult, has financial implications, and may impair quality of life (QOL; Hill et al., 2005). Lack of adherence to a GFD could affect as many as two-thirds of people with CD, and adherence may be especially challenging for children and adolescents (Branski, 2012; Newton & Singer, 2012). Compliance in the pediatric population is often unsatisfactory (Nachman et al., 2010).
Health-related quality of life (HRQOL) involves the implications of various chronic diseases on QOL and the person’s perception of the impact of the condition on his or her objective functioning and subjective assessment of well-being (Eisen, Locke, & Provenzale, 1999; Grootenhuis, Koopman, Verrips, Vogels, & Last, 2007). Measuring children’s HRQOL presents a challenge because perceptions may change during childhood and adolescence (Solans et al., 2008). Previous research worldwide has found disparities between the self-reported HRQOL of children with various chronic conditions, including CD, and their parents’ perceptions of their HRQOL, with parents tending to rate their children’s HRQOL lower than do the children (e.g., Pico, Spirito, & Roizen, 2012; van Doorn, Winkler, Zwinderman, Mearin, & Koopman, 2008). Such findings emphasize the need to understand both perceptions and to provide children the opportunity to express their own perception apart from their parents. Although the prevalence of CD in Israel is similar to that worldwide (Shamir et al., 2002), to the best of our knowledge, no local research to date has been published regarding self-reported HRQOL of children with CD.
Health is supported and promoted by engagement or participation in everyday activities in the home, school, work, and social environments, and participation is an important outcome for occupational therapy (American Occupational Therapy Association [AOTA], 2014; Piškur, 2014). In addition, leisure is a core concept in occupational therapy. It plays a central role in building children’s competence and self-determination, social and personality development, and adolescents’ initiative, which occurs during structured leisure activities such as sports, hobbies, and arts (AOTA, 2014; Larson, 2000).
The relationship among human functioning, disability, and health is described in the International Classification of Functioning, Disability and Health (ICF; World Health Organization [WHO], 2001) and in the Children and Youth version of the ICF (WHO, 2007). According to the ICF and the Occupational Therapy Practice Framework: Domain and Process (3rd ed.; AOTA, 2014), autoimmune diseases and digestive system body functions are among the client factors that may affect occupational performance. Although CD clearly fits this classification, no theoretical or clinical reference has been found that relates to occupational therapy practice and CD. This occupation-based study was developed in light of the HRQOL multidimensional approach. Eating is one of life’s meaningful activities and takes place in various environments and life contexts, including social encounters (Contento, Williams, Michela & Franklin, 2006; Segal, 2009). Therefore, in the case of CD, engagement in food-related leisure occupations is discussed.
The purpose of the current study was to compare the HRQOL perceptions of children with CD with their parents’ perceptions and to evaluate their overall and specific food-related leisure participation characteristics to examine the relationships between leisure participation and HRQOL.
Method
Participants
Children with CD ages 8–15 yr and their parents were recruited by publication through the Israeli Celiac Association and via local online CD support forums. Only children whose parents reported strict adherence to a GFD, with negative antibodies or antibodies in the process of decline and with neither physical nor neurological disabilities, were included in the study. A control group included friends of the study group participants, without CD or any other diagnosed medical condition, matched on age, gender, place of residence, and educational and social environment criteria. Informed written consent was obtained from both the children and their parents. The university’s institutional ethics committee approved this cross-sectional study.
Measures
Parents completed a demographic questionnaire for both themselves and their children (e.g., age, gender, CD health status, education level).
Celiac Disease DUX.
The Celiac Disease DUX (CDDUX; van Doorn et al., 2008) is a disease-specific HRQOL self-report questionnaire for children with CD ages 8–18 yr. It is used to obtain information about how children feel about their condition. This 12-item validated questionnaire is accompanied by a validated proxy version that parents complete about their children’s feelings. The CDDUX has three distinct subscales: Diet, which concerns how the child feels about lifelong dietary adherence; Communication, which concerns how the child feels when speaking about CD; and Having CD, which concerns the child’s feelings when offered or thinking about food containing gluten. Responses are rated on a five-picture facial expression Likert scale on which a higher score indicates a better quality of life. The HRQOL outcome consists of a score on each of the three scales and a total score. The CDDUX was translated into Hebrew (translation and back-translation) with the authors’ permission. The internal reliability of the translated version of the CDDUX was found to be acceptable for the total scores on the child form (Cronbach’s α = .86) and the parent form (Cronbach’s α = .81). Scale scores also showed acceptable levels of internal reliability for the child form (Cronbach’s α ranged from .75 to .85) and the parent form (Cronbach’s α ranged from .63 to .80).
Children’s Leisure Assessment Scale.
The Children’s Leisure Assessment Scale (CLASS; Rosenblum, Sachs, & Schreuer, 2010) is a validated multidimensional scale that examines school-age children’s engagement in leisure activities such as watching TV, dancing, playing ball games, and going to a restaurant. Activities are evaluated on four leisure participation dimensions: (1) variety of activities; (2) frequency (how often), rated on a 4-point Likert scale (1 = once in a few months, 2 = once a month, 3 = once or twice a week, 4 = everyday); (3) sociability (with whom), rated on a 4-point Likert scale (1 = alone, 2 = with a relative [e.g., parent, sibling], 3 = with a friend, 4 = with a few friends); and (4) preference, rated on a scale ranging from 1 to 10 (1 = not like at all and 10 = like very much ; Rosenblum et al., 2010). The CLASS has four activity factors defining the type of activity. For the purpose of this study, an additional dummy factor, food-related activities, was created that included four of the original CLASS activities: going to a restaurant, visiting relatives, picnic, and shopping at malls (Cronbach’s α = .60). As with the original four factors, the food-related activities were analyzed according to the four leisure dimensions (variety, frequency, sociability, and preference).
Procedure
Parents who responded to the recruitment call were contacted by telephone, and the study aim and procedure were explained. The requirement that parents and children complete their questionnaires separately was emphasized. Each participant recruited a friend, whose parents were then contacted, thus creating the control group. Questionnaires and research contact details were mailed together with an addressed and stamped return envelope. All parents completed the demographic questionnaire, children with CD and their parents each completed the CDDUX, and all the children completed the CLASS.
Data Analysis
Data were analyzed with IBM SPSS Statistics (Version 19; IBM Corp., Armonk, NY). Descriptive statistics were used to describe the participants. The internal reliabilities of the translated version of the CDDUX and the food-related activities factor of the CLASS were determined with Cronbach’s α. Multivariate analysis of variance was performed to test for differences in children’s and parents’ CDDUX scale scores and group CLASS scores. Paired t tests were used to compare children’s and parents’ CCDUX total scores, and the significance level was set at .05. The nonparametric Wilcoxon signed-rank test was used to analyze differences between each of the CDDUX’s 12 items. Finally, a Pearson correlation coefficient was calculated to test correlation between HRQOL and leisure participation.
Results
Demographics
Sixty-eight informants participated in this study: children ages 8–15 yr diagnosed with CD at least 6 mo before recruitment (n = 34) and their parents (n = 34). The matched control group of children also numbered 34. The mean (M) ages of the CD group and the matched control group were 10.5 yr (standard deviation [SD] = 2.4) and 10.6 yr (SD = 2.3), respectively, and 73.5% were female. Parents reported CD diagnosis by means of biopsy (88.2%) or blood tests (11.8%), and present CD medical status was determined by the presence of negative antibodies (82.4%) and antibodies in decline (17%). The children had been diagnosed between ages 1 and 13 yr, with the majority diagnosed between ages 4 and 6 yr (44.1%). No significant differences were found between the groups in children’s general health status, parents’ age (CD group, M = 42.1 yr for mothers, M = 44.6 yr for fathers; control group, M = 41.3 yr for mothers and M = 44.1 yr for fathers) or mothers’ years of education (CD group, M = 16.6 yr; control group, M = 15.7 yr).
Comparison Between Children’s and Parents’ Reports
As shown in Table 1, parents’ ratings on the Diet and Having CD scales showed that they perceived their children’s HRQOL significantly more negatively than did children, with both groups having lower ratings on the Having CD scale than on the Diet scale. Yet, no significant difference in perceptions was found on the Communication scale. Results of the paired t tests between the children’s and parents’ ratings on the three scales are also presented in Table 1. A significant correlation was found between age and QOL for children’s ratings on the Having CD scale (r = .39, p = .02). However, no significant correlation was found between children’s age and QOL for either the children’s total score (r = .06, p = .06) and scale scores or the parents’ total score (r = .11, p = .54) and all other scale scores.
Means, Standard Deviations, and Differences in Quality of Life of Children With CD
Note. 1–20 = very bad, 21–40 = bad, 41–60 = neutral, 61–80 = good, 81–100 = very good. — = not applicable. CD = celiac disease; CDDUX = Celiac Disease DUX; M = mean; SD = standard deviation.
p < .05. **p < .01.
Comparison of Specific Celiac Disease DUX Items
Because this is a primary study of children with CD in Israel and to deepen the understanding of the children’s and parents’ perceptions, a comparison between the two groups was performed on each CDDUX item, and the results are displayed in Table 2. A highly significant difference was found on Item 4 (p = .002) and Item 12 (p = .004). The children’s rating was higher than the parents’ rating on these items. In addition, significant differences were found on Items 6, 8, and 9. The item rated highest by both the children and parents was “Talking about celiac disease.” An interesting finding was that the children’s report of “Not being able to eat just everything I want, I feel . . .” yielded the lowest score. In contrast, the parents thought that their children felt worst about being given food containing gluten at school.
Comparison of CDDUX Items as Reported by Children and Parents
Note. CD = celiac disease; CDDUX = Celiac Disease DUX; M = mean; SD = standard deviation.
p < .05.
Comparison of Leisure Participation for Children With and Without Celiac Disease
No significant differences were found between the two groups of children in the amount of leisure participation, F(4, 63) = 0.444, p ≥ .05, η2 = .027, or on any of the four CLASS dimensions. In addition, no significant differences were found between the groups specifically concerning food-related activities, F(3, 68) = 0.970, p ≥ .05, η2 = .003.
Correlation Between Health-Related Quality of Life and Leisure Participation Among Children With Celiac Disease
A significant correlation was found in the children’s self-reports between the sociability dimension of the CLASS food-related activities factor and the total CDDUX score (r = .465, p = .006) and the three scales (Table 3).
Correlations Between CLASS Food Activities and Quality of Life of Children With CD
Note. n = 34. CD = celiac disease; CDDUX = Celiac Disease DUX; CLASS = Children’s Leisure Assessment Scale.
p < .05. **p < .01.
Discussion
This study presents HRQOL self-reports of children with CD and parent reports in Israel and is the first study to associate HRQOL with CD and an occupational view of participation. As expected, Israeli children reported a higher HRQOL than their parents’ proxy report. A significant finding was that children rated their own HRQOL as good (average total score = 62.6) and their parents perceived their children’s HRQOL to be neutral or less than good (57.6). Parents’ central role in supervising and developing new required dietary habits and maintaining a long-term dietary routine may play a role in their different experience.
Among all populations, people recognize the benefits of a healthy diet at different points in their lifetime (Persch, Lamb, Metzler, & Fristad, 2015). For children with food limitations, regardless of the child’s age at time of diagnosis, parents are mainly responsible for their children’s food consumption, especially in situations outside of the home (Marklund, Ahlstedt, & Nordström, 2006). Generally, young children require more mediation and supervision, yet during childhood and adolescence changes occur in the transactional interaction between the person, his or her everyday life activities (occupations), and the environments (AOTA, 2014; Law et al., 1996).
In the context of CD, condition-specific interactions are reflected in changes in the habits and daily routines of children and adolescents while participating in food-related occupations in the changing environments and conditions in which they take place. Development of health management routines, such as nutritional routines, promotes health and wellness (AOTA, 2014). As they develop, children and adolescents with CD need sophistication and refinement in instrumental activities of daily living, such as health management and maintenance in activities that take place in wider, more diverse environments and further away from home (AOTA, 2014; Rodger & Brown, 2006; Segal, 2009).
Adolescence is characterized by the need for a sense of security from parents combined with a need for personal identity and autonomy, the move toward independence from parents, and the ultimate desire to belong and to be the same as their peers (Vroman, 2014). Although these are typical developmental processes, children and adolescents with CD are confronted with food-related experiences and challenges that may influence their participation in age-related occupations. Their parents are confronted with challenging difficulties concerning cooperation with educational staff, friends, and family members about their children’s dietary needs (Cederborg, Hultman, & Magnusson, 2012). These challenges may cause the differences found in this study between parents’ and children’s responses, resulting in the parents’ lower perceptions of their child’s HRQOL.
The findings also correspond with evidence of differences between children and parents concerning disease-related perceptions, indicating that parents tend to perceive their child’s HRQOL more negatively than do the children themselves (Byström, Hollén, Fälth-Magnusson, & Johansson, 2012; Kurppa, Collin, Mäki, & Kaukinen, 2011; Upton, Lawford, & Eiser, 2008). This divergence emphasizes the importance of hearing children’s voice to profoundly understand their perception of QOL and daily experiences.
Observation of the scale scores revealed that highest scores on the Communication scale obtained from children and parents in Israel were similar to those previously reported in other countries (Pico et al., 2012; van Doorn et al., 2008). This scale involves talking to other people about CD and explaining it to them. On this scale, the best reported feeling referred to talking about CD with peers. CD is an externally invisible disease, yet its daily management requires cooperation and communication with the environment (Olsson, Lyon, Hörnell, Ivarsson, & Sydner, 2009). In fact, the relatively high scores on this scale may represent the children’s and parents’ comfort with sharing their nutritional needs with their environment. However, the poorer feeling expressed on the other two scales may reflect the everyday reality in which the social or cultural environment has difficulty in hearing and responding to the nutritional needs of those living with CD and thus inhibit engagement in food-related activities.
Subsequently, results of the comparison between each of the CDDUX items yielded interesting findings. The single question referring directly to an environment outside of the home, the school, yielded the parents’ lowest average score and among the children’s lowest scores and represented an average bad feeling in both groups. This mutual bad feeling may possibly express the difficulty families have managing CD outside the home, as found in other literature concerning specific difficulties of people with CD (e.g., Lee, Ng, Diamond, Ciaccio, & Green, 2012; Olsson et al., 2009). Contrary to their parents, the children with CD described “not being able to eat just everything I want . . .” as the worst feeling of all. One way to look at the GFD is as avoidance of gluten consumption. However, looking at the situation as avoiding participation in food-related activities validates the subjective eating experience for the person with CD in his or her own everyday experiences.
Finally, as reflected in the CLASS results, characteristics of participation in food-related leisure activities of children with and without CD appear to be similar. However, the literature concerning the quality of life of this population has suggested close observation of research results to enable the identification of unique characteristics (Grootenhuis et al., 2007; Kurppa et al., 2011). Consequently, analysis of food-related leisure activities and children’s self-report HRQOL scores revealed a significant correlation between the social dimension of the CLASS food-related activities factor and self-report HRQOL scores. Children who felt better about their CD as reported on the CDDUX rated their social participation in CLASS food-related activities, namely, that with friends, higher. In fact, a lower rating on the sociability dimension reflected children’s performance closer to the home and family environment, and a higher rating reflected their performance in a broader environment, farther away from the home environment and with peers.
Social participation is considered an indicator of health and well-being (Piškur, 2014). Moreover, use of self-organization strategies to obtain occupational participation enables children to adapt their age-appropriate and developing skills to cope with environmental demands (Humphry, 2002). Thus, this correlation may possibly indicate that feeling better HRQOL enables the child to cope better and develop efficient strategies to independently manage the chronic condition while participating in food-related leisure activities together with friends out of the home environment.
Limitations and Future Directions
These findings should be interpreted with caution because of the small sample size and because the participants were a convenience sample. Consequently, participants do not necessarily represent the entire local celiac community and hence may bias the results. In addition, CD medical status and adherence to the GFD relied on parents’ reports. Moreover, the influence of factors such as parent education level and accessibility of local celiac information and services was not investigated but may have affected the nature of CD coping strategies.
The results of the current study may be used as a baseline for future studies. Additional environments, such as the school environment, in which children and adolescents spend substantial time in their daily lives should be examined. Further larger scale sample research including both parents’ and children’s perspectives is required to understand the needs of those living with CD in Israel and in other countries and to deepen the understanding of CD’s influence on their QOL and well-being. This understanding can lead to the development of performance-based assessment tools and supportive intervention programs for people with CD at all stages of life.
Implications for Occupational Therapy Practice
The results of this study have the following implications for occupational therapy practice:
Health care services do not sufficiently accommodate the dietary restrictions of people with CD and may limit their participation in various occupational situations.
Occupational therapy’s focus on routines and habits can facilitate the healthy development of children and adolescents with CD and assist them to maintain their health and well-being and self-manage their condition.
Further research can lead to development of occupation-based assessment tools to identify the factors that support or hinder food-related occupational performance in people’s everyday life with CD.
Conclusions
The results of this study indicate significant differences between children’s report of their QOL with CD and their parents’ proxy report. These differences in perception stress the importance of obtaining information from both sources to determine the HRQOL of children with CD. The findings of this study are supported by the basic assumptions of the ICF (WHO, 2001) and the Occupational Therapy Practice Framework (AOTA, 2014) concerning how a digestive system autoimmune disease such as CD may restrict participation in leisure activities and therefore influence QOL. Children’s and adolescents’ management of CD requires adapting strategies to changing environments, specifically in transitions (e.g., elementary to middle and high school) and across the lifespan. Occupational therapy can contribute to the adherence to healthy dietary habits by the population with CD while focusing on participation and promoting independence. It may also possibly bridge the gap and provide a link between the medical system and the sociocultural system (Baum, 2011; Persch et al., 2015).