This is, to our knowledge, the first qualitative study exploring daily life consequences of receiving a screening-detected CD diagnosis. We found that the diagnosis had varying impact on quality of life that related both to changes in perceived health and to the adolescents' experiences of living with CD in terms of social sacrifices.
Previous research on CD and gluten-free diet's effect on quality of life has mainly utilized quantitative methods [21–26]. Although these quantified measures may facilitate reproducibility, they do not allow for capturing the complexity of the patients' lived experiences. By using qualitative methods, our study accessed adolescents' and parents' own perspective which allowed for a holistic description of changes in perceived health as well as impact on daily life. The study was characterized by an emergent design, purposive sampling of informants, reflective field notes, and oscillation between data collection and analysis. To further increase the credibility of the study, continued peer debriefing sessions were held within the research group and an audit trail with analytical memo notes was maintained throughout the study. The integrity of the study was strengthened by the moderators of the group discussions not being involved in the health care provided to the adolescents.
Focus group discussions build on group interaction and can facilitate sharing experiences, especially when eliciting children's views . However, in our study the willingness to participate in the focus group discussions may have been influenced by more positive experiences of the screening. Also, few descriptions of symptoms were communicated by the adolescents, maybe because of being reticent to share descriptions of symptoms among peers. On the other hand, the parents shared rich descriptions of their child's signs, symptoms, and well-being both before and after diagnosis and treatment. In addition, the collection of individually written narratives enabled us to capture more personal and sensitive experiences and also to explore the variation in experiences of the adolescents and their parents.
We found a large variation in perceived health before diagnosis among screening-detected CD adolescents, which has also been described by others [2, 3, 18, 20]. Together these results confirm that not all screening-detected CD cases perceive themselves as healthy. Our findings that some had experienced health problems, and sought health care, without receiving a correct diagnosis indicate that further educational efforts to increase CD awareness are needed. The observed phenomena of retrospective recognition of symptoms in relation to a screening-detected CD diagnosis is in line with other studies [18, 20], and seems to reflect both an increased understanding of symptoms and a reassurance of the benefits of having received the diagnosis.
In this study, we observed a varying impact on quality of life in terms of social sacrifices for the adolescents. Whereas some had, or were provided with, tools to successfully manage daily life, others found CD to be truly burdensome with considerable negative impact on their lives. A prominent experience was that adhering to the dietary restrictions limited daily life and caused feelings of being a burden or an outsider. Thus, adhering to the gluten-free diet related to felt stigma, as defined by Scambler and Hopkins . In line with Goffman's work on stigma management , we found that the adolescents had adopted strategies such as withdrawing from social contacts, attempting to hide their condition, or compensating by being overly nice. These findings build on to the findings of another study on clinically diagnosed adolescents reporting on stigma experiences related to gluten-free diet , by indicating that mode of diagnosis probably does not affect stigma experiences.
Our results also indicated that stigma experiences may be linked to gender differences in management strategies. In general, boys described more efforts to conceal their disease and reluctance to incorporate the disease into their social identities than girls. However, those who had chosen to abandon the gluten-free diet were girls. We saw a tendency that boys asked for support in their efforts to change external structures, whereas girls took on an active role themselves. These results are in line with studies about adolescents with asthma and diabetes showing that gendered meanings of stigma influence the strategies used to cope with the disease and treatment .
Mass screening for CD is still questioned, although most of the World Health Organization's criteria for implementation of mass screening programs are fulfilled [11–18]. A common argument against CD mass screening is that the diagnosis and treatment would be harder to accept and manage among those experiencing no prior symptoms compared to patients with clinically-detected CD. This assumes that screening-detected CD cases do not experience symptoms, while we and others have shown that screening also captures unrecognized symptomatic cases [19, 20]. Furthermore, it assumes that experiencing health improvement facilitates the acceptance of the diagnosis. However, we found that the adolescents' feelings and attitudes about living with CD did not have a direct relation to whether or not experiencing health improvement. Those with great health benefits could be the ones suffering most in terms of social consequences, and vice versa, indicating that there are many aspects, apart from perceived health benefits, influencing the adolescents' experiences. Thus, CD screening as a public health intervention needs to be evaluated by balancing intended positive outcome in terms of health benefits against unintended negative consequences in terms of social sacrifices [38, 39]. Further qualitative studies on psychological and social reactions as well as attitudes and feeling towards a CD screening are needed to fully understand the implications for designing and evaluating full scale screening programs among children or adolescents. Preferably such studies should also involve other age groups and cultural settings.