The HRQoL for adolescents with screening-detected CD, as self-reported on the EQ-5D, is similar to that of their peers, both before and one year after diagnosis and GFD, except in the dimension of pain at follow-up. A sex stratified analysis revealed that this is due to the difference between boy cases and referents at follow-up, in which 4.3% of the screening-detected boys report problems and 18.8% of the referent boys report problems (Adjusted OR 0.17, 95% CI 0.04-0.73) (Table
We present a unique study in which adolescents with screening-detected CD report their HRQoL before and one year after diagnosis and treatment. A strength of our study is that the adolescents report baseline HRQoL before knowledge of their CD diagnosis, unlike many studies in which patients are asked to recall how they felt at the time of their diagnosis. We have previously published baseline data, including participants from the same screening study, in which there was also no significant difference in HRQoL for those with unrecognized CD compared to their peers without CD at the time of the screening
. However, in this current study we explore the adolescents’ HRQoL before and after diagnosis and treatment.
Another strength of this study is the fact that the adolescents had their CD detected as a result of a screening study, and not clinically or because they were considered high risk. This means that our results may be more reflective of those living with unrecognized CD, which has been seen as a limitation in other studies that attempt to address the QoL for those with undiagnosed CD
. In a review of literature that addresses how screening, diagnosing, and treating “asymptomatic screening-detected individuals” affects their QoL, done by Aggarwal et al.
, most of the studies involved CD patients who were identified from high risk groups
[19, 26] or were compared based on what type of symptoms led to their diagnosis; i.e., typical, not typical, or those who reported they had not experienced any symptoms
[23, 24, 28]. However, two of the studies included individuals that could be considered as screened from the general population
[3, 35, 36]. In those studies, the individuals who had “typical” symptoms showed an improvement in QoL scores after one year on the GFD, while those who were supposedly symptom free had scores comparable to healthy controls at baseline and at follow-up,
[35, 36] similar to the screening-detected adolescents in our study.
Even though the EQ-5D instrument is a validated tool and we have used the Swedish child-friendly pilot version, this tool may have limitations in the context of this study. Perhaps, it is not the ideal tool for capturing problems with subtle symptoms or feelings caused by unrecognized CD. Also, these adolescents may have adapted to their current health situation as normal and at baseline rated their health status as high as possible and similar to their peers. If they did experience improvement after diagnosis and treatment, they would be unable to demonstrate improvement from the high health status previously reported. These screening-detected adolescents were also invited to participate in another follow-up study, where they participated in focus group discussions (n = 31) and wrote narratives (n = 91), and when they were asked specifically about change in well-being after diagnosis, 53.8% reported feeling better
. In that study, it was also shown that some of the screening-detected children only realized they had been experiencing symptoms after they had been diagnosed and treated
Although it is beyond the scope of this study to provide an explanation, the finding that fewer screening-detected boys (4.3%) reported problems at follow-up in the dimension of pain than the boy referents (18.8%) is interesting to consider and warrants further investigation. In a previous Swedish study, a doubled risk for symptomatic CD in girls compared to boys was found
. In this screening study, the male to female ratio for those with clinically diagnosed CD was 1:2 compared to 1:1 for those with screening-detected CD
. This difference reveals that, at the time of the screening, a larger proportion of girls compared to boys had already been diagnosed with CD in routine clinical practice
. It could be that the boys with unrecognized CD were further progressed in their disease at baseline because they were not as likely as the girls to have been found in clinical practice. One could speculate that, even though they may not have realized the extent of their problems at baseline, they experienced benefit from the treatment resulting in fewer of these boys reporting problems with pain than the boys in the referent group, a phenomenon which was not captured for the girls. In studies involving adults, men and women have been shown to experience the burden of CD differently
[27, 39, 40]. There is also research that shows men and women access health care differently
[41–43] and perhaps the boys were less likely to seek or receive care. It is also a possibility that clinicians expect girls to have a higher risk of developing CD and more readily recognize and diagnose girls.
In our study there were no significant differences in the anxiety dimension between cases and referents, suggesting that for these screening-detected adolescents the CD diagnosis and GFD have not caused excess anxiety (at least at one year follow-up). Other studies have shown that those with a strict GFD can have the same HRQoL as healthy children/adolescents
[44, 45]. However, in the qualitative study mentioned previously
, it was revealed that some of these adolescents perceived the GFD and lifestyle changes as inconvenient and causing feelings of stigma while others adapted well to the disease.