CeD and IBD are both chronic gastrointestinal conditions differing in their clinical presentation and limitations in daily life with a GFD as the only treatment option for children with CeD [22], while children with IBD often have to take long-term medication with potential side effects or undergo surgery [23] . Therefore, it could be speculated that children with IBD may have lower QoL scores than children with CeD. To date, no studies exist comparing QoL of paediatric IBD patients to CeD patients and to healthy controls.
Interestingly, the median PedsQL™ scores of children with CeD and IBD did not differ in our cohort. Also, no differences in the total scores by sex or age were found. This is consistent with previous studies which have not shown any significant impact by sex or age on total QoL scores of neither children with CeD [24] or children with IBD [25, 26] nor healthy children [27].
However, regarding subscales of the PedsQL™ 4.0 SF, our results differ from previous studies, which showed lower Emotional Functioning in female children with CeD [3] and in female patients with IBD [26], which we could not confirm in our cohort. Nevertheless, we found differences in children with CeD and their parents reporting lower Emotional Functioning and more Food and Drink Limits than children with IBD while Blood in Poop was lower in children with IBD and their parents compared to children with CeD. This is not surprising as rectal bleeding is one of the main symptoms of IBD [28] and Food and Drink Limits in children with CeD are evident as the GFD is restrictive. Similar to our findings, previous studies have described impaired Emotional Functioning in children with CeD, with a high prevalence of anxiety and depressive symptoms [30]. On the one hand, Emotional Functioning may be impaired due to intrinsic depressive symptoms and anxiety that can occur from untreated CeD [31]. As we have not assessed the degree of adherence to GFD, nor have we found a correlation between QoL and elevated tTG-IgA levels, no statement can be made about a possible relationship. On the other hand, Emotional Functioning may be impaired due the constantly encountered food restrictions and the fear of accidentally eating gluten-containing food. Unexpectedly, children with CeD reported lower QoL in terms of Trouble Swallowing and Heart Burn and Reflux. Interestingly, a study with adult CeD patients showed that newly diagnosed CeD patients had significantly increased reflux symptoms compared to healthy controls, which rapidly improved under GFD [32]. As adherence to the GFD was not assessed in our study, no correlation can be made here. However, no correlation between reflux symptoms and tTG-IgA levels was shown.
In the assessment of children with IBD and their parents, elevated calprotectin levels correlated with significantly lower overall QoL, lower Physical Functioning and lower Social Functioning. Calprotectin indicates inadequate disease control, which may be associated with various debilitating symptoms. For example, diarrhoea and the fear of using public toilets have already been associated with significantly poorer Physical and Social Functioning [29] resulting in impaired QoL. Consequently, in contrast, the therapeutic goal of achieving clinical and biochemical remission leads to better HRQoL. Elevated calprotectin levels can be considered a warning sign for being at risk for a reduced HRQoL.
In our study, both children and parents were interviewed. In contrast to other studies [9], the agreement between children and parents on overall QoL ratings did not differ significantly, although parents’ ratings were significantly lower in areas such as Emotional Functioning and Social Functioning. Haller et al. suggested that this may be due to parents having limited information about their child’s internal feelings and relations with peers [34]. Therefore, it is necessary to assess both the parents’ view on their child’s QoL and the child’s QoL to minimise parents’ concerns about and to accurately assess parents’ views on their child’s QoL and to avoid misjudgement and overtreatment.
To estimate the impairment of QoL assessed in our study cohort, we compared our children’s outcomes with those in studies on patients of similar age (5-18 years) and their parents (2-18 years) who had either gastrointestinal disorders (CD, UC, gastroesophageal reflux) or functional gastrointestinal disorders (FGID) (chronic constipation, functional abdominal pain, irritable bowel syndrome, functional dyspepsia) or who were healthy controls (Table 3).
TABLE 3. Comparison of the mean scores of the PedsQl™ 4.0 to the literature.
|
|
CeDa
Mean (SD)
|
IBDb
Mean (SD)
|
FGIDc
Mean (SD)
|
GId
Mean (SD)
|
HCe
Mean (SD)
|
Child: PedsQL™ 4.0
|
n=56
|
n=45
|
n=281
|
n=298
|
n=936
|
Total score
|
83.5 (11.5)
|
85.4 (15.8)
|
70.2 (17.0)
|
78.0 (14.6)
|
85.6 (11.9)
|
Physical Health
|
91.9 (11.4)
|
89.8 (17.6)
|
73.1 (20.3)
|
80.5 (16.6)
|
89.6 (12.1)
|
Psychosocial Health
|
80.7 (13.5)
|
83.9 (16.3)
|
68.6 (17.8)
|
76.6 (15.7)
|
83.5 (13.6)
|
Emotional Functioning
|
73.6 (17.2)
|
80.2 (21.0)
|
66.4 (23.6)
|
74.7 (21.2)
|
81.3 (17.9)
|
Social Functioning
|
88.1 (18.2)
|
90.7 (16.4)
|
79.2 (19.5)
|
87.3 (16.1)
|
87.4 (15.9)
|
School Functioning
|
80.4 (17.5)
|
80.8 (19.7)
|
60.3 (22.4)
|
67.8 (20.5)
|
81.8 (16.1)
|
|
|
|
|
|
|
Parents: PedsQL™ 4.0
|
n=67
|
n=50
|
n=356
|
n=301
|
n=1106
|
Total score
|
82.5 (12.4)
|
82.4 (19.1)
|
70.5 (19.1)
|
75.6 (17.5)
|
85.2 (12.8)
|
Physical Health
|
94.3 10.5
|
87.2 (21.4)
|
72.1 (22.8)
|
77.9 (21.1)
|
88.7 (15.2)
|
Psychosocial Health
|
78.5 (14.9)
|
80.8 (19.3)
|
69.7 (19.9)
|
74.4 (17.6)
|
83.3 (13.6)
|
Emotional Functioning
|
68.8 (19.4)
|
77.0 (21.3)
|
65.3 (24.3)
|
71.3 (21.7)
|
81.9 (16.4)
|
Social Functioning
|
86.7 (18.2)
|
87.2 (21.2)
|
79.4 (21.0)
|
82.8 (19.2)
|
87.2 (16.4)
|
School Functioning
|
80.0 (20.4)
|
77.9 (24.2)
|
63.0 (24.8)
|
68.9 (22.4)
|
80.6 (17.0)
|
a coeliac disease; b inflammatory bowel disease; c functional gastrointestinal disorder; d gastrointestinal disorders; e healthy controls. Confidence interval 95%.
Compared to children in the United States with gastrointestinal disorders (n=298) and FGID (n=281) [35] as well as healthy control subjects (n=936) [18,27] the mean QoL scores of our cohort interestingly showed no significant differences to healthy controls (CeD: 83.5±11.5, IBD: 85.4±15.8, healthy controls: 85.6±11.9. However, Emotional Functioning was reduced in children with CeD compared to healthy controls (p<0.01, CI 95%). Remarkably, higher mean QoL scores (p<0.01, CI 95%) were found in our cohort compared to children with gastrointestinal disorders and FGID from the literature. Mean QoL scores of children with gastrointestinal disorders (78.0±14.6) and FGID (70.2±17.0) were significantly lower than those of our cohort (CeD: 83.5±11.5; IBD: 85.4±15.8). The comparison of the parents' assessments showed similar results.
Conflicting statements in the literature exist regarding QoL of children with CeD and IBD compared to other children. While there are studies showing that QoL of children with CeD is comparable to the QoL of healthy controls and even better than the QoL of children with other gastrointestinal diseases such as functional constipation [36], other studies showed contrary results with significantly lower QoL of children with CeD compared to healthy controls and other gastrointestinal disorders [37]. Likewise, children with IBD in remission were found to have a similar QoL comparable to healthy controls whereas children with active IBD were found to have a significantly lower QoL compared to healthy children [38].
It must also be assumed that the patients’ history and intensity of disease vary between different studies and that there may also be a certain selection of participating patients in the surveys. Our children show a good QoL comparable to healthy children. A reason for this could be the very regular monitoring and consistent treatment and, therefore, patients with active disease and potentially low QoL consequently had access to more intensive medical and psychosocial care. However, the outcome could also be biased by the group of families who decided to participate in the study. This aspect should certainly be seen as a weakness of our study, as voluntary participation did not allow us to represent the entire group of patients. In addition, due to the limited sample size, patients in both disease groups were not matched for sex and age, and all patients were recruited from a single centre in Germany. As this is a cross-sectional study, the results may be influenced by the current situation, and findings may not be generalisable. Furthermore, adherence to the GFD, which may have an impact on Emotional Functioning as well as Heart Burn and Reflux, was not assessed. Calprotectin and tTG-IgA values were not always checked close to the time of the interview.
However, this study has several strengths including the use of a validated generic and disease-specific gastrointestinal questionnaire to assess QoL. It is the first study to provide a comparative insight into the QoL of paediatric CeD and IBD patients. The results of this study are important for paediatricians who counsel patients with newly diagnosed CeD or IBD. Children with CeD and IBD can achieve a similar QoL as healthy peers, in contrast to other chronic paediatric conditions like e.g. type I diabetes mellitus [39] or children with moderate-to-severe plaque psoriasis [40].