Health-related quality-of-life in children with celiac disease, measured prior to receiving their diagnosis through screening

Department of Public Health and Clinical Medicine, Epidemiology and Global Health, Umeå University, 901 85 Umeå, Sweden.
Journal of Medical Screening (Impact Factor: 3.1). 11/2011; 18(4):187-92. DOI: 10.1258/jms.2011.011081
Source: PubMed


To compare the health-related quality-of-life (HRQoL) of children with screening-detected coeliac disease (CD), before they learned of their diagnosis, with that of children without CD and in those previously diagnosed with CD.
In a cross-sectional CD screening study ('ETICS': Exploring the Iceberg of Coeliacs in Sweden), of 10,041 Swedish 12-year-olds invited, 7567 (75%) consented to participate, and 7208 (72%) children without previously diagnosed CD had serological markers analysed. Before the screening results were known, 7218 children (72%) and 6524 of their parents (65%) answered questionnaires. Questionnaires included the Swedish child-friendly pilot version of the EQ-5D instrument and proxy version of the EQ-5D instrument, which are generic tools used to describe HRQoL.
We found no significant difference in HRQoL between the groups of children with screening-detected CD, without CD, and those previously diagnosed with CD.
The HRQoL reported by 12-year-olds with screening-detected CD, before they learned of their diagnosis, was not worse than that of the children without CD or those previously diagnosed with CD. Thus, mass screening for CD should not be justified on the basis that children with unrecognized CD have a poor HRQoL. However, because these children rated their HRQoL before diagnosis and treatment, they may not have recognized or perceived symptoms as severe enough to seek medical attention which demonstrates how difficult clinical/active case finding can be. Mass screening may still, therefore, be considered if the aim is early detection and prevention of future complications.

Download full-text


Available from: Katrina Nordyke, Jan 30, 2014
  • Source
    • "All children were invited to provide a blood sample and alongside to fill in a questionnaire. Data collection was performed in the schools by research nurses with assistance from school health nurses and teachers [16,29]. Afterwards, blood samples were analyzed for serological markers; anti-tissue-transglutaminase antibodies of IgA-type (tTG) for all children and for some also anti-endomysial antibodies IgA (EMA), or in case of IgA-deficiency serological markers of IgG type. "
    [Show abstract] [Hide abstract]
    ABSTRACT: Background Knowledge regarding the health-related quality of life (HRQoL) of children with celiac disease remains limited and inconclusive. We investigated the HRQoL of three groups of 12-year-olds with: i) undetected celiac disease ii) clinically diagnosed celiac disease, and iii) without celiac disease. Methods A school-based cross-sectional multicenter screening study invited 18 325 children, whereof 68% consented to participate. Participants provided a blood sample, which was later analyzed for anti-tissue-tranglutaminase antibodies, and alongside filled in a questionnaire. When anti-tissue-tranglutaminase antibodies were elevated, a small intestinal biopsy verified the screening-detected celiac disease diagnosis. Self-reported HRQoL was measured using Kidscreen, a generic 52 items instrument with proven reliability and validity. Scores were linearly transformed into a 0–100 scale with higher values indicating better HRQoL. Mean values with standard deviations (mean ± SD) were compared, and uni- and multivariate logistic regression models tested the odds of a low HRQoL among children with undetected or diagnosed celiac disease, respectively. Results Children with undetected celiac disease (n = 238) reported similar HRQoL as children without celiac disease (n = 12 037) (83.0 ± 11.0 vs. 82.5 ± 11.3, P = 0.51), and also similar HRQoL (82.2 ± 12.2, P = 0.28) to that of children with diagnosed celiac disease (n = 90), of whom 92% were adherent to treatment. Having undetected celiac disease did not increase the odds of low overall HRQoL, independent of sex, area of residence, study year and occurrence of gastrointestinal symptoms (adjusted odds ratio 0.77, 95% CI 0.54-1.10). Comparable results were seen for diagnosed celiac disease cases (adjusted odds ratio 1.11, 95% CI 0.67-1.85). Conclusion Children with undetected celiac disease reported comparable HRQoL as their peers with diagnosed celiac disease, and those without celiac disease, when reporting prior to receiving the diagnosis through screening. Thus, children with celiac disease, both untreated and diagnosed, perceive their HRQoL as unimpaired by their disease.
    BMC Public Health 05/2014; 14(1):425. DOI:10.1186/1471-2458-14-425 · 2.26 Impact Factor
  • Source
    • "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 [34]. However, in this current study we explore the adolescents’ HRQoL before and after diagnosis and treatment. "
    [Show abstract] [Hide abstract]
    ABSTRACT: Celiac disease (CD) is a chronic disorder in genetically predisposed individuals in which a small intestinal immune-mediated enteropathy is precipitated by dietary gluten. It can be difficult to diagnose because signs and symptoms may be absent, subtle, or not recognized as CD related and therefore not prompt testing within routine clinical practice. Thus, most people with CD are undiagnosed and a public health intervention, which involves screening the general population, is an option to find those with unrecognized CD. However, how these screening-detected individuals experience the diagnosis and treatment (gluten-free diet) is not fully understood. The aim of this study is to investigate the health-related quality of life (HRQoL) of adolescents with screening-detected CD before and one year after diagnosis and treatment. A prospective nested case-referent study was done involving Swedish adolescents who had participated in a CD screening study when they were in the sixth grade and about 12 years old. Screening-detected adolescents (n = 103) and referents without CD who participated in the same screening (n = 483) answered questionnaires at the time of the screening and approximately one year after the screening-detected adolescents had received their diagnosis that included the EQ-5D instrument used to measure health status and report HRQoL. The HRQoL for the adolescents with screening-detected CD is similar to the referents, both before and one year after diagnosis and initiation of the gluten-free diet, except in the dimension of pain at follow-up. In the pain dimension at follow-up, fewer cases reported problems than referents (12.6% and 21.9% respectively, Adjusted OR 0.50, 95% CI 0.27-0.94). However, a sex stratified analysis revealed that the significant difference was for boys at follow-up, where fewer screening-detected boys reported problems (4.3%) compared to referent boys (18.8%) (Adjusted OR 0.17, 95% CI 0.04-0.73). The findings of this study suggest that adolescents with unrecognized CD experience similar HRQoL as their peers without CD, both before and one year after diagnosis and initiation of gluten-free diet, except for boys in the dimension of pain at follow-up.
    BMC Public Health 02/2013; 13(1):142. DOI:10.1186/1471-2458-13-142 · 2.26 Impact Factor

  • Journal of pediatric gastroenterology and nutrition 05/2012; 55(4):365. DOI:10.1097/MPG.0b013e31825f1617 · 2.63 Impact Factor
Show more