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Health-related quality-of-life in children with coeliac disease, measured prior to receiving their diagnosis through screening
Umeå University, Faculty of Medicine, Department of Public Health and Clinical Medicine, Epidemiology and Global Health.
Umeå University, Faculty of Medicine, Department of Public Health and Clinical Medicine, Epidemiology and Global Health.ORCID iD: 0000-2021-0028-7401
Umeå University, Faculty of Medicine, Department of Public Health and Clinical Medicine, Epidemiology and Global Health.ORCID iD: 0000-0002-1633-2179
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2011 (English)In: Journal of Medical Screening, ISSN 0969-1413, E-ISSN 1475-5793, Vol. 18, no 4, 187-192 p.Article in journal (Refereed) Published
Abstract [en]

OBJECTIVE: 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.

METHODS: 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.

RESULTS: We found no significant difference in HRQoL between the groups of children with screening-detected CD, without CD, and those previously diagnosed with CD.

CONCLUSION: 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.

Place, publisher, year, edition, pages
Royal Society of Medicine Press, 2011. Vol. 18, no 4, 187-192 p.
National Category
Public Health, Global Health, Social Medicine and Epidemiology
Identifiers
URN: urn:nbn:se:umu:diva-54066DOI: 10.1258/jms.2011.011081PubMedID: 22106434OAI: oai:DiVA.org:umu-54066DiVA: diva2:515489
Available from: 2012-04-13 Created: 2012-04-13 Last updated: 2017-12-07Bibliographically approved
In thesis
1. The burden of celiac disease and the value of having it diagnosed
Open this publication in new window or tab >>The burden of celiac disease and the value of having it diagnosed
2012 (English)Doctoral thesis, comprehensive summary (Other academic)
Alternative title[sv]
Bördan av att leva med celiaki och värdet av att bli diagnostiserad
Abstract [en]

Background: Celiac disease is a chronic disease characterized by intolerance to gluten. It is considered a public health problem affecting about 1% of Western populations, but, with most cases still undiagnosed. A glutenfree diet is the only effective treatment for the disease.

Objectives: To investigate the burden of celiac disease and the value of having it diagnosed. Additionally, the implications for a potential future celiac disease mass screening are discussed.

Methods: A questionnaire was sent during 2009 to 1,560 randomly selected adult members of the Swedish Society for Coeliacs, in equal-sized age- and sex strata, and 1,031 (66%) responded. Members were asked about symptoms, health-related quality of life as measured by EQ-5D, and health care consumption during the year prior to diagnosis and during the past year. They were also asked about the delay in having their celiac disease diagnosed and the appearance of other immune-mediated diseases. A school-based celiac disease screening of 12-year-olds was performed during 2005-2006. After blood sampling the 7,567 participating children and their parents received a questionnaire including the EQ-5D instrument in order to measure the child’s health-related quality of life. Comparisons were made between children with screening-detected celiac disease, those with previously diagnosed disease and those without the disease. Parents were asked about their willingness to pay for a celiac disease screening of their child, which was compared with the actual cost of a screening.

Results: Adult celiac disease patients had a poorer health-related quality of life than the general population, and a high prevalence of symptoms before celiac disease diagnosis. The mean delay from symptoms to diagnosis was 9.7 years. After initiated treatment with a gluten-free diet, health-related quality of life was improved to the level of the general population, and symptom relief and reduction in health care consumption were also reported. For children, health-related quality of life was similar across groups. The average cost per child for a screening was 47 EUR. Parents’ mean willingness to pay for a screening was 79 EUR, median 10 EUR.

Conclusion: The delay in celiac disease diagnosis is long, and treatment with a gluten-free diet substantially improved health for clinically detected cases. For screening-detected celiac disease the health benefits are still uncertain. A mass screening might nevertheless be justified to avoid the burden of living with undiagnosed disease, and parents’ willingness to pay indicates that performing it in childhood might be economically motivated. However, as both the cost-effectiveness of a screening and the short- and long term health benefits for screening-detected cases have not yet been sufficiently investigated, it is too early to make a recommendation for a celiac disease mass screening.

Place, publisher, year, edition, pages
Umeå: Umeå Universitet, 2012. 68 p.
Series
Umeå University medical dissertations, ISSN 0346-6612 ; 1489
National Category
Public Health, Global Health, Social Medicine and Epidemiology
Research subject
Public health; Epidemiology
Identifiers
urn:nbn:se:umu:diva-54058 (URN)978-91-7459-392-1 (ISBN)
Public defence
2012-05-11, Aulan, vårdvetarhuset, Umeå, 09:00 (Swedish)
Opponent
Supervisors
Available from: 2012-04-20 Created: 2012-04-12 Last updated: 2015-04-29Bibliographically approved
2. Mass screening for celiac disease: a public health intervention from the participant perspective
Open this publication in new window or tab >>Mass screening for celiac disease: a public health intervention from the participant perspective
2013 (English)Doctoral thesis, comprehensive summary (Other academic)
Abstract [en]

Background  Celiac disease (CD) is a chronic disorder in genetically predisposed individuals in which damage to the small intestine is caused by eating foods containing gluten. The prevalence has been shown to vary from around 1-3%, but most people with CD are undiagnosed. An option for finding those with unrecognized CD would include screening the general population, i.e., mass screening. However, screening identifies a pre-disease or disease condition in people who are presumed healthy and have not sought help. Therefore, the impacts of the screening process and being diagnosed through screening should be explored before such a public health intervention is considered. A population-based CD screening study involving 12-year-olds was undertaken in Sweden and provided an opportunity to explore these issues related to CD screening.

Aims To make inferences about the potential impacts mass screening for CD can have on participants by exploring experiences and outcomes for participants involved in CD screening study.

Methods and Subjects  Both qualitative (short written narratives) and quantitative (questionnaires with EQ-5D instrument) methods were used. Children who participated in the CD screening study were invited to write narratives at the time of the screening, before screening results were known, describing their experience with the screening (n=240). The EQ-5D instrument was used to measure and compare health-related quality of life reported by participants at the time of the screening and one year after the screening-detected participants received their diagnosis (screening-detected n=103, referents n=483). Those with screening-detected CD were also invited to write narratives one and five years after their diagnosis. In these narratives the adolescents described how it felt to be diagnosed with CD, how it felt to live with CD, and if they thought all children should be screened (one-year follow-up n=91, five-year follow-up n=72).

Results  Even though some children experienced fear and anxiety during the screening, overall they had, or were provided with, tools that allowed them to cope well with the screening. The health-related quality of life reported by those with screening-detected CD was similar before and one year after diagnosis (and similar to that of the referents). We also found that after five years of living with the diagnosis there had been maintenance and evolution in the beliefs and practices of these adolescents. Being detected through screening and the threat of complications impacted how they felt about the diagnosis, coped with the gluten-free diet, and what they thought about CD screening. Five years after the screening-detected diagnosis the adolescents have adjusted to the disease and adapted new habits and coping strategies to deal with the gluten-free diet. However, there are still those who doubt the accuracy and benefit of the diagnosis.

 

Conclusions  Our findings suggest that it is possible for participants to avoid excess anxiety during CD screening. However, there was not consensus among participants that being detected and treated had improved their health-related quality of life or that the immediate benefits outweighed the harm caused by being detected in this way. When considering mass screening, the affect on the participants is important to take into account and our findings shed light on some of the potential impacts a CD mass screening could have on participants.

Place, publisher, year, edition, pages
Umeå: Umeå University, 2013. 89 p.
Series
Umeå University medical dissertations, ISSN 0346-6612 ; 1597
Keyword
adolescents, celiac disease, children, EQ-5D, intervention, public health, narrative, screening, screening-detected
National Category
Public Health, Global Health, Social Medicine and Epidemiology
Research subject
Public health
Identifiers
urn:nbn:se:umu:diva-81689 (URN)978-91-7459-726-4 (ISBN)
Public defence
2013-11-15, Aulan, Care of Science Building (Vårdvetarhuset), Norrlands University Hospital (NUS), Umeå, 09:00 (English)
Opponent
Supervisors
Available from: 2013-10-24 Created: 2013-10-21 Last updated: 2015-04-29Bibliographically approved

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