RESUMO
BACKGROUND & AIMS: Celiac disease (CD) is a common yet underdiagnosed autoimmune disease with substantial long-term consequences. High-accuracy point-of-care tests for CD antibodies conducted at youth primary health care centers may enable earlier identification of CD, but evidence about the cost-effectiveness of such strategies is lacking. We estimated the long-term cost-effectiveness of active case finding and mass screening compared with clinical detection in the Netherlands. METHODS: A decision tree and Markov model were used to simulate a cohort of 3-year-old children with CD according to each strategy, taking into account their impact on long-term costs (from a societal perspective) and quality-adjusted life-years (QALYs). Model parameters incorporated data from the GLUTENSCREEN project, the Dutch Celiac Society, the Dutch Pediatric Surveillance Unit, and published sources. The primary outcome was the incremental cost-effectiveness ratio (ICER) between strategies. RESULTS: Mass screening produced 7.46 more QALYs and was 28,635 more costly compared with current care (ICER: 3841 per QALY), and case finding produced 4.33 more QALYs and was 15,585 more costly compared with current care (ICER: 3603 per QALY). At a willingness to pay of 20,000 per QALY, both strategies were highly cost-effective compared with current care. Scenario analyses indicated that mass screening is likely the optimal strategy, unless no benefit in detecting asymptomatic cases is assumed. CONCLUSIONS: An earlier identification of CD through screening or case finding in children using a point-of-care tests leads to improved health outcomes and is cost-effective in the long-term compared with current care. If the feasibility and acceptability of the proposed strategies are successful, implementation in Dutch regular care is needed.
RESUMO
Introduction: Coeliac disease (CD) occurs in 1% of the population, develops early in life and is severely underdiagnosed. Undiagnosed and untreated disease is associated with short-term and long-term complications. The current healthcare approach is unable to solve the underdiagnosis of CD and timely diagnosis and treatment is only achieved by active case finding. Aim: to perform a case finding project to detect CD children who visit the Youth Health Care Centres (YHCCs) in a well-described region in the Netherlands to evaluate whether it is feasible, cost-effective and well accepted by the population. Methods/analysis: Prospective intervention cohort study. Parents of all children aged 12 months and 4 years attending the YHCCs for a regular visit are asked whether their child has one or more CD-related symptoms from a standardised list. If so, they will be invited to participate in the case finding study. After informed consent, a point of care test (POCT) to assess CD-specific antibodies against tissue transglutaminase (TG2A) is performed onsite the YHCCs. If the POCT is positive, CD is highly suspected and the child will be referred to hospital for definitive diagnosis according to the Guideline Coeliac Disease of the European Society for Pediatric Gastroenterology, Hepatology and Nutrition guideline. Main outcomes: Incidence rate of new CD diagnoses in the study region in comparison to the one in the same age diagnosed by standard of care in the rest of the Netherlands.Feasibility and cost-effectiveness of active CD case finding at the YHCCs. All costs of active case finding, diagnostics and treatment of CD and the potential short-term and long-term consequences of the disease will be calculated for the setting with and without case finding.Ethical acceptability: by questionnaires on parental and healthcare professionals' satisfaction.A statistical analysis plan was prepared and is published on the GLUTENSCREEN website (Statistical-Analysis-Plan-11-5-2021_def.pdf (glutenscreen.nl) and added as annex 1). Ethics and dissemination: The Medical Ethics Committee Leiden approved this study. If we prove that case finding at the YHCC is feasible, cost-effective and well accepted by the population, implementation is recommended. Trial registration number: NL63291.058.17.