Published online August 1, 2006
PEDIATRICS Vol. 118 No. 2 August 2006, pp. 594-602 (doi:10.1542/peds.2005-2123)
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ARTICLE

Screening for Celiac Disease in Asymptomatic Children With Down Syndrome: Cost-effectiveness of Preventing Lymphoma

Nancy L. Swigonski, MD, MPH, Heather L. Kuhlenschmidt, MD, Marilyn J. Bull, MD, Mark R. Corkins, MD and Stephen M. Downs, MD, MS

Department of Pediatrics, Indiana University School of Medicine, Indianapolis, Indiana

BACKGROUND. Studies demonstrate an increased prevalence of celiac disease in persons with Down syndrome, leading some organizations and authors to recommend universal screening of children with Down syndrome. However, many children with Down syndrome are asymptomatic, and the long-term implications of screening are unknown. The complication of celiac disease that leads to mortality in the general population is non-Hodgkin's lymphomas.

OBJECTIVES. The purpose of this research in asymptomatic children with Down syndrome was to (1) calculate the number needed to screen to prevent a single case of lymphoma and (2) present a cost-effectiveness study of screening.

METHODS. We constructed a decision tree using probabilities derived from the published literature for Down syndrome or from the general population where Down syndrome-specific data were not available. Celiac disease was determined by serologic screening and confirmation with intestinal biopsy. Sensitivity analysis was used to alter probability estimates affecting the cost of preventing lymphoma.

RESULTS. Using our baseline values, the no-screen strategy is dominant; that is, screening not only costs more but also results in fewer quality-adjusted life-years. A screening strategy costs more than $500000 per life-year gained. Screening all asymptomatic children with Down syndrome for celiac disease costs almost $5 million to prevent a single case of lymphoma.

CONCLUSION. These analyses do not support the cost-effectiveness of screening, and more data are needed before recommendations to screen asymptomatic children with Down syndrome for celiac disease can be made.

Key Words: Down syndrome • celiac disease • cost-effectiveness

Abbreviations: CD—celiac disease • DS—Down syndrome • SIR—standardized incidence ratio • SMR—standardized mortality ratio • Ig—immunoglobin • GFD—gluten-free diet • EMA—endomysial antibody • tTG—tissue transglutaminase antibody • CHD—congenital heart disease • CI—confidence interval • QALY—quality-adjusted life-year

Accepted Mar 7, 2006.




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