title: Screening for celiac disease in asymptomatic children with Down syndrome: cost-effectiveness of preventing lymphoma.
abstract: 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.
  
  
  OBJECTIVE
  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 $500,000 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.
  
  
  CONCLUSIONS
  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.
authors.affiliation: Department of Pediatrics, Indiana University School of Medicine, Indianapolis, Indiana, USA. nswigons@iupui.edu
authors.forename: Nancy L
authors.initials: NL
authors.lastname: Swigonski
authors.forename: Heather L
authors.initials: HL
authors.lastname: Kuhlenschmidt
authors.forename: Marilyn J
authors.initials: MJ
authors.lastname: Bull
authors.forename: Mark R
authors.initials: MR
authors.lastname: Corkins
authors.forename: Stephen M
authors.initials: SM
authors.lastname: Downs
mesh_terms: D002446:Celiac Disease / Q000150:complications / Q000175:diagnosis* / Q000191:economics / Q000453:epidemiology; D002648:Child; D015897:Comorbidity; D003362:Cost-Benefit Analysis; D003365:Costs and Cost Analysis; D003663:Decision Trees; D004198:Disease Susceptibility; D004314:Down Syndrome / Q000150:complications*; D005770:Gastrointestinal Neoplasms / Q000191:economics / Q000453:epidemiology / Q000209:etiology / Q000517:prevention & control; D006801:Humans; D007349:Insurance, Health, Reimbursement / Q000191:economics / Q000706:statistics & numerical data; D008228:Lymphoma, Non-Hodgkin / Q000191:economics / Q000453:epidemiology / Q000209:etiology / Q000517:prevention & control*; D008403:Mass Screening / Q000191:economics; D008484:Medicaid / Q000191:economics / Q000706:statistics & numerical data; D006278:Medicare / Q000191:economics / Q000706:statistics & numerical data; D008962:Models, Theoretical; D015995:Prevalence; D019057:Quality-Adjusted Life Years; D012306:Risk; D012680:Sensitivity and Specificity