PEDIATRICS Vol. 118 No. 2 August 2006, pp. 816-817 (doi:10.1542/10.1542/peds.2006-1194)
COMMENTARY |
Screening for Celiac Disease in Asymptomatic Children With Down Syndrome: Cost-effectiveness of Preventing Lymphoma
Division of Pediatric Gastroenterology, Nutrition and Liver Diseases, Hasbro Children’s Hospital, Brown Medical School, Providence, Rhode Island
Celiac disease is now recognized as a relatively common disease. The North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition subcommittee on celiac disease recently published recommendations for testing and treatment.1 The subcommittee recommended that high-risk groups such as first-degree relatives of patients with celiac disease and individuals with type 1 diabetes mellitus, Turner’s syndrome, short stature, permanent dentition enamel erosion, and Down syndrome be screened with the antitissue transglutaminase antibody test.
Screening for a disease is appropriate if the disease has serious consequences, if before onset of the disease there is a detectable latent phase, and there exists a treatment that is more effective at an earlier stage.2 In patients with symptomatic celiac disease, a gluten-free diet for as little as 5 years can reduce or prevent gut lymphoma. Thus, with respect to gut lymphoma, celiac disease can be considered a premalignant condition.3–5 Furthermore, in a substantial proportion of cancer patients, the underlying celiac disease is not recognized until around the time of cancer diagnosis. Therefore, it would seem intuitive that identification and treatment of celiac disease at an earlier, asymptomatic stage would be even more effective.
Swigonski et al6 show in their decision analysis that treatment of asymptomatic celiac disease at an earlier stage in children with Down syndrome would have no effect on this serious outcome. They further show that screening asymptomatic children with Down syndrome would do more harm than good. The children’s quality of life would be negatively affected, and society’s resources would be diverted away from other priorities. Non-Hodgkin’s lymphoma is seen largely in individuals in their 60s and older. Given that the average person with Down syndrome does not yet live past his/her 50s, the conclusion that lymphoma would not be significantly prevented is evident.7
Non-Hodgkin’s lymphoma is not the only consequence of celiac disease. Therefore, the larger issue is whether treating celiac disease at an asymptomatic stage is more effective for other outcomes and in other groups also at risk. The authors site growth failure as one of the common findings at diagnosis of celiac disease in children with Down syndrome. Impairment of linear growth becomes evident late in the course of a disease. One would expect that screening and, therefore, earlier treatment would prevent growth failure. Unfortunately, no studies have addressed this and other outcomes among asymptomatic first-degree relatives. The findings are, at best, controversial in children with type 1 diabetes.8,9
Absence of evidence of effect is not synonymous with lack of effect. Swigonski et al and others remind us that despite good intentions harm could be done and resources may be wasted. Every effort should be made to verify the actual efficacy of screening asymptomatic children in the various risk groups. High-quality randomized clinical trials of screening versus no-screening strategies would provide the best evidence.
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FOOTNOTES |
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Accepted Apr 25, 2006.
Address correspondence to Neal S. LeLeiko, MD, PhD, Pediatric Gastroenterology, Brown University, 593 Eddy St, MPH 126, Providence, RI 02903. E-mail: neal_leleiko{at}brown.edu
The authors have indicated they have no financial relationships relevant to this article to disclose.
Opinions expressed in this commentary are those of the author and not necessarily those of the American Academy of Pediatrics or its Committees.
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- Aschengrau A, Seage GR. Essentials of Epidemiology and Public Health. Sudbury, MA: Jones and Bartlett Publishers, Inc; 2003
- Green PHR, Jabri B. Celiac disease and other precursors to small-bowel malignancy. Hematol Oncol Clin North Am. 2003;17 :611 –624[CrossRef]
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- Swigonski NL, Kuhlenschmidt HL, Bull MJ, Corkins MR, Downs SM. Screening for celiac disease in asymptomatic children with Down syndrome: cost-effectiveness of preventing lymphoma.
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[Abstract/Free Full Text] - Bittles AH, Glasson EJ. Clinical, social, and ethical implications of changing life expectancy in Down syndrome. Dev Med Child Neurol. 2004;46 :282 –286[Web of Science][Medline]
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[Free Full Text] - Rami B, Sumnik Z, Schober E, et al. Screening detected celiac disease in children with type 1 diabetes mellitus: effect on the clinical course (a case control study). J Pediatr Gastroenterol Nutr. 2005;41 :317 –321[CrossRef][Web of Science][Medline]
PEDIATRICS (ISSN 1098-4275). ©2006 by the American Academy of Pediatrics
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