Original ArticlesAccuracy and cost-effectiveness of a new strategy to screen for celiac disease in children with Down syndrome☆,☆☆
Section snippets
SUBJECTS AND METHODS
Inclusion criteria for the children participating in the study were: having DS, being a member of the Dutch Down Syndrome Foundation, having been born in the province of “Zuid Holland,” and having had gluten introduced into the diet. Informed consent was obtained from the parents of all 137 children who were screened for CD in 19947 (mean age, 5.3 years ± 3.7 SD; range, 1-17.6; male/female ratio = 1:1). Screening was offered again in 1996 and in 1999.
Clinical assessment was performed at home to
RESULTS
In 1994, 8 of the 137 screened children were given a diagnosis of CD (Fig 1).7In 1996, 125 of the 129 children without CD were approached again for screening: 3 children could not be contacted, and 1 child was not invited because he had developed leukemia. The parents of 85 individuals (mean age ± SD, 7.5 ± 6.5 years; range, 2.7-19.8 years; male/female ratio = 1:1.2) gave informed consent. Five children had IgA-AEA and
DISCUSSION
Our study confirms that CD is much more frequent in persons with DS than in the general Dutch population (0.5%) and that early detection and treatment of CD in children with DS improves their clinical condition and reverses the small bowel damage. Therefore all children with DS should be screened for CD. However, which approach should be used? The determination of IgA-AEA is widely accepted to be the most specific screening test.4, 7, 8 Confirming the results from others in the general
Acknowledgements
We thank Dr J. Bouquet (Sophia Children’s Hospital, Rotterdam) for providing clinical data, Dr G. M. T. Schreuder (Leiden University Medical Center) for HLA-typing analysis, and Prof Dr J. M. Wit (Leiden University Medical Center), Prof Dr S. P. Verloove-Vanhorick (TNO-PG, Leiden), and E. A. B. de Graaf (Dutch Down Syndrome Foundation) for critically reading the manuscript.
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Supported by the Dutch Ministry of Health, Welfare and Sports, Praeventiefonds/ZON, the Leiden University Research Information Center, and Astra Zeneca, the Netherlands.
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Reprint requests: M. Luisa Mearin, MD, PhD, Leiden University Medical Center, Department of Paediatrics, J6-S-220, PO Box 9600, 2300 RC Leiden, The Netherlands.