Prospective human leukocyte antigen, endomysium immunoglobulin A antibodies, and transglutaminase antibodies testing for celiac disease in children with Down syndrome

J Pediatr. 2009 Feb;154(2):239-42. doi: 10.1016/j.jpeds.2008.08.007. Epub 2008 Sep 25.

Abstract

Objective: To assess the effect of a prospective screening strategy for the early diagnosis of celiac disease (CD) in children with Down syndrome (DS).

Study design: Blood samples were taken from 155 children with DS. Buccal swabs were also taken from 9 of these children for determination of human leukocyte antigen (HLA)-DQ2 or HLA-DQ8 positivity. Independently, immunoglobulin A anti-endomysium-(EMA) and anti-tissue transglutaminase antibodies (TGA) were tested. An intestinal biopsy was performed to confirm the diagnosis of CD.

Results: Sixty-three children (40.6%) had test results that were positive for HLA-DQ2 or HLA-DQ8. Results of HLA DQ-typing of DNA isolated from blood and buccal swabs were identical. Eight of the children in whom test results were positive for HLA-DQ2/8 also had positive test results for EMA and TGA. CD was confirmed in 7 of these children with an intestinal biopsy, and in 1 child, CD was suggested with improvement on a gluten-free diet.

Conclusions: We found a prevalence of CD in children with DS of 5.2% (10 times higher than the general Dutch population). We recommend HLA-DQ2/8 typing from buccal swabs in the first year of life and initiating serologic screening of children with DS in whom test results are positive for HLA-DQ2 or DQ8 at age 3 years. Early knowledge of negative HLA-DQ2/8 status can reassure most parents that their children do not have a CD risk.

MeSH terms

  • Adolescent
  • Adult
  • Autoantibodies / blood*
  • Biopsy
  • Celiac Disease / diagnosis*
  • Celiac Disease / immunology
  • Child
  • Child, Preschool
  • Down Syndrome / complications*
  • Down Syndrome / genetics
  • Duodenum / pathology
  • Early Diagnosis
  • Feasibility Studies
  • Female
  • HLA Antigens / blood*
  • HLA-DQ Antigens / blood
  • Heterozygote
  • Homozygote
  • Humans
  • Immunoglobulin A / immunology*
  • Infant
  • Intestinal Mucosa / pathology
  • Male
  • Mass Screening
  • Prospective Studies
  • Transglutaminases / immunology*
  • Young Adult

Substances

  • Autoantibodies
  • HLA Antigens
  • HLA-DQ Antigens
  • HLA-DQ2 antigen
  • HLA-DQ8 antigen
  • Immunoglobulin A
  • Transglutaminases