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Have serological tests changed the face of childhood coeliac disease? A retrospective cohort study
Hélène Garnier-Lengliné1, Nicole Brousse, Sophie Candon
1Pediatric Gastroenterology Unit, Hôpital Necker-Enfants Malades, Paris, France.
Insights
The introduction of antitransglutaminase (tTG) and antiendomysium (EM) antibody testing has identified a new profile of childhood coeliac disease (CD). This includes more asymptomatic cases diagnosed through screening, with less severe symptoms than previously observed.
Area of Science:
- Pediatric Gastroenterology
- Immunology
- Clinical Diagnostics
Background:
- Coeliac disease (CD) diagnosis has evolved with the advent of serological antibody testing.
- Understanding the impact of these diagnostic tools on the clinical presentation of CD in children is crucial.
Purpose of the Study:
- To evaluate how the use of antitransglutaminase (tTG) and antiendomysium (EM) antibodies has altered the profile of childhood coeliac disease.
- To compare clinical, biological, and histological characteristics of CD patients before and after the widespread adoption of antibody testing.
Main Methods:
- Retrospective cohort study comparing two groups of pediatric CD patients.
- Historical cohort (1976-1992) diagnosed without antibody use vs. recent cohort (1994-2007) diagnosed with antibody use.
- Analysis of clinical, biological, and histological data at diagnosis.
Main Results:
- The median age of CD diagnosis increased from 1 year to 2.7 years in the recent cohort.
- Patients in the historical cohort presented with more severe gastrointestinal symptoms and failure to thrive.
- Morphological lesions (villous atrophy) were significantly more severe in the historical cohort compared to the recent cohort.
Conclusions:
- Serological tests have identified a new cohort of children with paucisymptomatic or asymptomatic coeliac disease, often diagnosed via screening.
- While symptoms in the recent cohort were less severe, the long-term risks of untreated silent CD require further investigation.
- Screening in at-risk populations has led to the diagnosis of silent CD.
Objectives:
The aim of this study was to evaluate if the use of antitransglutaminase (tTG) and antiendomysium (EM) antibodies has modified the profile of coeliac disease (CD) in children.
Design:
Retrospective cohort study.
Setting:
Monocentric study, in one major tertiary centre in Paris. Two cohorts of patients were compared; the first included patients before the use of antibodies, and the second included patients after the use of antibodies.
Participants:
All patients from the same physician diagnosed with a CD between 1976 and 1992 (historical cohort), and between 1994 and 2007, were included in the study. 56 patients were included in the historical cohort, 59 in the recent cohort.
Primary And Secondary Outcome Measures:
Clinical, biological and histological profiles at diagnosis have been studied.
Results:
The recent cohort diagnosis of CD was based in 27% on a systematic screening (type I diabetes, n=10; CD in siblings, n=6). On comparison of CD patients in the historical to the recent cohort, the following significant differences were observed: Median age at diagnosis increased from 1 year to 2.7 years (p<0.0001). Patients in the historical cohort had more gastrointestinal symptoms (93% vs 63%, p=0.0001) and failure to thrive (98% vs 80%, p=0.0025). Nutritional deficiencies and morphological lesions were more severe in the historical cohort (90% subtotal or total villous atrophy vs 51%, p<0.0001). Differences observed between the two cohorts were mainly due to the presence of screened patients.
Conclusions:
A new type of patients, with a paucisymptomatic or asymptomatic CD, has been identified using serological tests. Silent disease has been diagnosed by screening in a target population. In the other patients of the recent cohort, symptoms were similar but less severe than those observed before. Long-term risks of untreated silent CD are not well determined as yet, and have to be evaluated in prospective studies.
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