Incidental abnormalities are seldom missed in children satisfying the European Society for Paediatric Gastroenterology, Hepatology and Nutrition non-biopsy criteria: A retrospective single-center study.

Abstract

Background and Aim: Obtaining duodenal mucosal biopsy samples during gastroduodenoscopy has long been the gold standard in celiac disease (CeD) diagnosis. However, this paradigm was challenged in 2012 by the release of the European Society for Paediatric Gastroenterology, Hepatology and Nutrition (ESPGHAN) non-biopsy criteria. Under the 2020 ESPGHAN guidelines, pediatric patients satisfying the serological criteria may receive a diagnosis of CeD without duodenal biopsy samples and subsequent histological confirmation being obtained. We aimed todetermine the frequency of incidental abnormalities missed by omitting biopsies, in accordance with the 2020 ESPGHAN non-biopsy criteria, in an Australian pediatric population. Method(s): We performed a retrospective case notes review of all patients who had undergone investigation for CeD with subsequent histological diagnosis from 2013 to 2018 at a large tertiary center in Melbourne. Eligible patients were selected on meeting our selection criteria of age < 18 years, anti-tissue transglutaminase IgA > 10 x upper limit of normal (ULN), and deamidated gliadin peptide (DGP) IgA or IgG > 10 x ULN. Our center's pathology database and electronic medical records were consulted to derive the final subset of pediatric patients included in our study. Histopathological reports of all biopsy samples obtained from eligible patients were reviewed, and any incidental abnormalities identified by consultant pathologists were noted. Incidental abnormalities identified were reviewed for clinical significance, which we defined as those potentially requiring further treatment or follow-up. Result(s): During the study period, 252 children were diagnosed with CeD; however, only 44 (17.5%) (25 female, 19 male; median ages, 6 and 7 years, respectively) satisfied our inclusion criteria for non-biopsy diagnosis. A total of 369 biopsy samples were obtained: 260 duodenal (median, 6), 82 gastric (median, 2), and 27 esophageal (median, 2). All patients had duodenal biopsies, with additional esophageal and gastric biopsies performed in 12 (27.3%) and 43 (97.7%), respectively. We found that nine of 44 patients (20.5%), of whom eight were female, had incidental abnormalities. These were lymphocytic gastritis (five female patients, one male) and esophagitis (four female patients). One female patient had both esophagitis and lymphocytic gastritis. Conclusion(s): Our study suggests that significant abnormalities occur infrequently in an Australian pediatric population qualifying for the ESPGHAN non-biopsy approach and, therefore, the implementation of ESPGHAN criteria in the diagnosis of CeD is safe when applied appropriately. We identified four of 44 patients (9.1%) with esophagitis, which is of potential significance. Lymphocytic gastritis is a common concurrent finding in patients with CeD that is unlikely to alter the patient's clinical management and thus was not considered significant. Our study incorporated DGP IgA/IgG serology instead of endomysial antibodies (EMA) to reflect ESPGHAN criteria of a second positive celiac serological marker, as EMA is not part of routine celiac serology in Australia. Our finding that 9.1% of patients had concurrent incidental abnormalities of clinical significance is consistent with prior studies. This suggests that duodenal biopsies are still highly relevant for a large proportion of pediatric patients and have a prominent role in the ongoing diagnosis of CeD.