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The Characteristics of Isolated Bulb Celiac Disease in Children
Supriya Behl1, Muhammad Rehan Khan2, Yasmine Ismail3
1From the Department of Pediatric and Adolescent Medicine, Mayo Clinic, Rochester, MN.
Insights
In celiac disease (CD) diagnosis, isolated duodenal bulb biopsies are crucial, especially in children with lower antibody levels. Separating biopsies aids in identifying this specific patient group.
Area of Science:
- Pediatric Gastroenterology
- Celiac Disease Diagnosis
- Gastrointestinal Pathology
Background:
- Celiac disease (CD) diagnosis relies on detecting mucosal injury, which can be patchy.
- Up to 12% of CD patients may exhibit mucosal changes exclusively in the duodenal bulb.
- Current guidelines recommend biopsies from both the duodenal bulb and distal duodenum.
Purpose of the Study:
- To describe a cohort of children diagnosed with isolated celiac disease of the duodenal bulb.
- To evaluate the diagnostic benefit of separating duodenal bulb biopsies from distal duodenal biopsies.
Main Methods:
- Retrospective chart review of pediatric CD patients undergoing endoscopy with separated bulb and distal duodenum biopsies.
- Analysis of data from January 2011 to January 2022 across two medical centers.
- Pathologist review using Marsh-Oberhuber grading on selected biopsies.
Main Results:
- 15% (33/224) of pediatric CD patients had isolated bulb CD.
- Patients with isolated bulb CD were older at diagnosis (10 vs. 8 years) and had lower median anti-tissue transglutaminase IgA (TTG IgA) levels (2.8 vs. 16.7 x ULN).
- Approximately 88% of isolated bulb CD patients had TTG IgA levels below 10x ULN; biopsy distinction was challenging in one-third of cases.
Conclusions:
- Separating duodenal bulb biopsies is valuable for celiac disease diagnosis in children, particularly those with TTG IgA levels <10x ULN.
- Further prospective studies are needed to determine if isolated bulb CD represents a distinct entity or an early stage of conventional CD.
Objectives:
Mucosal injury in celiac disease (CD) patients can be patchy, and up to 12% of CD patients can have mucosal changes limited to the duodenal bulb. Hence, recent guidelines recommend obtaining bulb biopsies in addition to distal duodenum. This study aimed to describe a cohort of children with isolated bulb CD and assess the benefit of separating bulb biopsies.
Methods:
A retrospective chart review between January 2011 and January 2022 at 2 medical centers was conducted. We included children with CD who underwent endoscopy with separated biopsies from the bulb and distal duodenum. A blinded pathologist performed Marsh-Oberhuber grading on selected cases.
Results:
We identified 224 CD patients, of which 33 (15%) had histologically confirmed isolated bulb CD. Patients with isolated bulb CD were older at diagnosis (10 vs 8 years; P = 0.03). Median anti-tissue transglutaminase immunoglobulin A (TTG IgA) level was lower in isolate bulb CD (2.8 vs 16.7 times the upper limit of normal [ULN], P < 0.001). Almost 88% (29/33) of isolated bulb CD patients had an anti-TTG IgA value of less than 10 times the ULN. Time to anti-TTG IgA normalization (mean 14 months) was similar between the 2 groups. A pathologist review of diagnostic biopsies could not distinguish between the bulb and distal duodenum biopsies in approximately one-third of the reviewed samples.
Conclusions:
Separating bulb from distal duodenum biopsies can be considered during CD diagnosis, particularly in children with anti-TTG IgA levels less than 10 times the ULN. Larger prospective cohorts are needed to decide whether isolated bulb CD is a unique cohort or an early stage of the conventional CD.
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