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Tsuge, Mitsuru Department of Pediatric Acute Diseases, Okayama University Academic Field of Medicine, Dentistry, and Pharmaceutical Sciences ORCID Kaken ID researchmap
Shigehara, Kenji Department of Pediatrics, Okayama University Hospital
Uda, Kazuhiro Department of Pediatrics, Okayama University Hospital
Kawano, Seiji Department of Gastroenterology and Hepatology, Okayama University Hospital ORCID
Iwamuro, Masaya Department of Gastroenterology and Hepatology, Okayama University Hospital ORCID Kaken ID publons researchmap
Saito, Yukie Department of Pediatrics, Okayama University Hospital
Yashiro, Masato Department of Pediatrics, Okayama University Hospital Kaken ID
Ikeda, Masanori Department of Pediatrics, Okayama University Hospital Kaken ID publons researchmap
Tsukahara, Hirokazu Department of Pediatrics, Okayama University Academic Field of Medicine, Dentistry, and Pharmaceutical Sciences Kaken ID publons researchmap
Abstract
Background Non-esophageal eosinophilic gastrointestinal disorder (non-EoE-EGID) is a rare disease in which eosinophils infiltrate parts of the gastrointestinal tract other than the esophagus; however, the number of patients with non-EoE-EGID has been increasing in recent years. Owing to its chronic course with repeated relapses, it can lead to developmental delays due to malnutrition, especially in pediatric patients. No established treatment exists for non-EoE-EGID, necessitating long-term systemic corticosteroid administration. Although the efficacy of dupilumab, an anti-IL-4/13 receptor monoclonal antibody, for eosinophilic esophagitis, has been reported, only few reports have demonstrated its efficacy in non-EoE EGIDs.
Case presentation A 13-year-old boy developed non-EoE-EGID with duodenal ulcers, with chicken eggs as the trigger. He was successfully treated with an egg-free diet, proton pump inhibitors, and leukotriene receptor antagonists. However, at age 15, he developed worsening upper abdominal pain and difficulty eating. Blood analysis revealed eosinophilia; elevated erythrocyte sedimentation rate; and elevated levels of C-reactive protein, total immunoglobulin E, and thymic and activation-regulated chemokines. Upper gastrointestinal endoscopy revealed a duodenal ulcer with marked mucosal eosinophilic infiltration. Gastrointestinal symptoms persisted even after starting systemic steroids, making it difficult to reduce the steroid dose. Subcutaneous injection of dupilumab was initiated because of comorbid atopic dermatitis exacerbation. After 3 months, the gastrointestinal symptoms disappeared, and after 5 months, the duodenal ulcer disappeared and the eosinophil count decreased in the mucosa. Six months later, systemic steroids were discontinued, and the duodenal ulcer remained recurrence-free. The egg challenge test result was negative; therefore, the egg-free diet was discontinued. Blood eosinophil count and serum IL-5, IL-13, and eotaxin-3 levels decreased after dupilumab treatment. The serum levels of IL-5 and eotaxin-3 remained within normal ranges, although the blood eosinophil counts increased again after discontinuation of oral prednisolone.
Conclusions Suppression of IL-4R/IL-13R-mediated signaling by dupilumab may improve abdominal symptoms and endoscopic and histologic findings in patients with non-EoE-EGID, leading to the discontinuation of systemic steroid administration and tolerance of causative foods.
Keywords
Duodenal ulcer
Dupilumab
Eosinophilic gastroenteritis
Eotaxin-3
Food allergy
Interleukin-5
Interleukin-13
Non-esophageal eosinophilic gastrointestinal disorder
Note
The version of record of this article, first published in Allergy, Asthma & Clinical Immunology, is available online at Publisher’s website: http://dx.doi.org/10.1186/s13223-023-00859-3
Published Date
2023-12-05
Publication Title
Allergy, Asthma & Clinical Immunology
Volume
volume19
Issue
issue1
Publisher
BMC
Start Page
103
ISSN
1710-1492
Content Type
Journal Article
language
English
OAI-PMH Set
岡山大学
Copyright Holders
© The Author(s) 2023.
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isVersionOf https://doi.org/10.1186/s13223-023-00859-3
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http://creativecommons.org/licenses/by/4.0/
Citation
Tsuge, M., Shigehara, K., Uda, K. et al. Successful use of dupilumab for egg-induced eosinophilic gastroenteritis with duodenal ulcer: a pediatric case report and review of literature. Allergy Asthma Clin Immunol 19, 103 (2023). https://doi.org/10.1186/s13223-023-00859-3