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Published in: Allergy, Asthma & Clinical Immunology 1/2024

Open Access 01-12-2024 | Omalizumab | Case report

Eosinophilic gastritis and gluten-sensitive enteropathy manifested as hypoproteinemia and treated with omalizumab: a case report

Authors: Zhirong Du, Zixi Wang, Weixun Zhou, Jia Yin, Yuxiang Zhi

Published in: Allergy, Asthma & Clinical Immunology | Issue 1/2024

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Abstract

Background

Eosinophilic gastritis (EoG) has rarely been reported in conjunction with gluten-sensitive enteropathy (GSE). When this does occur, patients typically present with gastrointestinal symptoms. To our knowledge, hypoproteinemia has not been reported as the primary manifestation. Anti-IgE therapy, such as omalizumab, lowers eosinophil counts in the blood, lungs, and gut. Its efficiency in treating active EoG remain unknown.

Case presentation

We report a 33-month-old boy with a history of food allergy and atopic dermatitis who developed recurrent edema, hypoproteinemia, and eosinophilia at the age of 14 months. The diagnoses of EoG and GSE were confirmed based on the clinical presentation and results of gastrointestinal biopsies and serological testing. Although prednisone and dietary intervention were initially effective, the boy developed prednisone-related facial swelling. After stopping prednisone, his symptoms relapsed. Subsequent treatment with omalizumab, combined with dietary intervention, showed good efficacy and safety.

Conclusions

To our knowledge, this is the first case of concurrent EoG and GSE that presented primarily with hypoproteinemia. We highlight the rare manifestations of these two diseases to raise clinical suspicion and prevent missed and delayed diagnoses. The pathogenesis of EoG is heterogeneous and complex. Omalizumab showed good efficacy, indicating that IgE-mediated processes may be involved in the pathogenesis of this patient’s diseases.
Literature
1.
go back to reference Dellon ES, Gonsalves N, Abonia JP, Alexander JA, Arva NC, Atkins D, et al. International Consensus recommendations for Eosinophilic Gastrointestinal Disease nomenclature. Clin Gastroenterol Hepatol. 2022;20:2474–84.CrossRefPubMedPubMedCentral Dellon ES, Gonsalves N, Abonia JP, Alexander JA, Arva NC, Atkins D, et al. International Consensus recommendations for Eosinophilic Gastrointestinal Disease nomenclature. Clin Gastroenterol Hepatol. 2022;20:2474–84.CrossRefPubMedPubMedCentral
2.
go back to reference Mansoor E, Saleh MA, Cooper GS. Prevalence of eosinophilic gastroenteritis and colitis in a Population-based study, from 2012 to 2017. Clin Gastroenterol Hepatol. 2017;15:1733–41.CrossRefPubMed Mansoor E, Saleh MA, Cooper GS. Prevalence of eosinophilic gastroenteritis and colitis in a Population-based study, from 2012 to 2017. Clin Gastroenterol Hepatol. 2017;15:1733–41.CrossRefPubMed
3.
go back to reference Yuan J, Zhou C, Gao J, Li J, Yu F, Lu J, et al. Prevalence of Celiac Disease autoimmunity among adolescents and young adults in China. Clin Gastroenterol Hepatol. 2017;15:1572–9.CrossRefPubMed Yuan J, Zhou C, Gao J, Li J, Yu F, Lu J, et al. Prevalence of Celiac Disease autoimmunity among adolescents and young adults in China. Clin Gastroenterol Hepatol. 2017;15:1572–9.CrossRefPubMed
4.
go back to reference Chen PH, Anderson L, Zhang K, Weiss GA. Eosinophilic Gastritis/Gastroenteritis. Curr Gastroenterol Rep. 2021;23:13.CrossRefPubMed Chen PH, Anderson L, Zhang K, Weiss GA. Eosinophilic Gastritis/Gastroenteritis. Curr Gastroenterol Rep. 2021;23:13.CrossRefPubMed
5.
go back to reference McAllister BP, Williams E, Clarke K. A Comprehensive Review of Celiac Disease/Gluten-Sensitive enteropathies. Clin Rev Allergy Immunol. 2019;57:226–43.CrossRefPubMed McAllister BP, Williams E, Clarke K. A Comprehensive Review of Celiac Disease/Gluten-Sensitive enteropathies. Clin Rev Allergy Immunol. 2019;57:226–43.CrossRefPubMed
6.
go back to reference Walker MM, Potter M, Talley NJ. Eosinophilic gastroenteritis and other eosinophilic gut diseases distal to the oesophagus. Lancet Gastroenterol Hepatol. 2018;3:271–80.CrossRefPubMed Walker MM, Potter M, Talley NJ. Eosinophilic gastroenteritis and other eosinophilic gut diseases distal to the oesophagus. Lancet Gastroenterol Hepatol. 2018;3:271–80.CrossRefPubMed
7.
go back to reference Sato H, Honma T, Owaki T, Tominaga K, Yokoyama J, Terai S. Clinical and pathological profile of eosinophilic gastroenteritis. Eur J Gastroenterol Hepatol. 2019;31:157–62.CrossRefPubMed Sato H, Honma T, Owaki T, Tominaga K, Yokoyama J, Terai S. Clinical and pathological profile of eosinophilic gastroenteritis. Eur J Gastroenterol Hepatol. 2019;31:157–62.CrossRefPubMed
8.
go back to reference Halaby H, Bakheet SM, Shabib S, Powe JE, Al Mehaidib A, Nazer H. 99mTc-human serum albumin scans in children with protein-losing enteropathy. J Nucl Med. 2000;41:215–9.PubMed Halaby H, Bakheet SM, Shabib S, Powe JE, Al Mehaidib A, Nazer H. 99mTc-human serum albumin scans in children with protein-losing enteropathy. J Nucl Med. 2000;41:215–9.PubMed
10.
go back to reference Sunkara T, Rawla P, Yarlagadda KS, Gaduputi V. Eosinophilic gastroenteritis: diagnosis and clinical perspectives. Clin Exp Gastroenterol. 2019;12:239–53.CrossRefPubMedPubMedCentral Sunkara T, Rawla P, Yarlagadda KS, Gaduputi V. Eosinophilic gastroenteritis: diagnosis and clinical perspectives. Clin Exp Gastroenterol. 2019;12:239–53.CrossRefPubMedPubMedCentral
11.
go back to reference Gonsalves N, Doerfler B, Zalewski A, Yang GY, Martin LJ, Zhang X, et al. Prospective study of an amino acid-based elemental diet in an eosinophilic gastritis and gastroenteritis nutrition trial. J Allergy Clin Immunol. 2023;152:676–88.CrossRefPubMed Gonsalves N, Doerfler B, Zalewski A, Yang GY, Martin LJ, Zhang X, et al. Prospective study of an amino acid-based elemental diet in an eosinophilic gastritis and gastroenteritis nutrition trial. J Allergy Clin Immunol. 2023;152:676–88.CrossRefPubMed
12.
13.
go back to reference Levitt DG, Levitt MD. Protein losing enteropathy: comprehensive review of the mechanistic association with clinical and subclinical disease states. Clin Exp Gastroenterol. 2017;10:147–68.CrossRefPubMedPubMedCentral Levitt DG, Levitt MD. Protein losing enteropathy: comprehensive review of the mechanistic association with clinical and subclinical disease states. Clin Exp Gastroenterol. 2017;10:147–68.CrossRefPubMedPubMedCentral
14.
go back to reference Brown IS, Smith J, Rosty C. Gastrointestinal pathology in celiac disease: a case series of 150 consecutive newly diagnosed patients. Am J Clin Pathol. 2012;138:42–9.CrossRefPubMed Brown IS, Smith J, Rosty C. Gastrointestinal pathology in celiac disease: a case series of 150 consecutive newly diagnosed patients. Am J Clin Pathol. 2012;138:42–9.CrossRefPubMed
15.
go back to reference Talley NJ, Kephart GM, McGovern TW, Carpenter HA, Gleich GJ. Deposition of eosinophil granule major basic protein in eosinophilic gastroenteritis and celiac disease. Gastroenterology. 1992;103:137–45.CrossRefPubMed Talley NJ, Kephart GM, McGovern TW, Carpenter HA, Gleich GJ. Deposition of eosinophil granule major basic protein in eosinophilic gastroenteritis and celiac disease. Gastroenterology. 1992;103:137–45.CrossRefPubMed
16.
go back to reference Desreumaux P, Delaporte E, Colombel JF, Capron M, Cortot A, Janin A, Similar. IL-5, IL-3, and GM-CSF syntheses by eosinophils in the jejunal mucosa of patients with celiac disease and dermatitis herpetiformis. Clin Immunol Immunopathol. 1998;88:14–21.CrossRefPubMed Desreumaux P, Delaporte E, Colombel JF, Capron M, Cortot A, Janin A, Similar. IL-5, IL-3, and GM-CSF syntheses by eosinophils in the jejunal mucosa of patients with celiac disease and dermatitis herpetiformis. Clin Immunol Immunopathol. 1998;88:14–21.CrossRefPubMed
17.
go back to reference Björck S, Lindehammer SR, Fex M, Agardh D. Serum cytokine pattern in young children with screening detected coeliac disease. Clin Exp Immunol. 2015;179:230–5.CrossRefPubMedPubMedCentral Björck S, Lindehammer SR, Fex M, Agardh D. Serum cytokine pattern in young children with screening detected coeliac disease. Clin Exp Immunol. 2015;179:230–5.CrossRefPubMedPubMedCentral
18.
go back to reference Butterfield JH, Murray JA. Eosinophilic gastroenteritis and gluten-sensitive enteropathy in the same patient. J Clin Gastroenterol. 2002;34:552–3.CrossRefPubMed Butterfield JH, Murray JA. Eosinophilic gastroenteritis and gluten-sensitive enteropathy in the same patient. J Clin Gastroenterol. 2002;34:552–3.CrossRefPubMed
19.
go back to reference Bennett RA, Whitelock T 3rd, Kelley JL Jr. Eosinophilic gastroenteritis, gluten enteropathy, and dermatitis herpetiformis. Am J Dig Dis. 1974;19:1154–61.CrossRefPubMed Bennett RA, Whitelock T 3rd, Kelley JL Jr. Eosinophilic gastroenteritis, gluten enteropathy, and dermatitis herpetiformis. Am J Dig Dis. 1974;19:1154–61.CrossRefPubMed
20.
go back to reference Turner D, Wolters VM, Russell RK, Shakhnovich V, Muise AM, Ledder O, et al. Anti-TNF, infliximab, and adalimumab can be effective in eosinophilic bowel disease. J Pediatr Gastroenterol Nutr. 2013;56:492–7.CrossRefPubMed Turner D, Wolters VM, Russell RK, Shakhnovich V, Muise AM, Ledder O, et al. Anti-TNF, infliximab, and adalimumab can be effective in eosinophilic bowel disease. J Pediatr Gastroenterol Nutr. 2013;56:492–7.CrossRefPubMed
21.
go back to reference Li K, Ruan G, Liu S, Xu T, Guan K, Li J, et al. Eosinophilic gastroenteritis: Pathogenesis, diagnosis, and treatment. Chin Med J (Engl). 2023;136:899–909.CrossRefPubMed Li K, Ruan G, Liu S, Xu T, Guan K, Li J, et al. Eosinophilic gastroenteritis: Pathogenesis, diagnosis, and treatment. Chin Med J (Engl). 2023;136:899–909.CrossRefPubMed
22.
go back to reference Foroughi S, Foster B, Kim N, Bernardino LB, Scott LM, Hamilton RG, et al. Anti-IgE treatment of eosinophil-associated gastrointestinal disorders. J Allergy Clin Immunol. 2007;120:594–601.CrossRefPubMedPubMedCentral Foroughi S, Foster B, Kim N, Bernardino LB, Scott LM, Hamilton RG, et al. Anti-IgE treatment of eosinophil-associated gastrointestinal disorders. J Allergy Clin Immunol. 2007;120:594–601.CrossRefPubMedPubMedCentral
23.
go back to reference Caldwell JM, Collins MH, Stucke EM, Putnam PE, Franciosi JP, Kushner JP, et al. Histologic eosinophilic gastritis is a systemic disorder associated with blood and extragastric eosinophilia, TH2 immunity, and a unique gastric transcriptome. J Allergy Clin Immunol. 2014;134:1114–24.CrossRefPubMedPubMedCentral Caldwell JM, Collins MH, Stucke EM, Putnam PE, Franciosi JP, Kushner JP, et al. Histologic eosinophilic gastritis is a systemic disorder associated with blood and extragastric eosinophilia, TH2 immunity, and a unique gastric transcriptome. J Allergy Clin Immunol. 2014;134:1114–24.CrossRefPubMedPubMedCentral
24.
go back to reference Okayama Y, Matsumoto H, Odajima H, Takahagi S, Hide M, Okubo K. Roles of omalizumab in various allergic diseases. Allergol Int. 2020;69:167–77.CrossRefPubMed Okayama Y, Matsumoto H, Odajima H, Takahagi S, Hide M, Okubo K. Roles of omalizumab in various allergic diseases. Allergol Int. 2020;69:167–77.CrossRefPubMed
25.
go back to reference Zuberbier T, Wood RA, Bindslev-Jensen C, Fiocchi A, Chinthrajah RS, Worm M, et al. Omalizumab in IgE-Mediated food allergy: a systematic review and Meta-analysis. J Allergy Clin Immunol Pract. 2023;11:1134–46.CrossRefPubMed Zuberbier T, Wood RA, Bindslev-Jensen C, Fiocchi A, Chinthrajah RS, Worm M, et al. Omalizumab in IgE-Mediated food allergy: a systematic review and Meta-analysis. J Allergy Clin Immunol Pract. 2023;11:1134–46.CrossRefPubMed
26.
go back to reference Al-Toma A, Volta U, Auricchio R, Castillejo G, Sanders DS, Cellier C, et al. European Society for the Study of Coeliac Disease (ESsCD) guideline for coeliac disease and other gluten-related disorders. United Eur Gastroenterol J. 2019;7:583–613.CrossRef Al-Toma A, Volta U, Auricchio R, Castillejo G, Sanders DS, Cellier C, et al. European Society for the Study of Coeliac Disease (ESsCD) guideline for coeliac disease and other gluten-related disorders. United Eur Gastroenterol J. 2019;7:583–613.CrossRef
Metadata
Title
Eosinophilic gastritis and gluten-sensitive enteropathy manifested as hypoproteinemia and treated with omalizumab: a case report
Authors
Zhirong Du
Zixi Wang
Weixun Zhou
Jia Yin
Yuxiang Zhi
Publication date
01-12-2024
Publisher
BioMed Central
Published in
Allergy, Asthma & Clinical Immunology / Issue 1/2024
Electronic ISSN: 1710-1492
DOI
https://doi.org/10.1186/s13223-024-00878-8

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