Eosinophilic gastroenteritis with refractory ulcer disease and gastrointestinal bleeding as a rare manifestation of seronegative gastrointestinal food allergy

Abstract

Gastrointestinal bleeding and iron deficiency anaemia may cause severe symptoms and may require extensive diagnostics and substantial amounts of health resources.This case report focuses on the clinical presentation of a 22 year old patient with recurrent gastrointestinal bleeding from multilocular non-healing ulcers of the stomach, duodenum and jejunum over a period of four years. Extensive gastroenterological and allergological standard diagnostic procedures showed benign ulcerative lesions with tissue eosinophilia, but no conclusive diagnosis. Multiple diagnostic procedures were performed, until finally, endoscopically guided segmental gut lavage identified locally produced, intestinal IgE antibodies by fluoro-enzyme-immunoassay.IgE antibody concentrations at the intestinal level were found to be more-fold increased for total IgE and food-specific IgE against nuts, rye flour, wheat flour, pork, beef and egg yolk compared with healthy controls.Thus, a diet eliminating these allergens was introduced along with antihistamines and administration of a hypoallergenic formula, which resulted in complete healing of the multilocular ulcers with resolution of gastrointestinal bleeding. All gastrointestinal lesions disappeared and total serum IgE levels dropped to normal within 9 months. The patient has been in remission now for more than two years.Eosinophilic gastroenteritis (EG) is well known to induce refractory ulcer disease. In this case, the mechanisms for intestinal damage and gastrointestinal bleeding were identified as local gastrointestinal type I allergy. Therefore, future diagnostics in EG should also be focused on the intestinal level as identification of causative food-specific IgE antibodies proved to be effective to induce remission in this patient.

Figure 1

Endoscopic and histologic findings in the patient with recurrent gastrointestinal bleeding and seronegative gastrointestinal bleeding before and after antiallergic treatment. A: Several confluating fibrinous ulcers and swelling within the bulb of the duodenum persisting for more than 3 years despite successful helicobacter eradication. B: Chronic inflammation of the duodenum with dense eosinophils within the mucosa. C: Endoscopic view to distal bulb of the duodenum without ulcers or swelling 9 months after allergen elimination, but beginning deformation of the bulb. D: Chronic inflammation of the duodenum, but decrease of eosinophils.

Figure 2

Luminal immune diagnostics (median, 25-75 ^th percentile) in the patient with eosinophilic gastroenteritis compared to healthy controls. Endoscopically guided segmental lavage was performed at duodenum, mid jejunum, cecum and rectosigmoid according to the methods published previously [2, 5]. Medians and 25–75% percentile were calculated from the values obtained at the four lavage sites and compared with the normal range of immune parameters from 12 healthy controls.

Figure 3

Intestinal food-specific IgE concentrations in the patient with eosinophilic gastroenteritis. Endoscopically guided segmental lavage was performed at duodenum, mid jejunum, cecum and rectosigmoid according to the methods published previously [2, 5, 7]. Medians and 25–75% percentile are calculated from the values obtained at the four lavage sites. Food-specific IgE was measured by ThermoFischer Cap-System with significantly elevated levels of >0.35 KU/mg protein as reported from other investigators [3, 5, 7, 12, 15, 16]. Following allergens were negative with specific IgE titers <0.35 KU/mg protein: Egg white, caseine, lactalbumine, gliadin, rice, soy bean, soy Gly m4, potato, celery.

Figure 4

Hemoglobin and serum IgE levels in the patient with recurrent gastrointestinal bleeding and seronegative gastrointestinal allergy. After elimination of the causative allergens along with antiallergic treatment a continuous increase of hemoglobin, iron and ferritin levels was noted, while serum IgE dropped to nearly normal values. The laboratory changes were accompanied by a complete endoscopic and histologic healing of gastrointestinal ulcers (Figure 1).