LAUSR

Eosinophilic gastrointestinal diseases (EGIDs) are characterized by abnormal eosinophilic infiltration of different segments of the gastrointestinal tract in the absence of identifiable secondary causes. EGIDs are often associated with other atopic conditions. Comorbid food allergies have been noted in only 3.4% of patients with eosinophilic gastroenteritis (EGE). Although dietary treatments, including elimination diets and empirical food avoidance, are known treatment options for eosinophilic esophagitis, the effectiveness of dietary therapies for EGE remains to be clarified. Since responses to dietary therapies do not always correlate with the specific food sensitization determined by skin test and/or serum antigen-specific IgE, it is difficult to identify a causative food for empirical avoidance. In this report we describe a case of EGE that developed after long-term sustained unresponsiveness of IgE-mediated cow’s milk (CM) allergy that was effectively treated with the elimination of dairy products. A female infant, exclusively breastfed from birth, experienced several episodes of urticaria within 1 h of ingesting CM and boiled egg during weaning. Because her serum-specific IgE (sIgE) (ImmunoCAP, Phadia, Uppsala, Sweden) to CM and egg white was 0.48 and 6.32 kUa/L, respectively, she was diagnosed as having IgE-mediated CM and egg allergies. Dairy and egg products were eliminated from her diet. At 2 years 10 months of age, a follow-up examination revealed decreases in sIgE. Dairy and egg products were reintroduced, and their intakes were gradually increased without elicitation of any symptoms. At 4 years 7 months of age, when viral enterocolitis was epidemic in the child’s kindergarten, the child presented with vomiting and diarrhea. She was treated with Bifidobacterium probiotics. Four weeks later, she was referred to our hospital because of weight loss, persistent diarrhea, hematochezia, and a remarkable eosinophilia (WBC 24 700/μL, eosinophils 40%). On admission, serum sIgE showed; egg yolk 0.38 kUa/L, egg white 3.64 kUa/L, and CM 0.44 kUa/L. An allergen-specific lympho-proliferation index against lactoferrin was positive, whereas those against j-casein, and human a-lactalbumin were negative. Abdominal computed tomography revealed a thickened intestinal wall. Upper and lower endoscopic examinations showed no apparent mucosal lesions, but duodenum biopsy specimens obtained by endoscopy revealed infiltration of eosinophils (> 100 eosinophils/high-power field) in the mucosa (Fig 1). Eosinophil infiltration was not remarkable in gastric and ileal mucosa. Bone marrow examination showed no monoclonal cell proliferation, and FIP1L1-PDGFa and JAK2 mutations were not detected. Parasite screening was negative. Chest computed tomography and echography did not show abnormal findings. The patient was diagnosed as having EGE. After 6 days of fasting and parenteral nutrition, her gastrointestinal symptoms disappeared and a diet excluding dairy and egg products was reinstated. Although egg products were resumed on the 18th day of hospitalization, absolute eosinophil counts continuously decreased until dairy products were reintroduced on the 25th hospital day. After commencement of dairy products, she complained of intermittent abdominal pains and presented with progressive diarrhea but no cutaneous or respiratory symptoms. Peripheral blood eosinophil counts increased over the following 5 days. These symptoms and eosinophilia disappeared after