Atypical Food protein-induced enterocolitis syndrome in children: Is IgE sensitization an issue longitudinally?

Abstract

Background: Food Protein-Induced Enterocolitis Syndrome (FPIES) is a clinically well-characterized, non-IgE mediated, food allergy syndrome yet its rare atypical presentation remain poorly understood.Objective: Aim of this study was to present the 10-year experience of a referral center highlighting the atypical FPIES cases and their long-term outcome.Methods: FPIES cases were prospectively, longitudinally evaluated in respect of food outgrowth and developing other allergic diseases with or without concomitant IgE sensitization later on in life.Results: Out of a total of 14,188 referrals (0.7%), 100 cases were identified. At presentation, 15 cases were found sensitized to the offending food. Fish was the most frequent eliciting food, followed by cow’s milk and egg. Tolerance acquisition was earlier for cow’s milk, followed by egg and fish, while found not protracted in atypical cases. Resolution was not achieved in half of the fish cases during the 10-year follow up time. Sensitization to food was not related to infantile eczema or culprit food but was related to sensitization to aeroallergens. In the long-term evaluation, persistence of the FPIES or aeroallergen’s sensitization was significantly associated with an increased hazard risk of developing early asthma symptoms.Conclusion: Sensitization to food was neither related to eczema, culprit food, nor to tolerance acquisition but rather to the development of allergic asthma through aero sensitization. In addition to an IgE profile in an early age, FPIES persistence may also trigger mechanisms switching FPIES cases to a Th2 immune response later in life, predisposing to atopic respiratory symptoms; albeit further research is required.