Food as a trigger for abdominal angioedema attacks in patients with hereditary angioedema
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RESEARCH
Open Access
Food as a trigger for abdominal angioedema attacks in patients with hereditary angioedema Urs C. Steiner1*, Lea Kölliker1, Christina Weber-Chrysochoou2, Peter Schmid-Grendelmeier2, Elsbeth Probst1, Walter A. Wuillemin3 and Arthur Helbling4
Abstract Background: Hereditary angioedema with C1 inhibitor deficiency (C1-INH-HAE) is a rare inherited disease. In most HAE-affected subjects, defined trigger factors precede angioedema attacks. Mechanisms of how trigger factors stimulate the contact activation pathway with bradykinin generation are not well elucidated. In recent studies, hypersensitivity reactions and food were stated as relevant triggers. We investigated HAE affected people for possible hypersensitivity reactions or intolerances and their relation in triggering angioedema attacks. Methods: A questionnaire was filled in, recording date of birth, gender, and self-reported angioedema attacks associated with the ingestion of foodstuffs, administration of drugs, hymenoptera stings and hypersensitivity reactions against inhalation allergens. All participants performed a skin prick test against inhalation allergens and food. In patients who stated an association of possible hypersensitivity with angioedema, a serological ImmunoCAP test was also performed. Results: From the 27 women and 15 men analyzed, 79% stated trigger factors. From those food was mentioned in 36%. The suspected food included tomato, green salad, fish, citrus fruits, apple, onion, garlic, cheese, chili, kiwi, milk, tree nuts, strawberry, pineapple, shrimps, bread, banana, leek, chicken and alcohol, and were associated with abdominal angioedema. Neither the skin prick test nor the ImmunoCAP-test turned out positive for the tested food allergens. Conclusion: Food seems to be a relevant trigger factor, causing angioedema in HAE affected patients. The reason, however, is not IgE-mediated hypersensitivity, but most probably an intolerance reaction to food products. Keywords: Hereditary angioedema (HAE), Trigger factors, Allergy, Intolerance
Background Hereditary angioedema with C1 inhibitor deficiency (C1-INH-HAE) is a rare inherited disease, due to mutations of the SERPING1 gene. Two different variants have been described. Type I is characterized by a quantitative decrease in C1-INH protein, in Type II, the C1-INH protein has a normal level, but is dysfunctional [1, 2]. C1-INH inhibits factors of the contact system including FXII and kallikrein, and controls the generation of vasoactive peptides such as bradykinin [3]. C1-INH-HAE is characterized by episodic attacks of subcutaneous and * Correspondence: [email protected] 1 Department of Clinical Immunology, University Hospital Zurich, Zurich, Switzerland Full list of author information is available at the end of the article
submucosal swelling affecting extremities, trunk, genitals, and face, and might be life threatening when upper airways and abdomen are affected [4, 5]. Although it is a systemic activation process, frequency and localization of angioedema are highly varia
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