Dupilumab for Treatment of Food-Dependent, Exercise-Induced Anaphylaxis: Report of One Case / 中国医学科学杂志(英文版)

Food-dependent, exercise-induced anaphylaxis (FDEIA) is a potentially life-threatening disorder that often occurs with exercise, and patients typically have eaten a specific food within hours before disease onset. This disease is exceedingly rare, with a prevalence of 0.02%. No well-recognized prevention or treatment strategy has been available for FDEIA except avoiding triggers strictly. Here we report an 11-year-old boy with a history of recurrent anaphylaxis of unknown etiology more than 10 times within two years. As the anaphylactic symptoms had not been controlled after traditional treatments, the patient was given subcutaneous injection of dupilumab seven times within 33 weeks. During dupilumab treatments, the patient was exposed to culprit mushrooms plus exercises at least twice a month but without notable anaphylaxis. Thus, Dupilumab may improve the allergic reactions in FDEIA patients.

A New Diagnostic Criteria of Wheat-Dependent, Exercise-Induced Anaphylaxis in China / 中华医学杂志(英文版)

Background@#Wheat-dependent, exercise-induced anaphylaxis (WDEIA) is an allergic reaction induced by intense exercise combined with wheat ingestion. The gold standard for diagnosis of WDEIA is a food exercise challenge; however, this test is unacceptable for Chinese WDEIA patients and unable to be approved by the Ethics Committee of Chinese hospitals due to substantial risk. There are no diagnostic criteria for Chinese WDEIA patients. The aim of present study was to propose new practical diagnosis criteria for Chinese WDEIA patients.@*Methods@#We prospectively included 283 clinically diagnosed WDEIA patients from January 1, 2010 to June 30, 2014, and in the meanwhile, three groups were enrolled which included 133 patients with the history of anaphylaxis induced by food other than wheat, 186 recurrent urticaria patients, and 94 healthy participants. Clinical comprehensive evaluation by allergists used as the reference gold standard, receiver operator characteristic (ROC) curves were plotted, areas under curve (AUC) for specific immunoglobin E (sIgE) were compared to evaluate the diagnostic value of IgE specific to wheat, gluten, and ω-5 gliadin. Patients were followed up by telephone questionnaire 1 year after diagnosis.@*Results@#We reviewed 567 anaphylactic reactions in 283 WDEIA patients. Of these anaphylactic reactions, 415 (73.3%) reactions were potentially life-threatening anaphylaxis. Among the 567 anaphylactic reactions, 75% (425/567) occurred during exercise. The highest AUC (0.910) was observed for sIgE for gluten, followed by omega-5 gliadin (AUC 0.879). Combined gluten- and ω-5 gliadin-specific IgE testing provided sensitivity and specificity of 73.1% and 99.0%, respectively. During the 1-year follow-up period, repeat anaphylaxis was rare when patients observed strict avoidance of wheat products combined with exercise or other triggering agents.@*Conclusions@#In this study, we proposed diagnostic criteria and management of WDEIA patients in China. Our present study suggested that confirmed anaphylactic reactions triggered by wheat with positive sIgE to gluten and omega-5-gliadin may provide supportive evidence for clinicians to make WDEIA diagnosis without performing a food exercise challenge.

A Case of Exercise-Induced Anaphylaxis Presenting with Lower Lid Angioedema

PURPOSE: To describe a case of exercise-induced anaphylaxis presenting with lower lid angioedema. CASE SUMMARY: A 35-year-old female patient for the past 3 years experienced lower lid edema in her left eye after exercising. In the treadmill exercise test, sneezing, coughing, and lower lid edema in her left eye appeared 13 minutes after initiating exercise. Additionally, contrast-enhanced computed tomography scan revealed minimal soft tissue thickening of the left inferior periorbital subcutaneous area with subtle enhancement after exercise. The serum immunoglobulin E (IgE) test showed elevated total IgE levels. The patient was diagnosed with exercise-induced anaphylaxis based on the above results. We informed the patient on her trigger factors and possible symptoms and prescribed an oral antihistamine and steroid. CONCLUSIONS: In patients presenting with facial edema or eyelid edema after exercise, the possibility of exercise-induced anaphylaxis should be considered. In addition, we have to aware of possibility of complication such as airway obstruction or a life-threatening condition, and it is needed to prevent a recurrence of anaphylaxis by cooperating with other department.

ANAFILAXIA INDUCIDA POR EL EJERCICIO: REVISIÓN DE LA LITERATURA / Exercise-induced anaphylaxis: a review

Durante los últimos años ha habido un aumento en la incidencia de un tipo particular de anafilaxia que solo se manifiesta al realizar algún tipo de actividad física y que se conoce como anafilaxia inducida por el ejercicio. Fue descripta por primera vez en 1979 y definida como una forma de alergia física inducida por el ejercicio, sola o asociada a otros factores precipitantes, caracterizada por una reacción severa, de inicio brusco y rápido. Existe además un subgrupo de pacientes que sólo presentan síntomas si el ejercicio se lleva a cabo dentro de unas pocas horas de ingerir alimentos. Este trastorno se denomina anafilaxia inducida por ejercicio e ingestión de alimentos. La etiopatogenia de la anafilaxia inducida por ejercicio no está del todo aclarada, pero se han postulado distintas teorías que explicarían la liberación final de mediadores vasoactivos. Las manifestaciones clínicas no difieren significativamente de otras anafilaxias y puede ir desde compromiso cutáneo hasta compromiso de vía aérea, shock anafiláctico y eventualmente la muerte del paciente. El diagnóstico de esta enfermedad se basa principalmente en una anamnesis detallada en búsqueda de todos los posibles agentes predisponentes, pruebas de sensibilización inmediata y pruebas de provocación de ejercicio, alimentos o fármacos, si corresponden. La base del tratamiento es evitar los factores desencadenantes, junto con el manejo agudo de la anafilaxia. (AU)

During the last years, there has been an increased incidence of a particular type of anaphylaxis that only manifests with some kind of physical activity and is known as exercise-induced anaphylaxis. Described for the first time in 1979 and defined as a form of physical allergy induced by exercise, alone or associated with other triggers, characterized by a severe reaction which appears very suddenly. There is also a subset of patients that only presents symptoms if the exercise is carried out within a few hours of eating. This condition is called exercise-induced anaphylaxis and food intake. The pathogenesis of the exercise-induced anaphylaxis is not fully clear, but several theories have been postulated to explain the final release of vasoactive mediators. Clinical manifestations do not differ significantly from other anaphylaxis and can range from cutaneous to airway involvement, anaphylactic shock an even death of the patient. The diagnosis of this disease is mainly based in a detailed history in search of all possible predisposing agents, immediate sensitization testing, and challenge tests of exercise, food or drugs if applicable. The mainstay of treatment is to avoid the triggers, along with the acute management of anaphylaxis.(AU)

Wheat - Dependent Exercise-Induced Anaphylaxis Occurred With a Delayed Onset of 10 to 24 hours After Wheat Ingestion: A Case Report

Wheat-dependent exercise-induced anaphylaxis (WDEIA) usually occurs 1 to 4 hours after wheat ingestion and the pathophysiology of WDEIA remains unknown. It is recommended that WDEIA patients refrain from exercise for 4 to 6 hours after wheat ingestion. We report a case of a 51-year-old man who experienced 5 anaphylaxis attacks; two of which occurred 10 to 24 hours after wheat ingestion and exercise. He has a history of chronic gastroenteritis that responds well to antihistamine drugs but not proton pump inhibitors (PPIs) and prokinetic agents. Abdominal CT results implied the possibility of superior mesenteric artery syndrome. We suggest that WDEIA occurs 6 hours after wheat ingestion in cases compounded by obstructive gastrointestinal diseases.

Wheat - Dependent Exercise-Induced Anaphylaxis Occurred With a Delayed Onset of 10 to 24 hours After Wheat Ingestion: A Case Report

Wheat-dependent exercise-induced anaphylaxis (WDEIA) usually occurs 1 to 4 hours after wheat ingestion and the pathophysiology of WDEIA remains unknown. It is recommended that WDEIA patients refrain from exercise for 4 to 6 hours after wheat ingestion. We report a case of a 51-year-old man who experienced 5 anaphylaxis attacks; two of which occurred 10 to 24 hours after wheat ingestion and exercise. He has a history of chronic gastroenteritis that responds well to antihistamine drugs but not proton pump inhibitors (PPIs) and prokinetic agents. Abdominal CT results implied the possibility of superior mesenteric artery syndrome. We suggest that WDEIA occurs 6 hours after wheat ingestion in cases compounded by obstructive gastrointestinal diseases.

A Case Report of Food-Dependent Exercise-Induced Anaphylaxis in a Patient who was Sensitive to Pork / 소아알레르기및호흡기학회지

Food-dependent exercise-induced anaphylaxis (FDEIA) is a specific variant of exercise-induced anaphylaxis that requires both vigorous physical activity and the ingestion of specific foods. In particular, it is rare occurrence for FDEIA to be associated with meat in Korea. A 15-year-old female had generalized urticaria, dyspnea, severe cough, headache, dizziness, and vomiting after singing and dancing for 1 hour and after ingesting grilled pork. Skin prick tests showed a strong positive reaction to pork, whereas the results of an oral food challenge and exercise provocation tests were negative. However, the exercise provocation test after pork ingestion showed a positive reaction manifested by generalized urticaria, cough, mild dyspnea, and a 23% decreased peak expiratory flow rate. Three allergens to pork (67 kDa, 90 kDa, and 15 kDa) reacted with the patient's serum on immunoglobulin E immunoblotting. We report a case of pork-dependent exercise-induced anaphylaxis in a patient who was sensitive to pork.

A Case of Wheat-Dependent Exercise-Induced Anaphylaxis

Foods are recognized as a common cause of urticaria; however, the role of food is considered to be more important in acute not chronic urticaria. Wheat is a basic ingredient found in many common foods. Food allergy to wheat is primarily described in children in the form of atopic dermatitis. It is rare in adults; where it is mainly reported in exercise-induced anaphylaxis. We report a case of wheat dependent exercise-induced anaphylaxis that occurred in a 54-year-old Korean woman.

Wheat-dependent, Exercise-induced Anaphylaxis: A Successful Case of Prevention with Ketotifen

Food-dependent, exercise-induced anaphylaxis (FDEIA) is the triggering of anaphylaxis after ingestion of certain foods when followed by physical exercise. Symptoms vary from the typical generalized urticaria to severe allergic reactions. We report the case of a 20-year-old woman who had a 7-year history of recurrent wheals and dyspnea after ingesting several kinds of food (wheat, pork, and beef) along with physical exercise. Based on a provocation test, she was diagnosed with wheat-dependent, exercise-induced anaphylaxis. She was instructed to take 2 mg of ketotifen 2 hours before ingestion of wheat to prevent the symptoms, and subsequently the provocation test did not elicit wheals. We therefore prescribed ketotifen (1 mg twice a day). She has not had recurrent wheals or dyspnea for 6 months. We herein report an interesting case of wheat-dependent, exercise-induced anaphylaxis with successful prevention by ketotifen.

Three Cases of Food-dependent Exercise-induced Anaphylaxis / 대한내과학회지

Food-dependent exercise-induced anaphylaxis(FDEIAn), first reported in 1979, is a rare condition characterized by acute cutaneous, gastrointestinal, cardiovascular, and respiratory symptoms occurring on exertion after certain meals. This article describes 3 patients who developed urticaria, dizziness, abdominal pain, and loss of consciousness if the ingestion of wheat flour-food was followed by exercise within 2 hours. But either exercise or wheat intake alone did not induce any symptoms. Skin prick test showed strong positive responses to wheat flour and bread in all patients. We could diagnose FDEIAn with specific food-exercise challenges.