Idiopathic Anaphylaxis in Infancy

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RATIONALE: Idiopathic anaphylaxis is a potentially life-threatening disease sharing the characteristics of anaphylaxis but lacking an identifiable cause. We present results of a literature search on pediatric idiopathic anaphylaxis and a case report of the youngest patient yet described with this condition.METHODS: OVID and PubMed were searched with the following terms: idiopathic anaphylaxis, idiopathic anaphylaxis infant, idiopathic anaphylaxis children, pediatric idiopathic anaphylaxis, pediatric anaphylaxis.RESULTS: A literature review of idiopathic anaphylaxis reveals that it is very infrequently observed in young children, with the youngest age reported as 11 months old. We were referred a 5-month-old female who developed anaphylaxis (severe swelling of the face, lips, tongue and hands as well as diffuse erythema, respiratory stridor and wheezing) at age 2 1/2 months. She responded to treatment with epinephrine and methylprednisolone. By the age of 5 months two similar episodes occurred, one requiring hospitalization. An extensive evaluation including CBC, C1 esterase inhibitor level, total IgE, baseline tryptase, serum specific IgE to milk, crab, fish, trout, flounder, codfish, salmon, and tuna (foods ingested by the mother) were all negative. Skin testing to other common foods and environmental allergens was also negative. The child was initially breastfed but switching to an elemental formula did not prevent recurrences. She was diagnosed with idiopathic anaphylaxis.CONCLUSIONS: Idiopathic anaphylaxis may occur at any age, even in early infancy. This is the youngest case yet reported. RATIONALE: Idiopathic anaphylaxis is a potentially life-threatening disease sharing the characteristics of anaphylaxis but lacking an identifiable cause. We present results of a literature search on pediatric idiopathic anaphylaxis and a case report of the youngest patient yet described with this condition. METHODS: OVID and PubMed were searched with the following terms: idiopathic anaphylaxis, idiopathic anaphylaxis infant, idiopathic anaphylaxis children, pediatric idiopathic anaphylaxis, pediatric anaphylaxis. RESULTS: A literature review of idiopathic anaphylaxis reveals that it is very infrequently observed in young children, with the youngest age reported as 11 months old. We were referred a 5-month-old female who developed anaphylaxis (severe swelling of the face, lips, tongue and hands as well as diffuse erythema, respiratory stridor and wheezing) at age 2 1/2 months. She responded to treatment with epinephrine and methylprednisolone. By the age of 5 months two similar episodes occurred, one requiring hospitalization. An extensive evaluation including CBC, C1 esterase inhibitor level, total IgE, baseline tryptase, serum specific IgE to milk, crab, fish, trout, flounder, codfish, salmon, and tuna (foods ingested by the mother) were all negative. Skin testing to other common foods and environmental allergens was also negative. The child was initially breastfed but switching to an elemental formula did not prevent recurrences. She was diagnosed with idiopathic anaphylaxis. CONCLUSIONS: Idiopathic anaphylaxis may occur at any age, even in early infancy. This is the youngest case yet reported.