Pediatric idiopathic anaphylaxis: Experience with 22 patients

Ditto, Anne Marie; Krasnick, Jane; Greenberger, Paul A.; Kelly, Kevin J.; McGrath, Kris G.; Patterson, Roy

doi:10.1016/s0091-6749(97)70244-6

Cited by 32 publications

(10 citation statements)

References 20 publications

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“…A higher incidence (60%) in women has been described, although the real overall incidence is hard to estimate due to limitations of data reported by old literature . On the other hand, IA seems to be less common in children . Patients with IA were found to have a high incidence of atopy, as high as 58% in one series…”

Section: Epidemiologymentioning

confidence: 90%

Idiopathic anaphylaxis

Bilò

Martini

Tontini

et al. 2019

Clin Experimental Allergy

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Idiopathic anaphylaxis (IA) or spontaneous anaphylaxis is a diagnosis of exclusion when no cause can be identified. The exact incidence and prevalence of IA are not known. The clinical manifestations of IA are similar to other known causes of anaphylaxis. A typical attack is usually acute in onset and can worsen over minutes to a few hours. The pathophysiology of IA has not yet been fully elucidated, although an IgE‐mediated pathway by hitherto unidentified trigger/s might be the main underlying mechanism. Elevated concentrations of urinary histamine and its metabolite, methylimidazole acetic acid, plasma histamine and serum tryptase have been reported, consistent with mast cell activation. There is some evidence that corticosteroids reduce the frequency and severity of episodes of IA, consistent with a steroid‐responsive condition. Important differential diagnoses of IA include galactose alpha‐1,3 galactose (a carbohydrate contained in red meat) allergy, pigeon tick bite (Argax reflexus), wheat‐dependent exercise‐induced anaphylaxis, Anisakis simplex allergy and mast cell disorders. Other differential diagnoses include “allergy‐mimics” such as asthma masquerading as anaphylaxis, undifferentiated somatoform disorder, panic attacks, globus hystericus, vocal cord dysfunction, scombroid poisoning, vasoactive amine intolerance, carcinoid syndrome and phaeochromocytoma. Acute treatment of IA is the same as for other forms of anaphylaxis. Long‐term management is individualized and dictated by frequency and severity of symptoms and involves treatment with H1 and H2 receptor blockers, leukotriene receptor antagonist and consideration for prolonged reducing courses of oral corticosteroids. Patients should possess an epinephrine autoinjector with an anaphylaxis self‐management plan. There are anecdotal reports regarding the use of omalizumab. For reasons that remain unclear, the prognosis of IA is generally favourable with appropriate treatment and patient education. If remission cannot be achieved, the diagnosis should be reconsidered.

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Section: Epidemiologymentioning

confidence: 90%

Idiopathic anaphylaxis

Bilò

Martini

Tontini

et al. 2019

Clin Experimental Allergy

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“…In a cohort of 175 patients with idiopathic anaphylaxis reported in 1990, the most common symptoms were urticaria or angioedema (100%), upper airway obstruction (62%), bronchospasm (31%), syncope (24%), hypotension (25%), gastrointestinal tract symptoms (17%) and flushing (16%) [22]. Idiopathic anaphylaxis may also occur in a pediatric population, with symptoms and severity similar to that in adults [23], and recurrent anaphylaxis has been a presenting feature in some cases of pediatric mastocytosis [24]. …”

Section: Discussionmentioning

confidence: 99%

Prevention of Mast Cell Activation Disorder-Associated Clinical Sequelae of Excessive Prostaglandin D<sub>2</sub> Production

Butterfield

Weiler²

2008

Int Arch Allergy Immunol

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Background: Patients with systemic mastocytosis have increased numbers of mast cells in the bone marrow and other organs, such as the liver, spleen, gastrointestinal tract and skin. Symptoms result from the local and remote effects of mediator release from mast cells and from the local effects of increased mast cell numbers in various organs. Patients with mast cell activation experience many of the same clinical symptoms as do patients with systemic mastocytosis from chronic or spontaneous release of mast cell mediators. We report 4 patients with mast cell activation symptoms from selective release of prostaglandin (PG) D₂, but not histamine, and their improvement with aspirin therapy. Methods: Bone marrow biopsy specimens obtained from 4 patients with symptoms suggestive of mastocytosis were examined by tryptase immunostaining. Baseline levels of serum tryptase and urinary 11β-PGF₂_α and N-methylhistamine were obtained. In 2 of the 4 patients, urinary 11β-PGF₂_α and N-methylhistamine samples were also measured during acute symptoms. Results: Baseline increase in urinary excretion of the PGD₂ metabolite 11β-PGF₂_α was found in 2 patients. In the remaining 2 patients, baseline levels of urinary 11β-PGF₂_α and N-methylhistamine were normal, but during acute symptoms, the excretion of 11β-PGF₂_α increased markedly. Treatment with aspirin resulted in normalization of 11β-PGF₂_α excretion in the 2 patients with elevated baseline levels and in prevention of symptoms in all 4 patients. Conclusions: These results suggest that mast cell activation may be manifested by a selective excessive release of PGD₂. These patients respond to administration of aspirin but not to antihistamines.

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“…In addition, the classification and treatment algorithm previously applied in the adult population are used in the pediatric population. 13,14 In a retrospective review of medical records of children presenting to a pediatric allergy clinic, 8 children diagnosed with IA were identified. The patients' ages ranged from 11 months to 19 years at disease onset.…”

Section: Epidemiologymentioning

confidence: 99%

“…12 In studies from Northwestern University, the overwhelming majority of patients achieve remission. 13,19,23 If a remission cannot be achieved, other diagnoses should be reconsidered. Recurrence of anaphylaxis in idiopathic anaphylactic patients does occur.…”

Section: Prognosismentioning

confidence: 99%

Idiopathic Anaphylaxis

Fenny

Grammer

2015

Immunology and Allergy Clinics of North America

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Idiopathic anaphylaxis is a diagnosis of exclusion after other causes have been thoroughly evaluated and excluded. The pathogenesis of idiopathic anaphylaxis remains uncertain, although increased numbers of activated lymphocytes and circulating histamine-releasing factors have been implicated. Signs and symptoms of patients diagnosed with idiopathic anaphylaxis are indistinguishable from the manifestations of other forms of anaphylaxis. Treatment regimens are implemented based on the frequency and severity of patient symptoms and generally include the use of epinephrine autoinjectors, antihistamines, and steroids. The prognosis of idiopathic anaphylaxis is generally favorable with well-established treatment regimens and effective patient education.

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Pediatric idiopathic anaphylaxis: Experience with 22 patients

Cited by 32 publications

References 20 publications

Idiopathic anaphylaxis

Idiopathic anaphylaxis

Prevention of Mast Cell Activation Disorder-Associated Clinical Sequelae of Excessive Prostaglandin D<sub>2</sub> Production

Idiopathic Anaphylaxis

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