{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,20]],"date-time":"2026-08-20T14:57:41Z","timestamp":1787237861298,"version":"build-2736575974"},"reference-count":15,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2019,12,3]],"date-time":"2019-12-03T00:00:00Z","timestamp":1575331200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Allergy Immunology"],"published-print":{"date-parts":[[2020,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Episodic angioedema with eosinophilia (EAE, Gleich syndrome) is a rare disease, consisting of recurrent angioedema with hypereosinophilia and frequent increased serum immunoglobulin M levels. Less than 100 patients have been reported, mainly adults, sometimes with underlying lymphocytic variant of hypereosinophilic syndrome (HES\n                      <jats:sub>L<\/jats:sub>\n                      ). The aim of this study was to identify and describe pediatric cases.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We performed a retrospective study of all pediatric cases of EAE referred within the French National Referral Center for Hypereosinophilic Syndrome (CEREO). Next, the PRISMA guidelines were applied in order to perform a systematic review (data sources: PubMed, Web of Science).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Among the two reported and 15 previously published cases of EAE occurring in children, the main clinical findings mimicked those of adults, including recurrent angioedema, hives, and weight gain. The median time between the first angioedema flare and the diagnosis of EAE was 5\u00a0years in published cases. Hypereosinophilia was constant, usually worsening with each attack, but seldom disappeared between flares. Total IgM serum levels were elevated in 16 patients. Four children had evidence of abnormal CD3\n                      <jats:sup>\u2010<\/jats:sup>\n                      CD4\n                      <jats:sup>+<\/jats:sup>\n                      T cells. First\u2010line therapy relied on oral corticosteroids in all patients, and further lines (used in five patients) included interferon\u2010\u03b1, methotrexate, and cyclosporin. Two children developed eosinophilic myocarditis during follow\u2010up.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Pediatricians should be aware that EAE is a diagnosis to consider in children. T\u2010cell immunophenotyping is warranted in this setting. Prognosis seems fair, yet eosinophil\u2010related organ damage may occur in patients with persistent eosinophilia.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/pai.13173","type":"journal-article","created":{"date-parts":[[2019,11,14]],"date-time":"2019-11-14T09:20:42Z","timestamp":1573723242000},"page":"297-302","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Episodic angioedema with eosinophilia (Gleich syndrome) in children: A clinical review"],"prefix":"10.1111","volume":"31","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0225-9046","authenticated-orcid":false,"given":"Val\u00e9rie","family":"Bertrand","sequence":"first","affiliation":[{"name":"Pediatric Unit Le Havre Hospital Le Havre France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olivia","family":"Boccara","sequence":"additional","affiliation":[{"name":"Department of Dermatology and Reference Center for Genodermatoses and Rare Skin Diseases (MAGEC) Universit\u00e9 Paris Descartes \u2010 Sorbonne Paris Cit\u00e9 Institut Imagine H\u00f4pital Universitaire Necker\u2010Enfants Malades APHP Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bruno","family":"Filhon","sequence":"additional","affiliation":[{"name":"Pediatric Unit Le Havre Hospital Le Havre France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Florian","family":"Manca","sequence":"additional","affiliation":[{"name":"Pediatric Unit Le Havre Hospital Le Havre France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guillaume","family":"Lef\u00e8vre","sequence":"additional","affiliation":[{"name":"National Referral Center for Hypereosinophilic Syndromes (CEREO) Suresnes France"},{"name":"D\u00e9partement de M\u00e9decine Interne et Immunologie Clinique Centre de R\u00e9f\u00e9rence des Maladies Auto\u2010immunes Syst\u00e9miques Rares du Nord et Nord\u2010Ouest de France (CeRAINO) CHU de Lille Universit\u00e9 de Lille Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthieu","family":"Groh","sequence":"additional","affiliation":[{"name":"National Referral Center for Hypereosinophilic Syndromes (CEREO) Suresnes France"},{"name":"Service de M\u00e9decin Interne H\u00f4pital Foch Universit\u00e9 Versailles\u2010Saint\u2010Quentin\u2010en\u2010Yvelines Suresnes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean\u2010Emmanuel","family":"Kahn","sequence":"additional","affiliation":[{"name":"National Referral Center for Hypereosinophilic Syndromes (CEREO) Suresnes France"},{"name":"Service de M\u00e9decine Interne H\u00f4pital Ambroise Par\u00e9 Universit\u00e9 Versailles\u2010Saint Quentin\u2010en\u2010Yvelines Boulogne\u2010Billancourt 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