{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T03:58:16Z","timestamp":1790913496292,"version":"4.1.0"},"reference-count":15,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2025,5,20]],"date-time":"2025-05-20T00:00:00Z","timestamp":1747699200000},"content-version":"vor","delay-in-days":139,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2025,1,1]],"date-time":"2025-01-01T00:00:00Z","timestamp":1735689600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100013322","name":"European Federation of Pharmaceutical Industries and Associations","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100013322","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["JP 60749951"],"award-info":[{"award-number":["JP 60749951"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatrics International"],"published-print":{"date-parts":[[2025,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Hereditary angioedema (HAE) is a rare genetic disorder that causes recurrent edema and abdominal\/laryngeal attacks. However, the number of pediatric HAE cases is unknown. We aimed to assess the number of pediatric HAE and the actual status of pediatric HAE management in Japan.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A mail questionnaire survey was conducted on 142 clinicians (representatives of institutes) with previous experience in HAE management. The survey items included the number of pediatric patients who were treated for HAE, history of attacks and its impact on quality of life, the presence of untested pediatric relatives of adult patients, and unmet needs.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      In total, 69 representatives (49% of overall institutes) responded. Twenty\u2010five (36% of respondents) had experience in pediatric HAE management. The number of cases managed by individual faculties ranged from 1 to 6, and most physicians (\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u200916, 64% of faculties with patient(s)) reported the management of a single case only. There were 26 (8 male,16 female) patients with one or more attacks. Nineteen facilities (28% of respondents) reported one or more pediatric relative(s) of patients who were hesitant to screen. Further, physicians reported various unmet needs.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Pediatric HAE is managed in many facilities in a dispersed manner. In some cases, the pediatric relatives of patients diagnosed with HAE did not undergo screening. This study identified unmet needs and challenges that reflect the absence of specialized pediatric management. Hence, the standardization of pediatric HAE management is an urgent concern in Japan.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/ped.70077","type":"journal-article","created":{"date-parts":[[2025,5,20]],"date-time":"2025-05-20T06:39:55Z","timestamp":1747723195000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["A nationwide questionnaire\u2010based surveillance on pediatric hereditary angioedema in Japan"],"prefix":"10.1111","volume":"67","author":[{"given":"Beverley Anne","family":"Yamamoto","sequence":"first","affiliation":[{"name":"Graduate School of Human Sciences Osaka University  Osaka Japan"},{"name":"Registered NPO Hereditary Angioedema Japan (HAEJ)  Hyogo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isao","family":"Ohsawa","sequence":"additional","affiliation":[{"name":"Saiyu Souka Hospital  Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shun","family":"Toriumi","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine Juntendo University  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Makiko","family":"Matsuyama","sequence":"additional","affiliation":[{"name":"Registered NPO Hereditary Angioedema Japan (HAEJ)  Hyogo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takahiro","family":"Saito","sequence":"additional","affiliation":[{"name":"Office of Management and Planning Osaka University  Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0364-0022","authenticated-orcid":false,"given":"Toshiaki","family":"Shimizu","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine Juntendo University  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9442-3666","authenticated-orcid":false,"given":"Eisuke","family":"Inage","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine Juntendo University  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,5,20]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.waojou.2022.100627"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2012.05.055"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.alit.2020.09.008"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2016-0575"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.2500\/aap.2021.42.210019"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2015.12.985"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/ped.13729"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.waojou.2021.100511"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.anai.2015.03.010"},{"key":"e_1_2_8_11_1","volume-title":"Hereditary angioedema \u2010 intractable disorder","author":"Ohsawa I","year":"2016"},{"key":"e_1_2_8_12_1","unstructured":"CSL Behring GmbH Berinert(C1 Esterase Inhibitor (Human)) [package insert]. U.S. Food and Drug Administration website.https:\/\/www.fda.gov\/media\/77803\/download?attachment. Accessed 15 Feb 2024"},{"key":"e_1_2_8_13_1","unstructured":"Takeda Pharmaceutical Co.Approval of Partial Change to the Manufacturing and Marketing Approval Matters of Filazil\u00ae Subcutaneous Injection 30 mg Syringe for the Treatment of Hereditary Angioedema (HAE) in Pediatric Patients in Japan [Press release].https:\/\/www.takeda.com\/ja\u2010jp\/announcements\/2022\/hae\u20102. Accessed 15 Feb 2024."},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.alit.2023.02.002"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40265-021-01475-4"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/1346-8138.16909"}],"container-title":["Pediatrics International"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/ped.70077","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/ped.70077","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/ped.70077","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T03:49:36Z","timestamp":1790912976000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/ped.70077"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,1]]},"references-count":15,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2025,1]]}},"alternative-id":["10.1111\/ped.70077"],"URL":"https:\/\/doi.org\/10.1111\/ped.70077","archive":["Portico"],"relation":{},"ISSN":["1328-8067","1442-200X"],"issn-type":[{"value":"1328-8067","type":"print"},{"value":"1442-200X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,1]]},"assertion":[{"value":"2024-05-09","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-02-25","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-05-20","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70077"}}