{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,22]],"date-time":"2026-06-22T16:17:19Z","timestamp":1782145039800,"version":"3.54.5"},"reference-count":5,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2023,1,26]],"date-time":"2023-01-26T00:00:00Z","timestamp":1674691200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Respirology Case Reports"],"published-print":{"date-parts":[[2023,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Alveolar capillary dysplasia with misalignment of the pulmonary veins (ACDMPV) is a rare congenital diffuse lung disorder, with a fatal course during the neonatal period. We describe an 18\u2010month\u2010old boy who presented with respiratory syncytial virus pneumonia and pulmonary hypertensive crisis requiring extracorporeal membrane oxygenation. Exome sequencing revealed a <jats:italic>FOXF1<\/jats:italic> frameshift variant, NM_001451.2:c.995_998delACTC, inherited from his asymptomatic mother. Genetic findings were compatible with histopathology findings from a lung biopsy. Based on the disease course, histopathology, and outcomes of this case, we believe ACDMPV should be considered a possibility in an infant presenting with hypoxemic respiratory failure, resistant pulmonary hypertension, and vasodilator\u2010induced pulmonary edema. Genetic testing can contribute to the diagnostic process.<\/jats:p>","DOI":"10.1002\/rcr2.1089","type":"journal-article","created":{"date-parts":[[2023,1,27]],"date-time":"2023-01-27T06:59:01Z","timestamp":1674802741000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Unusual presentation of alveolar capillary dysplasia with misalignment of the pulmonary veins in a child with respiratory syncytial virus pneumonia: A case report"],"prefix":"10.1002","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0441-8570","authenticated-orcid":false,"given":"Kantisa","family":"Sirianansopa","sequence":"first","affiliation":[{"name":"Division of Pulmonology and Critical Care Medicine, Department of Pediatrics, Faculty of Medicine Prince of Songkla University  Songkhla Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pharsai","family":"Prasertsan","sequence":"additional","affiliation":[{"name":"Division of Pulmonology and Critical Care Medicine, Department of Pediatrics, Faculty of Medicine Prince of Songkla University  Songkhla Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kanokpan","family":"Ruangnapa","sequence":"additional","affiliation":[{"name":"Division of Pulmonology and Critical Care Medicine, Department of Pediatrics, Faculty of Medicine Prince of Songkla University  Songkhla Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kantara","family":"Saelim","sequence":"additional","affiliation":[{"name":"Division of Pulmonology and Critical Care Medicine, Department of Pediatrics, Faculty of Medicine Prince of Songkla University  Songkhla Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9318-3974","authenticated-orcid":false,"given":"Phawin","family":"Kor\u2010anantakul","sequence":"additional","affiliation":[{"name":"Excellence Center for Genomics and Precision Medicine, King Chulalongkorn Memorial Hospital, Thai Red Cross Society, and Department of Pediatrics, Faculty of Medicine Chulalongkorn University  Bangkok Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,1,26]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.earlhumdev.2021.105323"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2017.10.026"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1136\/archdischild-2019-317121"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1177\/2045894018795143"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtcvs.2013.06.014"}],"container-title":["Respirology Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/rcr2.1089","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/rcr2.1089","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/rcr2.1089","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,19]],"date-time":"2023-08-19T18:04:32Z","timestamp":1692468272000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/rcr2.1089"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,1,26]]},"references-count":5,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2023,2]]}},"alternative-id":["10.1002\/rcr2.1089"],"URL":"https:\/\/doi.org\/10.1002\/rcr2.1089","archive":["Portico"],"relation":{},"ISSN":["2051-3380","2051-3380"],"issn-type":[{"value":"2051-3380","type":"print"},{"value":"2051-3380","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,1,26]]},"assertion":[{"value":"2022-11-10","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-01-09","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-01-26","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e01089"}}