{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,31]],"date-time":"2026-07-31T05:57:52Z","timestamp":1785477472448,"version":"3.56.0"},"reference-count":45,"publisher":"Springer Science and Business Media LLC","license":[{"start":{"date-parts":[[2026,6,29]],"date-time":"2026-06-29T00:00:00Z","timestamp":1782691200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,6,29]],"date-time":"2026-06-29T00:00:00Z","timestamp":1782691200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Pediatr Res"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Alveolar capillary dysplasia with misalignment of pulmonary veins (ACD\/MPV) is a rare developmental lung disorder associated with high neonatal mortality. We aimed to describe the epidemiology, resource utilization, and outcomes of neonates with ACD\/MPV.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Retrospective observational study using the Pediatric Health Information System database. ICD-10 diagnosis codes were used to identify neonates hospitalized with ACD\/MPV between 2016 and 2025. Demographic characteristics, associated congenital anomalies, resource utilization, length of stay, and mortality were analyzed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>We identified 58 neonates with ACD\/MPV during the study period. The demographic distribution includes 51.7% female sex, 60.3% Whites, and 28.4% Hispanics or Latinos. Median gestational age was 38 weeks with a birthweight of 2,883 grams. Cytogenetic testing was documented in 43.1% and surgical tissue biopsy in 53.3% of the neonates. Congenital anomalies were present in 91%, with cardiovascular malformations predominating. Invasive mechanical ventilation (IMV) was used in 93.1%, and extracorporeal membrane oxygenation (ECMO) in 63.8%. ECMO utilization was more common among non-survivors (72.0 vs. 12.5%). Mortality was 86.2%.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>ACD\/MPV is a rare neonatal disorder characterized by high mortality and substantial resource utilization. Congenital malformations are frequent, with cardiovascular anomalies predominating. ACD\/MPV should be considered in neonates with refractory pulmonary hypertension.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Impact<\/jats:title>\n                    <jats:p>\n                      <jats:list list-type=\"bullet\">\n                        <jats:list-item>\n                          <jats:p>This is the largest multicenter study in the U.S to date exploring the epidemiology, resource utilization, and outcomes of neonates with alveolar capillary dysplasia with misalignment of pulmonary veins (ACD\/MPV).<\/jats:p>\n                        <\/jats:list-item>\n                        <jats:list-item>\n                          <jats:p>ACD\/MPV is a rare neonatal disorder and carries a very high mortality (86%) despite advanced therapies.<\/jats:p>\n                        <\/jats:list-item>\n                        <jats:list-item>\n                          <jats:p>ACD\/MPV is frequently associated with congenital malformations, which were present in over 91% of the cases, with cardiovascular anomalies predominating.<\/jats:p>\n                        <\/jats:list-item>\n                        <jats:list-item>\n                          <jats:p>ACD\/MPV should be suspected in neonates with prolonged or refractory pulmonary hypertension, and early diagnostic evaluation, like genetic testing or lung biopsy, should be conducted to avoid prolonged futile interventions.<\/jats:p>\n                        <\/jats:list-item>\n                      <\/jats:list>\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1038\/s41390-026-05265-0","type":"journal-article","created":{"date-parts":[[2026,6,29]],"date-time":"2026-06-29T10:45:43Z","timestamp":1782729943000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Alveolar capillary dysplasia with misalignment of pulmonary veins in neonates: a multicenter cohort study"],"prefix":"10.1038","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1970-1507","authenticated-orcid":false,"given":"Prithvi","family":"Sendi","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"Martinez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kaitlin","family":"Kobaitri","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Balagangadhar R.","family":"Totapally","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,6,29]]},"reference":[{"key":"5265_CR1","first-page":"919","volume":"24","author":"MH Mac","year":"1948","unstructured":"Mac, M. 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