{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,22]],"date-time":"2026-08-22T05:05:12Z","timestamp":1787375112056,"version":"build-2736575974"},"reference-count":17,"publisher":"American Medical Association (AMA)","issue":"21","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JAMA"],"published-print":{"date-parts":[[2023,12,5]]},"abstract":"<jats:sec><jats:title>Importance<\/jats:title><jats:p>Early anhydramnios during pregnancy, resulting from fetal bilateral renal agenesis, causes lethal pulmonary hypoplasia in neonates. Restoring amniotic fluid via serial amnioinfusions may promote lung development, enabling survival.<\/jats:p><\/jats:sec><jats:sec><jats:title>Objective<\/jats:title><jats:p>To assess neonatal outcomes of serial amnioinfusions initiated before 26 weeks\u2019 gestation to mitigate lethal pulmonary hypoplasia.<\/jats:p><\/jats:sec><jats:sec><jats:title>Design, Setting, and Participants<\/jats:title><jats:p>Prospective, nonrandomized clinical trial conducted at 9 US fetal therapy centers between December 2018 and July 2022. Outcomes are reported for 21 maternal-fetal pairs with confirmed anhydramnios due to isolated fetal bilateral renal agenesis without other identified congenital anomalies.<\/jats:p><\/jats:sec><jats:sec><jats:title>Exposure<\/jats:title><jats:p>Enrolled participants initiated ultrasound-guided percutaneous amnioinfusions of isotonic fluid before 26 weeks\u2019 gestation, with frequency of infusions individualized to maintain normal amniotic fluid levels for gestational age.<\/jats:p><\/jats:sec><jats:sec><jats:title>Main Outcomes and Measures<\/jats:title><jats:p>The primary end point was postnatal infant survival to 14 days of life or longer with dialysis access placement.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The trial was stopped early based on an interim analysis of 18 maternal-fetal pairs given concern about neonatal morbidity and mortality beyond the primary end point despite demonstration of the efficacy of the intervention. There were 17 live births (94%), with a median gestational age at delivery of 32 weeks, 4 days (IQR, 32-34 weeks). All participants delivered prior to 37 weeks\u2019 gestation. The primary outcome was achieved in 14 (82%) of 17 live-born infants (95% CI, 44%-99%). Factors associated with survival to the primary outcome included a higher number of amnioinfusions (<jats:italic>P<\/jats:italic>\u2009=\u2009.01), gestational age greater than 32 weeks (<jats:italic>P<\/jats:italic>\u2009=\u2009.005), and higher birth weight (<jats:italic>P<\/jats:italic>\u2009=\u2009.03). Only 6 (35%) of the 17 neonates born alive survived to hospital discharge while receiving peritoneal dialysis at a median age of 24 weeks of life (range, 12-32 weeks).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions and Relevance<\/jats:title><jats:p>Serial amnioinfusions mitigated lethal pulmonary hypoplasia but were associated with preterm delivery. The lower rate of survival to discharge highlights the additional mortality burden independent of lung function. Additional long-term data are needed to fully characterize the outcomes in surviving neonates and assess the morbidity and mortality burden.<\/jats:p><\/jats:sec><jats:sec><jats:title>Trial Registration<\/jats:title><jats:p>ClinicalTrials.gov Identifier: <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"https:\/\/www.clinicaltrials.gov\/study\/NCT03101891\">NCT03101891<\/jats:ext-link><\/jats:p><\/jats:sec>","DOI":"10.1001\/jama.2023.21153","type":"journal-article","created":{"date-parts":[[2023,12,5]],"date-time":"2023-12-05T16:30:02Z","timestamp":1701793802000},"page":"2096","source":"Crossref","is-referenced-by-count":65,"title":["Neonatal Survival After Serial Amnioinfusions for Bilateral Renal Agenesis"],"prefix":"10.1001","volume":"330","author":[{"given":"Jena L.","family":"Miller","sequence":"first","affiliation":[{"name":"Center for Fetal Therapy, Department of Gynecology and Obstetrics, Johns Hopkins University, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ahmet A.","family":"Baschat","sequence":"additional","affiliation":[{"name":"Center for Fetal Therapy, Department of Gynecology and Obstetrics, Johns Hopkins University, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mara","family":"Rosner","sequence":"additional","affiliation":[{"name":"Center for Fetal Therapy, Department of Gynecology and Obstetrics, Johns Hopkins University, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yair J.","family":"Blumenfeld","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Stanford University School of Medicine, Stanford, California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julie S.","family":"Moldenhauer","sequence":"additional","affiliation":[{"name":"Center for Fetal Diagnosis and Treatment, Children\u2019s Hospital of Philadelphia, Philadelphia, Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anthony","family":"Johnson","sequence":"additional","affiliation":[{"name":"The Fetal Center, Department of Obstetrics and Gynecology, University of Texas Health Center, Houston"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mauro H.","family":"Schenone","sequence":"additional","affiliation":[{"name":"Division of Maternal and Fetal Medicine, Department of Obstetrics and Gynecology, Mayo Clinic, Rochester, Minnesota"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael V.","family":"Zaretsky","sequence":"additional","affiliation":[{"name":"Colorado Fetal Care Center, Children\u2019s Hospital of Colorado, Aurora"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ramen H.","family":"Chmait","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Keck School of Medicine, University of Southern California, Los Angeles"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juan M.","family":"Gonzalez","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, University of California, San Francisco"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Russell S.","family":"Miller","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Columbia University Irving Medical Center, New York, New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anita J.","family":"Moon-Grady","sequence":"additional","affiliation":[{"name":"Division of Cardiology, Department of Pediatrics, University of California, San Francisco"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ellen","family":"Bendel-Stenzel","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Department of Pediatrics and Adolescent Medicine, Mayo Clinic, Rochester, 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University, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel S.","family":"Warren","sequence":"additional","affiliation":[{"name":"Department of Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel F.","family":"Hanley","sequence":"additional","affiliation":[{"name":"Division of Brain Injury Outcomes, Johns Hopkins University, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joslynn A.","family":"Watkins","sequence":"additional","affiliation":[{"name":"Division of Pediatric Nephrology, Johns Hopkins University School of Medicine, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joshua","family":"Samuels","sequence":"additional","affiliation":[{"name":"Division of Pediatric Nephrology and Hypertension, McGovern School at the University of Texas Health Science Center, 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