Neonatal Survival After Serial Amnioinfusions for Bilateral Renal Agenesis: The Renal Anhydramnios Fetal Therapy Trial.

Autor:	Miller JL; Center for Fetal Therapy, Department of Gynecology and Obstetrics, Johns Hopkins University, Baltimore, Maryland., Baschat AA; Center for Fetal Therapy, Department of Gynecology and Obstetrics, Johns Hopkins University, Baltimore, Maryland., Rosner M; Center for Fetal Therapy, Department of Gynecology and Obstetrics, Johns Hopkins University, Baltimore, Maryland., Blumenfeld YJ; Department of Obstetrics and Gynecology, Stanford University School of Medicine, Stanford, California., Moldenhauer JS; Center for Fetal Diagnosis and Treatment, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania., Johnson A; The Fetal Center, Department of Obstetrics and Gynecology, University of Texas Health Center, Houston., Schenone MH; Division of Maternal and Fetal Medicine, Department of Obstetrics and Gynecology, Mayo Clinic, Rochester, Minnesota., Zaretsky MV; Colorado Fetal Care Center, Children's Hospital of Colorado, Aurora., Chmait RH; Department of Obstetrics and Gynecology, Keck School of Medicine, University of Southern California, Los Angeles., Gonzalez JM; Department of Obstetrics and Gynecology, University of California, San Francisco., Miller RS; Department of Obstetrics and Gynecology, Columbia University Irving Medical Center, New York, New York., Moon-Grady AJ; Division of Cardiology, Department of Pediatrics, University of California, San Francisco., Bendel-Stenzel E; Division of Neonatology, Department of Pediatrics and Adolescent Medicine, Mayo Clinic, Rochester, Minnesota., Keiser AM; Division of Neonatology, Department of Pediatrics, Johns Hopkins University, Baltimore, Maryland., Avadhani R; Division of Brain Injury Outcomes, Johns Hopkins University, Baltimore, Maryland., Jelin AC; Division of Maternal-Fetal Medicine, Department of Gynecology and Obstetrics, Johns Hopkins University, Baltimore, Maryland., Davis JM; Tufts Clinical and Translational Science Institute, Division of Newborn Medicine, Tufts Children's Hospital, Tufts University, Boston, Massachusetts., Warren DS; Department of Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland., Hanley DF; Division of Brain Injury Outcomes, Johns Hopkins University, Baltimore, Maryland., Watkins JA; Division of Pediatric Nephrology, Johns Hopkins University School of Medicine, Baltimore, Maryland., Samuels J; Division of Pediatric Nephrology and Hypertension, McGovern School at the University of Texas Health Science Center, Houston., Sugarman J; Berman Institute of Bioethics, Johns Hopkins University School of Medicine, Baltimore, Maryland., Atkinson MA; Division of Pediatric Nephrology, Johns Hopkins University School of Medicine, Baltimore, Maryland.
Jazyk:	angličtina
Zdroj:	JAMA [JAMA] 2023 Dec 05; Vol. 330 (21), pp. 2096-2105.
DOI:	10.1001/jama.2023.21153
Abstrakt:	Importance: 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. Objective: To assess neonatal outcomes of serial amnioinfusions initiated before 26 weeks' gestation to mitigate lethal pulmonary hypoplasia. Design, Setting, and Participants: 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. Exposure: Enrolled participants initiated ultrasound-guided percutaneous amnioinfusions of isotonic fluid before 26 weeks' gestation, with frequency of infusions individualized to maintain normal amniotic fluid levels for gestational age. Main Outcomes and Measures: The primary end point was postnatal infant survival to 14 days of life or longer with dialysis access placement. Results: 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' 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 (P = .01), gestational age greater than 32 weeks (P = .005), and higher birth weight (P = .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). Conclusions and Relevance: 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. Trial Registration: ClinicalTrials.gov Identifier: NCT03101891.
Databáze:	MEDLINE
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