Julio A Jiménez1, Elisenda Eixarch2, Philip DeKoninck3, João R Bennini4, Roland Devlieger3, Cleisson F Peralta5, Eduard Gratacos2, Jan Deprest6. 1. Department of Development and Regeneration, Group Biomedical Sciences, KU Leuven, Leuven, Belgium; Department of Obstetrics and Gynaecology, Clínica Alemana, Universidad del Desarrollo Santiago, Chile. 2. Fetal i+D Fetal Medicine Research Center, BCNatal-Barcelona Center for Maternal-Fetal and Neonatal Medicine (Hospital Clínic and Hospital Sant Joan de Deu), Institut d'Investigacions Biomediques August Pi i Sunyer, University of Barcelona, Centre for Biomedical Research on Rare Diseases, Barcelona, Spain. 3. Department of Development and Regeneration, Group Biomedical Sciences, KU Leuven, Leuven, Belgium; Division Woman and Child, Department of Obstetrics and Gynaecology, University Hospitals Leuven, Leuven, Belgium. 4. Department of Obstetrics and Gynecology, Faculty of Medical Sciences, State University of Campinas, Brazil. 5. Department of Obstetrics and Gynecology, Faculty of Medical Sciences, State University of Campinas, Brazil; Fetal Medicine Unit - The Heart Hospital, São Paulo, Brazil; Gestar Fetal Medicine and Surgery Center, São Paulo, Brazil. 6. Department of Development and Regeneration, Group Biomedical Sciences, KU Leuven, Leuven, Belgium; Division Woman and Child, Department of Obstetrics and Gynaecology, University Hospitals Leuven, Leuven, Belgium; UCL Institute for Women's Health (IWH), University College London, London, United Kingdom. Electronic address: jan.deprest@uzleuven.be.
Abstract
BACKGROUND: Isolated congenital diaphragmatic hernia defect allows viscera to herniate into the chest, competing for space with the developing lungs. At birth, pulmonary hypoplasia leads to respiratory insufficiency and persistent pulmonary hypertension that is lethal in up to 30% of patients. Antenatal measurement of lung size and liver herniation can predict survival after birth. Prenatal intervention aims at stimulating lung development, clinically achieved by percutaneous fetal endoscopic tracheal occlusion under local anesthesia. This in utero treatment requires a second intervention to reestablish the airway, either before birth or at delivery. OBJECTIVE: To describe our experience with in utero endotracheal balloon removal. MATERIALS AND METHODS: This is a retrospective analysis of prospectively collected data on consecutive patients with congenital diaphragmatic hernia treated in utero by fetal endoscopic tracheal occlusion from 3 centers. Maternal and pregnancy-associated variables were retrieved. Balloon removal attempts were categorized as elective or emergency and by technique (in utero: ultrasound-guided puncture; fetoscopy; ex utero: on placental circulation or postnatal tracheoscopy). RESULTS: We performed 351 balloon insertions during a 144-month period. In 9 cases removal was attempted outside fetal endoscopic tracheal occlusion centers, 3 of which were deemed impossible and led to neonatal death. We attempted 302 in-house balloon removals in 292 fetuses (217 elective [71.8%], 85 emergency [28.2%]) at 33.4 ± 0.1 weeks (range: 28.9-37.1), with a mean interval to delivery of 16.6 ± 0.8 days (0-85). Primary attempt was by fetoscopy in 196 (67.1%), by ultrasound-guided puncture in 62 (21.2%), by tracheoscopy on placental circulation in 30 (10.3%), and postnatal tracheoscopy in 4 cases (1.4%); a second attempt was required in 10 (3.4%) cases. Each center had different preferences for primary technique selection. In elective removals, we found no differences in the interval to delivery between fetoscopic and ultrasound-guided puncture removals. Difficulties during fetoscopic removal led to the development of a stylet to puncture the balloon, leading to shorter operating time and easier reestablishment of airways. CONCLUSION: In these fetal treatment centers, the balloon could always be removed successfully. In 90% this was in utero, with the use of fetoscopy preferred over ultrasound-guided puncture. Ex utero removal was a fall-back procedure. In utero removal does not seem to precipitate immediate membrane rupture, labor, or delivery, although the design of the study did not allow for a formal conclusion. For fetoscopic removals, the introduction of a stylet facilitated retrieval. Successful removal may rely on a permanently prepared team with expertise in all possible techniques.
BACKGROUND: Isolated congenital diaphragmatic hernia defect allows viscera to herniate into the chest, competing for space with the developing lungs. At birth, pulmonary hypoplasia leads to respiratory insufficiency and persistent pulmonary hypertension that is lethal in up to 30% of patients. Antenatal measurement of lung size and liver herniation can predict survival after birth. Prenatal intervention aims at stimulating lung development, clinically achieved by percutaneous fetal endoscopic tracheal occlusion under local anesthesia. This in utero treatment requires a second intervention to reestablish the airway, either before birth or at delivery. OBJECTIVE: To describe our experience with in utero endotracheal balloon removal. MATERIALS AND METHODS: This is a retrospective analysis of prospectively collected data on consecutive patients with congenital diaphragmatic hernia treated in utero by fetal endoscopic tracheal occlusion from 3 centers. Maternal and pregnancy-associated variables were retrieved. Balloon removal attempts were categorized as elective or emergency and by technique (in utero: ultrasound-guided puncture; fetoscopy; ex utero: on placental circulation or postnatal tracheoscopy). RESULTS: We performed 351 balloon insertions during a 144-month period. In 9 cases removal was attempted outside fetal endoscopic tracheal occlusion centers, 3 of which were deemed impossible and led to neonatal death. We attempted 302 in-house balloon removals in 292 fetuses (217 elective [71.8%], 85 emergency [28.2%]) at 33.4 ± 0.1 weeks (range: 28.9-37.1), with a mean interval to delivery of 16.6 ± 0.8 days (0-85). Primary attempt was by fetoscopy in 196 (67.1%), by ultrasound-guided puncture in 62 (21.2%), by tracheoscopy on placental circulation in 30 (10.3%), and postnatal tracheoscopy in 4 cases (1.4%); a second attempt was required in 10 (3.4%) cases. Each center had different preferences for primary technique selection. In elective removals, we found no differences in the interval to delivery between fetoscopic and ultrasound-guided puncture removals. Difficulties during fetoscopic removal led to the development of a stylet to puncture the balloon, leading to shorter operating time and easier reestablishment of airways. CONCLUSION: In these fetal treatment centers, the balloon could always be removed successfully. In 90% this was in utero, with the use of fetoscopy preferred over ultrasound-guided puncture. Ex utero removal was a fall-back procedure. In utero removal does not seem to precipitate immediate membrane rupture, labor, or delivery, although the design of the study did not allow for a formal conclusion. For fetoscopic removals, the introduction of a stylet facilitated retrieval. Successful removal may rely on a permanently prepared team with expertise in all possible techniques.
Authors: Ahmet A Baschat; Sean B Blackwell; Debnath Chatterjee; James J Cummings; Stephen P Emery; Shinjiro Hirose; Lisa M Hollier; Anthony Johnson; Sarah J Kilpatrick; Francois I Luks; M Kathryn Menard; Lawrence B McCullough; Julie S Moldenhauer; Anita J Moon-Grady; George B Mychaliska; Michael Narvey; Mary E Norton; Mark D Rollins; Eric D Skarsgard; KuoJen Tsao; Barbara B Warner; Abigail Wilpers; Greg Ryan Journal: Obstet Gynecol Date: 2022-05-02 Impact factor: 7.623
Authors: Francesca Maria Russo; Alexandra Benachi; Tim Van Mieghem; Jan De Hoon; Kristel Van Calsteren; Pieter Annaert; Jean-Marc Tréluyer; Karel Allegaert; Jan Deprest Journal: Trials Date: 2018-09-27 Impact factor: 2.279
Authors: Jan A Deprest; Kypros H Nicolaides; Alexandra Benachi; Eduard Gratacos; Greg Ryan; Nicola Persico; Haruhiko Sago; Anthony Johnson; Mirosław Wielgoś; Christoph Berg; Ben Van Calster; Francesca M Russo Journal: N Engl J Med Date: 2021-06-08 Impact factor: 176.079
Authors: Jan A Deprest; Alexandra Benachi; Eduard Gratacos; Kypros H Nicolaides; Christoph Berg; Nicola Persico; Michael Belfort; Glenn J Gardener; Yves Ville; Anthony Johnson; Francesco Morini; Mirosław Wielgoś; Ben Van Calster; Philip L J DeKoninck Journal: N Engl J Med Date: 2021-06-08 Impact factor: 176.079
Authors: Adalina Sacco; Lennart Van der Veeken; Emma Bagshaw; Catherine Ferguson; Tim Van Mieghem; Anna L David; Jan Deprest Journal: Prenat Diagn Date: 2019-02-27 Impact factor: 3.050
Authors: D Basurto; N Sananès; T Bleeser; I Valenzuela; N De Leon; L Joyeux; E Verbeken; S Vergote; L Van der Veeken; F M Russo; J Deprest Journal: Ultrasound Obstet Gynecol Date: 2021-01 Impact factor: 8.678
Authors: Lennart Van der Veeken; Francesca Maria Russo; Luc De Catte; Eduard Gratacos; Alexandra Benachi; Yves Ville; Kypros Nicolaides; Christoph Berg; Glenn Gardener; Nicola Persico; Pietro Bagolan; Greg Ryan; Michael A Belfort; Jan Deprest Journal: Gynecol Surg Date: 2018-05-08