Fetoscopic temporary tracheal occlusion for congenital diaphragmatic hernia: prelude to a randomized, controlled trial

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Abstract

Objective

As previously reported, high postnatal mortality seen in fetuses with congenital diaphragmatic hernia (CDH) with liver herniation and low lung-to-head ratio (LHR) appears to be improved in fetuses who undergo fetoscopic temporary tracheal occlusion (TO). To test whether further evolution of this technique produces results that justify a randomized controlled trial comparing prenatal intervention to postnatal care, the authors analyzed 11 additional cases and the cumulative experience with 19 cases.

Methods

The authors analyzed retrospectively the outcome of 11 new and 8 previously reported cases of fetoscopic temporary tracheal occlusion. Various factors were studied including maternal morbidity, antenatal outcome, physiologic lung response, and neonatal course.

Results

Temporary TO can be accomplished using 3 5-mm radially expanding uterine ports without hysterotomy. Obstetric morbidity included mild pulmonary edema in 6 cases, chorioamniotic separation and premature rupture of membranes in 12 patients, and preterm labor and delivery in all patients. Thirteen of 19 (68%) neonates survived for 90 days after delivery; one died in utero, and 5 died after birth. Late mortality included one death caused by sepsis and 2 by complications associated with tracheostomies. Morbidity from gastroesophageal reflux requiring Nissen fundoplication, tracheal injury requiring repair or tracheostomy, and recurrent hernias after diaphragmatic repair were characteristic in longterm survivors.

Conclusions

Fetoscopic temporary TO may improve outcome in poor-prognosis fetuses with CDH. However, complications related to tracheal dissection, premature delivery and late morbidity are significant. This experience has led to simpler techniques for fetoscopic tracheal occlusion and to an National Institutes of Health-sponsored randomized controlled trial comparing fetoscopic tracheal occlusion with optimal postnatal care.

Section snippets

Materials and methods

From January 1996 to April 1999, 19 fetuses underwent fetoscopic temporary tracheal occlusion (Fetendo clip) for congenital diaphragmatic hernia (CDH) at the University of California at San Francisco (UCSF) Fetal Treatment Center. We now update the previously reported experience with 8 left-sided CDHs3 with an additional 8 left-sided and 3 right-sided CDH patients.

Fetuses eligible for prenatal intervention met criteria including (1) diagnosis of CDH made before 25 weeks’ gestation, (2) a major

Operation

Although open hysterotomy was available as a back-up procedure if fetoscopic placement could not be accomplished for technical reasons (fetal position, visualization, bleeding), the last 16 cases (all new cases and the last 5 cases of the previous series) were accomplished without hysterotomy. Eleven of the 19 cases had the more favorable posterior placenta, whereas 8 had less-favorable anterior or fundal locations. Average operating time was 221 minutes ± 69 minutes. The first 8 cases averaged

Discussion

Two decades of extensive experimental investigation and agonizing and often frustrating clinical trials led to development of the prenatal strategy of temporary tracheal occlusion to enlarge the lungs of fetuses with the most severe form of CDH. Previous retrospective and prospective studies have found that without prenatal intervention, high-risk infants do poorly even with optimal postnatal care with mortality rates of 60% for LHRs less than 1.4 and nearly 100% for LHRs less than 1.0.1, 16

References (33)

Cited by (113)

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    Eventually, vascular occlusion balloons were chosen due to several advantages: avoided secondary surgical procedures, provided total tracheal occlusion while allowing tracheal growth, required a simpler technical approach, and minimized dysmorphism of the trachea. Eventually, with the advent of percutaneous fetoscopic access to the fetus, this strategy could be accomplished without hysterotomy and thus minimizing maternal risk.28 Although fetal tracheal occlusion demonstrated increased lung volume, ongoing pulmonary distention had shown significant decrease in the production of type 2 pneumocytes and surfactant production29–31 with an increase in the risk of PH in CDH-affected neonates after birth.32

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    Studies in animal models have revealed that changes to the tracheal architecture happens also at a microscopic level.129 Harrison et al.130 reported a small cohort of patients treated with prenatal TO who had stridor and vocal cord paralysis, two of whom also had tracheomalacia. Other authors did not find an association between the increase in airways’ width and short term131 or long-term tracheomegaly-related respiratory symptoms,126 and Cortes et al.24 found no difference in terms of 2 years outcome between CDH survivors who randomly underwent TO or not.

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