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PubMed2004

Recent advances in fetal surgery.

Fetal surgery is now an accepted modality for treatment of a variety of lethal and non-lethal congenital conditions. It represents a new, fast-moving frontier of medicine in which cooperative mulitdisciplinary effort and input are required to assure both fetal and maternal welfare. A wide range of therapeutic strategies from percutaneous to open invasive techniques has led to a complex list of different procedures for different diseases. This review identifies the most common disease entities managed by fetal intervention, examines the evolution in development of techniques to those currently used, and describes the prospective, randomized trials presently underway that are designed to establish the safety and determine true efficacy of treatment. Fetal surgery as a (multi)discipline continues to strive to minimize maternal and fetal risk. Undoubtedly, as tocolytic therapy and neonatal intensive efforts improve, fetal therapy will expand.

Bronchopulmonary Sequestration↗
PubMed2005

Fetal intervention for congenital diaphragmatic hernia: the European experience.

Fetuses with CDH presenting with liver herniation and a lung area-to-head circumference ratio of less than 1.0 have a high chance for neonatal death due to pulmonary hypoplasia. Fetal tracheal occlusion (TO) prevents egress of lung liquid, which triggers lung growth. In animal experiments, we were able to develop a minimally invasive technique for Fetoscopic Endoluminal Tracheal Occlusion (FETO) with a detachable balloon. In 2001, we demonstrated feasibility of FETO by percutaneous access in fetuses with severe CDH. In a retrospective multicenter review, we obtained LHR measurements and position of the liver in 134 cases of isolated left-sided CDH between 24 and 28 weeks. Eleven patients (8%) with LHR < 1.4 opted for termination. Overall survival of liveborn babies was 47% (58/123). LHR and position of the liver correlated both to survival. Combination of both variables predicted neonatal outcome better: liver up and LHR < 1.0 predicted a survival of 9%. When LHR < 0.6, there were no survivors irrespective of liver position. We could successfully perform endotracheal placement of the balloon in 20 cases at a median gestational age of 26 weeks. The mean duration of the operation was 22 (range 5-54) minutes. In 11 (55%) of these patients, there was postoperative prelabor (ie, <37 weeks) amniorrhexis. Membranes ruptured before 32 weeks in 35%, with a decreasing trend as experience increased. Ultrasound scans after FETO demonstrated an increase in the echogenicity of the lungs within 48 hours and improvement in the LHR from a median 0.7 (range 0.4-0.9) before FETO to 1.8 (range 1.1-2.9) within 2 weeks after surgery. The median gestation at delivery was 33.2 (range 27-38) weeks, and in 14 (70%) this occurred after 32 weeks. Surgical repair of the diaphragmatic hernia could be done in 13 babies, and in all but 1 the defect was extensive and required the insertion of a patch. Survival to discharge was 50%. These 10 long-term surviving babies are now aged 7 to 26 (median 19) months without known neurologic morbidity. Eight babies died in the neonatal period due to complications of the underlying disease. Two nonsurvivors died from other causes but with appropriately developed lungs. Improved survival coincided with increasing experience, in turn related to reduced incidence of postoperative amniorrhexis, later delivery, and a change in the policy on the timing of removal of the balloon from intrapartum to the prenatal period. Survival in eligible contemporary controls was 1/12 (8%). The presence of liver herniation and a low lung-to-head ratio (LHR <1.0) is a good predictor of poor prognosis at different tertiary centers around the world. Severe CDH may be successfully treated with FETO, which is minimally invasive and may improve postnatal survival.

Balloon Occlusion↗
PubMed2005

Fetal surgery for congenital diaphragmatic hernia: the North American experience.

The role for fetal surgery in treating fetuses with congenital diaphragmatic hernia (CDH) is unclear. Two decades of investigation have improved our understanding of the prenatal natural history, pathophysiology, and outcomes of these patients. During this same period, there have been advances in fetal surgery techniques including improvements in fetal monitoring, maternal-fetal anesthesia, tocolysis, and improved instrumentation to permit increased application of videoscopic approaches. Because of technical challenges, open fetal repair of CDH has been abandoned. Fetal tracheal ligation has shown promise, but a recently published prospective, randomized trial failed to show a benefit of fetoscopic tracheal ligation compared with expert postnatal treatment. Although there is evidence that postnatal outcomes for infants with this disease have improved with the adoption of gentilation ventilator management, high-frequency ventilation, and ECMO, there continues to be a subset of infants with severe CDH that die or suffer serious long-term morbidity despite advanced surgical care. The purpose of this article is to review issues related to prenatal diagnosis, patient selection, and outcomes for fetal surgery; and ultimately to assess whether there is a role for fetal surgery in treating fetuses with CDH.

Balloon Occlusion↗
PubMed2002

Fetoscopic treatment for discordant twins.

BACKGROUND/PURPOSE: In rare instances in monochorionic twin pregnancies, one twin can have a discordant anomaly (eg, cystic hygroma). If this twin dies in utero, neurologic injury and death can occur in the surviving cotwin. To protect the normal twin, the authors developed an approach to separate the circulations and ablate the umbilical cord of the abnormal twin. METHODS: From September 1998 to February 2001, 6 cases of discordant anomalous twins were diagnosed by prenatal ultrasound scan in which the anomaly was lethal or parents desired prenatal termination for this abnormal twin. All underwent surgical intervention with gestational ages varying from 19 to 24 weeks. RESULTS: Depending on cord insertion site and placental anatomy, blood flow was interrupted to the anomalous fetus by either radiofrequency ablation (RFA; 2 cases), cord transection (1 case), or cord transection after laser ablation of communicating vessels (3 cases). Fetal death occurred in one normal twin 4 days postoperatively. Average age at delivery for the 5 surviving fetuses was 34.5 weeks' gestation. On follow-up, all surviving infants are neurologically intact. CONCLUSION: An otherwise normal monochorionic twin threatened by an anomalous cotwin can be salvaged successfully with a strategy tailored to interrupt the vascular connections between the 2 twins.

Abnormalities, Multiple↗
PubMed2003

Evolution of a percutaneous fetoscopic access system for single-port tracheal occlusion.

BACKGROUND/PURPOSE: Prenatal tracheal occlusion currently is being assessed as a treatment modality for congenital diaphragmatic hernia (CDH). The development of a totally percutaneous fetoscopic access system would help avoid the need for maternal laparotomy and reduce the morbidity rate of fetal surgical procedures for the mother. Laparoscopic radial expansion sheaths and Seldinger technique-based vascular catheters both have been advocated as means of achieving amniotic cavity access. The authors have investigated these 2 systems in an attempt to develop a reliable method for achieving safe percutaneous fetoscopic access and present the first successful attempt to deploy an intratracheal balloon using an entirely percutaneous approach through a single port in an ovine model. METHODS: A number of prototype systems were evaluated sequentially over a 3-year period in an ovine model: (1) the radially expanding InnerDyne step port system, (2) a new rigid cannula with a bulbous/sharp end preloaded onto the radially expanding InnerDyne port, (3) a conical removable addition to the rigid cannula in 2, (4) a modified bulbous/sharp ended cannula incorporating a circumferential protective insert, (5) a rigid split sheath with the radially expanding port placed through the lumen of the split sheath, (6) a flexible introducer and dilator with the split sheath (used in the Seldinger placement of central lines), and (7) a 2-needle approach using a superelastic shape-memory alloy Nickel-Titanium wire with the flexible dilator and sheath, incorporating a side perfusion port. For balloon tracheal occlusion, live anaesthetized time-mated pregnant ewes were used at 110 days' gestation. Tracheobronchoscopy was achieved using a 3-mm 0 degrees telescope, and the cutaneotracheal tract was secured by a 3.3-mm sheath incorporating a side-perfusion port. The rigid telescope was replaced by a flexible choledochoscope preloaded with a silicone balloon. The balloon was deployed 2 cm above the carina proximal to the right upper lobe bronchus. RESULTS: The many problems encountered in the evolution of the preferred system related mainly to separation and tenting of the chorioamniotic membranes in the ovine uterus and inconsistent access to the fetal parts of interest. Each resulted in significant modifications to our approach. Furthermore, the use of rigid access devices commonly caused fetal injury. Successful access to the intrauterine cavity and cannulation of the trachea was achieved consistently with minimal trauma, irrespective of fetal position by method 7. Multiple port placement allowed visualization of the entry of all components of the system confirming minimal chorioamniotic membrane separation and tenting. Single port tracheal occlusion was undertaken first on 6 cadavers before being performed successfully on 3 live anaesthetized ewes. Fetoscopic access and cannulation of the trachea was achieved consistently in all live animals irrespective of fetal position. CONCLUSIONS: This modified Seldinger technique using the unique properties of the memory-shaped alloy wire for initial uterine access offers a safe method for the percutaneous placement of fetoscopic ports in the ovine model for prenatal intervention. Successful placement of a tracheal balloon entirely through a single percutaneously placed port represents a further advance in prenatal therapy for CDH.

Animals↗
PubMedDate not supplied

[Ultrasound and endoscopic image of exencephaly (acrania) in the 12th week of pregnancy].

BACKGROUND: Acrania is a rare congenital anomaly of unknown pathogenesis, in which the flat bones of the cranial vault are absent and the facial bones are normally present. MATERIALS AND METHODS: We present a case of a 4th gravida, who had already born 2 children with acrania and came for an ultrasound examination in the 12th week of gestation. RESULTS: By transvaginal ultrasonography it was possible to diagnose fetal acrania already in the 12th week. By hysteroscopy at the same time we could see, that the brain was covered by a thin membrane, the facial bone and the shape of the cranium were normal. CONCLUSIONS: By hysteroscopy it was possible to confirm the diagnosis and to detect additional malformations. This fetus showed a small omphalocele, hexadactylism on both sides and clubfeet on both sides. DISCUSSION: By hysteroscopy in this early week of pregnancy we could demonstrate, that acrania is an independent anomaly and not the first stage of an anencephaly as reported by other authors. Hysteroscopy also offers the advantage of viewing the intact fetus before pregnancy termination.

Abnormalities, Multiple↗
PubMedDate not supplied

Amniotic fluid index: correlation with amniotic fluid volume.

We calculated the amniotic fluid indexes (AFIs) of 310 women on 459 occasions. Normative data were analyzed and compared with data in several high-risk groups. In the normal gestations there was a progressive increase in AFI with advancing gestation until 32 weeks, after which there was a decline. The mean AFIs in abnormal gestations varied with the clinical diagnoses. These values were compared to those obtained by assessing amniotic fluid volume (AFV), that is a pocket more than 2 cm. There were 51 patients with abnormal AFVs. Forty-two had decreased fluid, six also had decreased AFIs; nine had increased AFVs and five (all with diabetes) also had increased AFIs. Thus, AFIs in normal pregnancies showed an orderly pattern of change with gestational age, and there was no accurate correlation between AFI and AFV. Thus, using AFV alone may lead to false interpretations of amniotic fluid status.

Amniotic Fluid↗
PubMed2000

Fetal hydrolaparoscopy and endoscopic cystotomy in complicated cases of lower urinary tract obstruction.

OBJECTIVE: Vesicoamniotic shunting may be difficult or impossible in selected cases of fetal lower obstructive uropathy. The purpose of this article is to describe the performance of fetal hydrolaparoscopy and endoscopic fetal cystotomy in two fetuses with complicated lower obstructive uropathy. STUDY DESIGN: Fetal hydrolaparoscopy-endoscopic fetal cystotomy was performed in a patient with a markedly thickened bladder that could not be entered percutaneously. A peritoneoamniotic (bridge) shunt was also placed. Fetal hydrolaparoscopy-endoscopic fetal cystotomy was performed in a second patient with a collapsed bladder from a previous vesicocentesis, because vesicoinfusion resulted in further ascites. Fetal cystoscopy was performed after endoscopic fetal cystotomy, and posterior urethral valves were ablated with neodymium:yttrium-aluminum-garnet laser energy. A vesicoamniotic shunt was left in place. RESULTS: Adequate bladder drainage was obtained in both cases. The first baby required bilateral nephrotomy and a permanent cystotomy at birth and is scheduled for a bladder expansion procedure at the age of year. The second patient had premature rupture of membranes and fetal death from treatment of this complication 5 days after the original procedure. CONCLUSION: Fetal hydrolaparoscopy-endoscopic fetal cystotomy can be performed in complicated cases of lower obstructive uropathy. The procedure involves the creation of a defect in the bladder dome under direct endoscopic visualization within a spontaneous or intentional hydroperitoneum. Peritoneoamniotic shunting, vesicoamniotic shunting, or ablation of posterior urethral valves may then be performed. Fetal hydrolaparoscopy-endoscopic fetal cystotomy should be reserved only for complicated cases of lower obstructive uropathy in which conventional vesicoamniotic shunting is not safely possible. Further experience with fetal hydrolaparoscopy-endoscopic fetal cystotomy is necessary to establish its risks and benefits.

Adult↗
PubMed2001

Fetoscopic temporary tracheal occlusion by means of detachable balloon for congenital diaphragmatic hernia.

Occlusion of the fetal trachea blocks the egress of fetal lung fluid and stimulates the growth of hypoplastic lungs in fetuses with diaphragmatic hernia. Accomplishing temporary and reversible occlusion of the fetal trachea has proven difficult without invasive fetal surgery. Using simultaneous real-time ultrasonography and fetal bronchoscopy through a single uterine port, we placed a detachable balloon in the trachea of 2 fetuses with severe diaphragmatic hernia. In both fetuses the fetal lung subsequently enlarged, allowing survival after birth.

Adult↗