A cystic mass at the base of the fetal tongue diagnosed antenatally is uncommon, relegated to case reports in the literature. Dr. Hartnick et al. [
Fetal airway obstruction at delivery is a potentially fatal complication. If a fetal oropharyngeal anomaly is noted antepartum, the neonatal airway can be secured intrapartum using the EXIT procedure, thus reducing potential neonatal morbidity and mortality [
We report a case of a pregnancy complicated by a fetal oropharyngeal cyst and successfully securing the fetal airway using the EXIT procedure. This report demonstrates the effectiveness of this procedure in securing the fetal airway while maintaining the fetoplacental circulation and adequate fetal oxygenation.
At our institution, an IRB approval is not required for a case report.
A 26-year-old primigravida presented to the Maternal Fetal Medicine (MFM) clinic for consultation regarding a 1 cm cystic structure located at the floor of the fetal mouth. The structure had been visualized on an ultrasound in the radiology department at 21-week gestation. The patient was healthy without any significant medical or family history. Her prenatal laboratory evaluation was normal, and her medications included prenatal vitamins. An ultrasound, performed by the MFM department, at 25-week gestation, revealed a 1.4 cm
Ultrasound image at 26-week gestation demonstrating oropharyngeal cyst (black arrow).
The case was discussed in the weekly NICU/MFM conference with the pediatric ENT physician in attendance. The options of securing the airway during an EXIT procedure or post delivery after clamping the umbilical cord were discussed. Since the oropharyngeal cyst had increased to 3.1 cm, the pediatric otolaryngologist felt that the fetal airway would be more safely managed during an EXIT procedure, allowing more time for controlled bronchoscope guided intubation, and if necessary, tracheotomy, while the fetus is being perfused and oxygenated with an intact fetal-placental unit. The team was kept informed of the progress during the antenatal course and would be available at all times for the delivery. In addition, the pediatric otolaryngologist consulted the pediatric anesthesiologist who would also be present during the EXIT procedure. Furthermore, the pediatric cardiologist would be in attendance for the delivery to monitor the fetal cardiac function. The EXIT procedure was scheduled for 39 weeks of gestation. Serial MFM ultrasounds were obtained at 32 and 36 weeks of gestation revealing dimensions of the mass at 2.1 cm
The patient presented in active labor at
The patient was then taken to the operating room where general endotracheal anesthesia was administered via rapid sequence induction with thiopental and succinylcholine. Paralysis was subsequently maintained with vecuronium. Deep inhalation anesthesia was achieved with high-dose isoflurane. Maternal laparotomy was then performed via a low transverse abdominal incision to expose the uterus. A low transverse incision was made in the lower uterine segment, and the infant was delivered to the level of the upper abdomen as shown in Figure
Intraoperative image of EXIT procedure. The fetus is delivered to level of upper abdomen with the fetal head and neck stabilized by the obstetrician. The pediatric anesthesiologist performs direct laryngoscopy.
The halogenated anesthetic was discontinued and oxytocin infusion was initiated following delivery of the placenta. Uterine massage and 40 units of intravenous oxytocin effected adequate uterine tone. The hysterotomy incision was repaired in two layers. Estimated blood loss from the procedure was 1200 mL. No intraoperative fetal or maternal complications occurred. The mother’s postoperative recovery was uncomplicated. She was discharged on the second postoperative day and had a normal 6-week postpartum checkup.
A neonatal MRI confirmed the cystic mass, arising from the midline at the base of the tongue, measuring 2.4 cm
The EXIT procedure was initially designed to reverse the tracheal occlusion that was done antenatally in a fetus with severe congenital diaphragmatic hernia [
In order to achieve successful fetal oxygenation and the deep maternal anesthesia that provides fetal anesthesia during the EXIT procedure, it is necessary to maintain uterine hypotonia. In this case, uterine hypotonia was achieved using isoflurane intraoperatively. Additional methods include intravenous terbutaline or nitroglycerin [
To ensure fetal paralysis during the EXIT procedure (as recommended by the pediatric anesthesiologist), we administered intramuscular rocuronium into the fetal thigh under ultrasound guidance preoperatively. Fetal paralysis occurred in four minutes after the injection and was confirmed by ultrasound. To our knowledge, this is the first report using a fetal paralytic agent for an EXIT procedure. The decision was made based on the above reason—the clinical opinion of the pediatric subspecialists.
The EXIT procedure is associated with maternal risks that are well documented in the literature. Since it is necessary to maintain uterine hypotonia to maximize uteroplacental blood flow, the mother is at risk for hemorrhage and placental abruption.
The surgeons and anesthesia providers must have a low threshold for terminating the EXIT procedure before a significant loss of blood occurs.
In one large case series of thirty-one patients, the average time on placental bypass was 30.7 minutes, with a range from 8 to 66 minutes. The average blood loss was 848 mL [
It is important to deliver the fetus only to a level where the airway and neck can be accessed for evaluation. When the remainder of the fetal body fills the intrauterine cavity, the possibility of placental detachment is decreased. Other authors have described a technique similar to amnioinfusion to allow for persistent uterine distention with a lower risk of placental separation [
The importance of a multispecialty approach with constant communication cannot be overemphasized. In our case, despite a procedure which was scheduled to occur at 39 weeks of gestation, our patient presented in active labor at 37 weeks. The multiple team members had to assemble promptly to perform the EXIT procedure. This multispecialty team had met on several occasions during the course of this pregnancy, and thus, the role of each subspecialist was clear. This ensured a smooth assembly of all necessary personnel and equipment on short notice.
In summary, in this case of a fetal oropharyngeal mass, the fetal airway was successfully and safely secured using the EXIT procedure. In addition, this case demonstrated the importance of a multidisciplinary approach in managing such cases.
The views expressed in this article are those of the author(s) and do not necessarily reflect the official policy or position of the Department of the Navy, Department of Defense, or the United States Government. We are military service members. This work was prepared as part of our official duties. Title 17 U.S.C. 105 provides that “Copyright protection under this title is not available for any work of the United States Government.’’ Title 17 U.S.C. 101 defines a United States Government work as a work prepared by a military service member or employee of the United States Government as part of that person’s official duties.