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Prenatal Diagnosis of Congenital Epulis: Implications for Delivery

Oral masses are uncommon in the fetus. The differential diagnosis is broad and includes both benign and malignant conditions. Oral masses may disrupt fetal swallowing and can affect neonatal breathing and feeding, thus potentially affecting immediate management of the neonate. Although most oral masses are diagnosed postnatally, some are detected prenatally. Accurate prenatal diagnosis is important for antenatal counseling and delivery planning, which may include the addition of surgical specialists at the delivery and, in some cases, use of the ex utero intrapartum treatment (EXIT) procedure. We present a case of a fetal oral mass, thought to be congenital epulis, which led to multidisciplinary delivery room management at the time of birth and subsequent planned surgical excision. A 32-year-old woman, gravida 2, para 0, presented to obstetric triage at a gestational age of 30 weeks 5 days with a report of leaking fluid. Her prenatal course had been otherwise uncomplicated except for placenta previa diagnosed at her 18-week anatomy scan; the previa had resolved on follow-up sonography, and there was no evidence of polyhydramnios during her prenatal course. Her medical and surgical histories were unremarkable. Preterm premature rupture of membranes was confirmed on the basis of a physical examination, vaginal pH, and microscopic evaluation. The patient started receiving latency antibiotics, was given a course of betamethasone, and was admitted to the hospital for expectant management. Sonography performed on hospital day 2 estimated the fetal weight at 1719 g (52nd percentile), in a female fetus who was in a frank breech presentation with an amniotic fluid index of 7.6. Sonography showed a 1to 2-cm spherical homogeneous solid and vascular mass protruding from the fetus’s mouth (Figure 1A). Fetal magnetic resonance imaging (MRI) performed the same day to examine the airway and further evaluate the mass showed a 1.6-cm soft tissue mass originating specifically from the gingiva of the left side of the maxilla, near the future site of the maxillary canine or lateral incisors (Figure 1B). No other abnormalities were detected. Based on the morphologic characteristics and location on MRI, the diagnosis of congenital epulis was made. Although the mass was considered to be small and unlikely to lead to substantial airway obstruction, there was concern for its growth before delivery. Thus, a plan was made for the presence of pediatric otorhinolaryngology in the delivery room for assistance with neonatal airway management, if necessary. At 32 weeks 6 days, the patient developed cramping and bleeding. Fetal heart tracing showed moderate variability with recurrent variable decelerations, and a plan was made to proceed with delivery. The female neonate was born by a classical cesarean delivery because of the breech presentation, prematurity, and an undeveloped lower uterine segment and was handed off to the awaiting neonatology and pediatric otorhinolaryngology teams. The neonate was found to have a 2-cm soft tissue mass arising from the left maxillary gingiva (Figure 1C). She did not have respiratory distress. Given prematurity, she was transferred to the neonatal intensive care unit for stabilization and was scheduled for mass excision on day 2 of life. Histologic findings of the excised mass were consistent with congenital epulis. Congenital epulis, also known as a congenital gingival granular cell tumor, is a rare benign tumor observed only in fetuses and neonates. This tumor, which can range in size from 1 mm to 9 cm, tends to arise from the anterior alveolar ridge, with the maxilla involved twice as often as the mandible. This tumor has the potential for obstruction of the oral cavity, leading to polyhydramnios prenatally and respiratory and feeding difficulty postnatally. As such, when technically possible, surgical excision is performed. The etiology of congenital epulis is unknown, although various theories of development have been postulated. A hormonal etiology has been suspected, as it is seen 10 times more often in female fetuses. It is typically found in chromosomally normal neonates without other congenital anomalies, although it has been associated with 47,XXX and polydactyly. Given that there have been no case reports of congenital epulis before 26 weeks, these tumors are thought to develop and grow in the late second and third trimesters. It is notable that in our case, 3 prior prenatal sonographic examinations, performed at 18, 22, and 28 weeks, did not show the mass; our review of the images showed normal profile views at the first 2 scans; however, views of the fetal lips and oral cavity were not obtained at the 28-week scan. First identified by Neumann in 1871, this tumor has been described in more than 200 case reports, documenting its clinical features, natural history, and surgical excision. Until recently, the tumor was primarily identified at the time of birth, but in the setting of improved ultrasound technology, prenatal diagnosis is increasingly more common. Prenatal diagnosis allows for optimal multidisciplinary delivery management and timely treatment of