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Publication: Ear, Nose & Throat Journal
Author: Bewley, Arnaud; Bloom, Jason D; Kherani, Safeena; Pawel, Bruce R
Date published: July 1, 2010
Language: English
PMID: 37298
ISSN: 01455613
CODEN: ENTJDO
Journal code: ENTJ

A 1 -day-old girl was transferred to the neonatal intensive care unit for management of a large intraoral mass that prevented her from feeding. A nasogastric tube was placed on arrival. The patient had been born during an uncomplicated delivery at full term to healthy parents after a normal pregnancy. All prenatal studies had been unremarkable.

On presentation, a firm, 2 ? 1 ? 1-cm polypoid, mucosalized mass with a broad-based stalk was attached in the midline to the apex of the inferior alveolar ridge (figure 1). The location and size of the mass did not allow for suckling. Findings on the remainder of the workup were unremarkable, and a clinical examination detected no other abnormalities.

The patient was scheduled for an elective excision of the mass under general endotracheal anesthesia. Following an uneventful intubation, the gingiva surrounding the lesion's stalk was infiltrated with 1% lidocaine with 1:100,000 epinephrine. The lesion was then removed by transection of the stalk with an iris scissors. Bipolar cautery was then used to obtain hemostasis.

Gross pathology demonstrated a 2.5 x 1 x 1-cm, tan, polypoid lesion. Histologically, the mass contained sheets of monomorphic polygonal cells with small round nuclei, inconspicuous nucleoli, and abundant pink granular cytoplasm (figure 2). These findings were consistent with a congenital epulis. Also noted within the lesion were abundant thin-walled vessels with perivascular acute and chronic inflammation. The overlying nonkeratinizing squamous epithelium was unremarkable and lacked pseudoepitheliomatous hyperplasia.

Congenital epulis is a rare, benign tumor of the soft tissue that presents at birth. It was first described by Neumann in 1871. ' Since then, only 161 cases have been reported in the literature before this one. Epulis most commonly arises on the anterior surface of the maxillary alveolus, and it usually affects the area of the canines and incisors.2,3 Congenital epulis is more common in girls; reported female-to-male ratios range from 3:2 to 10: 1.2,3 In 10% of cases, multiple tumors are seen.3 No known cause of epulis has been identified.

The most common presenting sign of epulis is interference with feeding; airway obstruction may also be seen. Congenital epulis has been associated with polyhydramnios secondary to an inability of the fetus to swallow amniotic fluid.4 The diagnosis of epulis can be made clinically, and it is confirmed by histologic findings. Epulis lesions are smooth, pedunculated, mucosalized masses that arise from the alveolar ridge. Prenatal diagnoses have been reported on ultrasonography.5,6 Kumar et al described a case of ultrasonographically detected epulis that was resected via an EXIT (ex utero intrapartum treatment) procedure in a neonate who was experiencing airway obstruction.5

Histologically, epulis is characterized by sheets of round cells with granular cytoplasm and small nonmitotic nuclei. Cells are often surrounded by a delicate stroma of collagen, and the overlying mucosal epithelium is often not thickened. As was the case with our patient, epulis specimens tend to contain numerous capillaries in a fine fibrovascular network. Epulis can be distinguished from more common granular cell tumors by its lack of pseudoepitheliomatous hyperplasia, the absence of S-IOO protein expression, and a positive reaction to carcinoembryonic antigen and HLA-DR antigen.3

Management involves nutritional support in the case of a mass large enough to interfere with normal suckling and establishment of a safe airway in the instance of a large obstructive mass. Simple excision is recommended under local or general anesthesia. Excision is considered curative and no instances of recurrence have been documented, so follow-up can be minimal.

References

1. Neumann E. Elin Fall von congenitaler Epulis. Arch Heilk 1871;12: 189.

2. Olson JL, Marcus JR, Zuker RM. Congenital epulis. J Craniofac Surg2005;16(1):161-4.

3. Aköz T, Erdogin B, Görgü M. Obstructive congenital epulis of the newborn. Plast Reconstr Surg 1997-,99(3):922-3.

4. Schwartz Z, Katz Z, Zosmer A, et al. Polyhydramnios caused by giant alveolar granular cell myoblastoma. Acta Obstet Gynecol Scand 1986;65(5):519-21.

5. Kumar P, Kim HH, Zahtz GD, et al. Obstructive congenital epulis: Prenatal diagnosis and perinatal management. Laryngoscope 2002;112(11):1935-9.

6. López de Lacalle JM, Aguirre I, Irizabal JC, Nogues A. Congenital epulis: Prenatal diagnosis by ultrasound. Pediatr Radiol 2001;31(6): 453-4.

Author affiliation:

Arnaud Bewley, MD; Jason D. Bloom, MD; Safeena Kherani, MD; Bruce R. Pawel, MD

Author affiliation:

From the Department of Otorhinolaryngology-Head and Neck Surgery, University of Pennsylvania School of Medicine, Philadelphia (Dr. Bewley and Dr. Bloom); and the Department of Otolaryngology (Dr. Kherani) and the Department of Pathology and Laboratory Medicine (Dr. Pawel), Children's Hospital of Philadelphia.



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