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Year : 2009  |  Volume : 52  |  Issue : 3  |  Page : 436-437
Congenital epulis of the newborn


1 Department of Pathology, Sultan Qaboos Hospital, Salalah, Sultanate of Oman
2 Department of Pediatric Surgery, Sultan Qaboos Hospital, Salalah, Sultanate of Oman

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Date of Web Publication12-Aug-2009
 

How to cite this article:
Gokhale UA, Malhotra CJ. Congenital epulis of the newborn. Indian J Pathol Microbiol 2009;52:436-7

How to cite this URL:
Gokhale UA, Malhotra CJ. Congenital epulis of the newborn. Indian J Pathol Microbiol [serial online] 2009 [cited 2019 Dec 10];52:436-7. Available from: http://www.ijpmonline.org/text.asp?2009/52/3/436/55020


A full-term normally delivered female child was referred to the department of pediatric surgery immediately after birth for a protruding, sessile soft tissue swelling from her mouth. It was arising from the mucosa of the upper alveolar ridge of the left side of the maxilla. It was interfering with the feeding but did not cause airway obstruction. Otherwise the infant was healthy. No other structural abnormalities were noted. The preoperative diagnosis considered was a granuloma. The tumor was excised under local anesthesia on the next day and subjected to histopathological examination. The postoperative period was uneventful.

Grossly, the mass was oval, grayish white and firm in consistency measuring 1.8 1.5 0.4 cm. The external surface was slightly irregular. The cut surface was grayish white and well-circumscribed [Figure 1].

Microscopically, H and E sections showed a well-circumscribed lesion composed of sheets of closely packed polygonal cells with abundant, coarsely granular, eosinophilic cytoplasm. The nuclei were small, uniform, round to oval and centrally or slightly eccentrically situated with well-defined, prominent nucleoli. There was a delicate network of fine capillaries traversing the lesion [Figure 2]a and b. It was covered by normal stratified squamous epithelium [Figure 3] showing focal ulceration. With this classical histological pattern, a diagnosis of congenital epulis of the newborn was rendered. Immunohistochemistry further confirmed the diagnosis as the tumor cells were diffusely and strongly positive for vimentin and negative for S-100 protein and desmin. Smooth muscle antigen (SMA) was positive within the blood vessels.

The excision was not complete as the tumor was seen at the surgical margin. The child has followed up for the last 18 months and there is no evidence of local recurrence.

Congenital epulis is a rare benign tumor, also known as congenital granular cell tumor of the newborn or Neumann's tumor. [1] Since its first description by Neumann in 1871, around 200 cases have been reported in the worldwide literature so far. [2] It presents as a smooth-surfaced, tan-colored, sessile or pedunculated, firm mass, typically seen protruding from the neonate's mouth. [1],[3] It is seen three times more frequently on the anterior alveolar ridge of the maxilla than from the mandible with a female to male ratio of 8 to 10:1. [2] Usually, it appears as a single lesion, but in 10% cases it may arise from multiple locations simultaneously. [4] Other than gingiva, a few cases of congenital epulis have been reported on the tongue. [5] The condition is not associated with any other dental abnormalities or congenital malformations. The size of the tumor may vary from a few millimeters to 9 cm. Larger lesions may cause prenatal hydramnios due to ineffective deglutition and postnatally feeding and breathing difficulties. [4]

There is a striking histological similarity between congenital epulis and the more common acquired granular cell tumors of any other site. Both have large cells with eosinophilic granular cytoplasm. The difference is made on the basis of lack of pseudoepitheliomatous hyperplasia of the overlying squamous epithelium, plexiform arrangement of capillaries and positivity for vimentin and lack of S-100 protein in immunohistochemistry in congenital epulis. Other distinguishing features that aid the diagnosis are its presence in newborn female babies and its unique location. [1],[2]

Prenatal ultrasound has been used to report congenital epulis in sporadic cases after 25th week of gestation, but the findings are nonspecific and ultrasonic differential diagnoses include congenital malformations and various benign and malignant intraoral tumors. [4],[5] It can, however, help in the management of pregnancy, delivery and postnatal management especially if the lesion is large. [4] In our case, the prenatal ultrasound did not reveal any intraoral mass.

The histogenesis of congenital epulis is uncertain and lacks consensus. Several theories have been postulated to explain the origin and pathology of the lesion viz. myoblastic, odontogenic, neurogenic, fibroblastic, histiocytic or pluripotent precursors of these cells. [5] However, most of the studies point its origin towards primitive mesenchymal cells with myofibroblastic differentiation. [1]

As congenital epulis has female preponderance, stimulation in utero by maternal hormones has also been suggested but not proved as estrogen and progesterone receptor studies were negative. [1]

Despite two spontaneous regressions reported in the literature, surgery is the treatment of choice. Radical resection is not warranted as it is likely to damage the underlying alveolar bone and developing tooth buds. No recurrence has been reported even when the excision has been incomplete. [3]

The tumor is often misdiagnosed prior to surgery because of its rarity and lack of awareness, as in our case. The clinical differential diagnoses include hemangioma, lymphangioma, fibroma, granuloma, rhabdomyoma, embryonal and alveolar rhabdomyosarcoma, chondrogenic and osteogenic sarcoma, schwanomma and heterotopic gastrointestinal cyst. [1],[3]


   Acknowledgement Top


We thank Dr. S. Thomas, Senior Consultant and Head, Department of Histopathology, Royal Hospital, Muscat, Oman for Immuno- histochemistry.

 
   References Top

1.Lapid O, Shaco-Levy R, Krieger Y, Kachko L, Sagi A. Congenital epulis. Pediatrics 2001;107:e22.   Back to cited text no. 1  [PUBMED]  [FULLTEXT]
2.Fister P, Volavsek M, Novosel Sever M, Jazbec J. A newborn baby with a tumor protruding from the mouth. Acta Dermatoven APA 2007;16:128-9.  Back to cited text no. 2    
3.Wittebole A, Bayet B, Veyckemans F, Gosseye S, Vanwijck R. Congenital epulis of the newborn. Acta Chir Belg 2003;103:235-7.  Back to cited text no. 3  [PUBMED]  
4.Kannan SK, Rajesh R. Congenital epulis - congenital granular cell lesion: A case report. J Indian Soc Pedod Prev Dent 2006;24:104-6.  Back to cited text no. 4    
5.Feller F, Wood NH, Singh AS, Raubenheimer EJ, Meyerov R, Lemmer J. Multiple congenital oral granular cell tumours in a newborn black female: A case report. Cases Journal 2008,1:13. Available from: http://www.casesjournal.com/content/1/1/13 . [cited 2008 May 30]  Back to cited text no. 5    

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Correspondence Address:
Uday A Gokhale
P.O. Box 1094, G.P.O. Salalah - 211, Sultanate of Oman

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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.55020

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    Figures

  [Figure 1], [Figure 2], [Figure 3]

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