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CASE REPORT
Year : 2006  |  Volume : 24  |  Issue : 2  |  Page : 104-106
 

Congenital epulis - congenital granular cell lesion: A case report


1 Dept. of Oral Medicine and Radiology, Sree Mookambika Institute of Dental Sciences, Kulasekharam, Kanyakumari, India
2 Dept. of Pedodontics, Sree Mookambika Institute of Dental Sciences, Kulasekharam, Kanyakumari, India

Correspondence Address:
Kannan S Karthiga
New Sankara Vilas, Puthukkadai, (PO) Kanyakumari District - 629 171, Tamilnadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.26026

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  Abstract 

Neumann first described congenital epulis (CE) of newborn in 1871. CE occurs at birth and has a striking predilection for females. It is most frequently located on the anterior maxillary alveolar ridge. CE clinically appears as a pedunculated protuberant mass. In cases with large lesions mechanical oral and nasal obstruction can impair fetal deglutition and neonatal respiratory efforts resulting in polyhydramnios prenatally or respiratory impairment postnatally. Histologically CE shows characteristic large cells with granular cytoplasm and spindle cells resembling fibroblasts. The exact histiogenesis is still uncertain, various theories of origin are epithelial, undifferentiated mesenchymal cells, pericytes, fibroblast, smooth muscle cells and nerve related cells. A newborn female child with CE in the maxillary alveolar ridge reported to our institution is presented with investigation, management and follow-up.


Keywords: Congenital epulis, congenital granular cell lesion, new born


How to cite this article:
Karthiga KS, Rajesh. Congenital epulis - congenital granular cell lesion: A case report. J Indian Soc Pedod Prev Dent 2006;24:104-6

How to cite this URL:
Karthiga KS, Rajesh. Congenital epulis - congenital granular cell lesion: A case report. J Indian Soc Pedod Prev Dent [serial online] 2006 [cited 2019 Dec 13];24:104-6. Available from: http://www.jisppd.com/text.asp?2006/24/2/104/26026



  Introduction Top


Congenital epulis is a reactive or degenerative lesion with a mesenchymal origin rather than a true neoplasm.[1],[2] Neumann first described CE in 1871, hence also known as Neumann's tumor.[3],[4],[5] CE occurs as a well-defined pedunculated mass with smooth or lobulated surface. CE commonly arises from the anterior maxillary alveolar ridge of newborn, not associated with any other congenital malformations.[3],[4] CE has a female predilection with 8:1 ratio; an endogenous (intrauterine) hormonal stimulus is proposed but disproved by the absence of receptor for estrogen and progesterone.[3],[4],[5],[6] This lesion is rare as reviewed by Zuker and Buenecha in 1993, who described only 167 reported cases in the literature.[5]


  Case Report Top


A two-day-old female infant was referred to the Dept. of Oral Medicine and Radiology, Sree Mookambika Institute of Dental Sciences from Velayudhan Pillai Memorial Hospital, Kulasekharam, with a growth in the anterior maxillary alveolus, which was noticed at birth. There was no difficulty in breast feeding or breathing and no other structural deformities. On intra oral examination a growth of one-centimeter diameter showing smooth surface, pink color with a narrow stalk was noticed. On palpation it was firm, not compressible or reducible and non-tender [Figure - 1]. The growth was excised under local anesthesia on the 30th day. There was minimal postoperative bleeding and the child was discharged on the same day with medications [Figure - 2]. The cut open specimen showed pale white appearance [Figure - 3], which on histopathological examination showed a growth covered with mildly acanthotic squamous epithelium and showed closely packed polygonal cells with centrally placed nuclei and markedly granular cytoplasm. No cross striations were seen. Vascularity was increased [Figure - 4]. Follow-up for three months didn't show recurrence [Figure - 5].


  Discussion Top


CE occurs in the mucosa of the maxillary alveolus as smooth surfaced pink mass, usually not associated with any other abnormality of the teeth or other congenital abnormalities. CE causing superior deviation of upper lip, flat nose with the absence of anterior nasal spine and reduced antero posterior dimension of maxilla has been reported,[3] in our case there is no other congenital abnormalities present. The exact histiogenesis is still uncertain, various proposed cell of origin are Odontogenic epithelium, undifferentiated mesenchymal cell, pericytes, fibroblast, smooth muscle cell, nerve related cell, histiocytes.[3] CE has an 8:1 female predilection and 3:1 maxillary alveolar site predilection,[3],[6] our case is a female child and lesion occurred in anterior maxillary gum pad. Like our case CE usually occurs as a single mass although 10% cases occur as multiple.[3],[4],[6] The size of the CE varies from few mm to 9 cm.[5],[6] Large lesion can interfere with fetal deglutition resulting in hydramnios and respiratory obstruction, difficulty in feeding postnatally.[3] As the size of the lesion in our case is one-centimeter diameter, it is not interfering with feeding and breathing.

Histologically CE shows highly vascularised fibrous tissue with nests of polygonal cells with large clear and granular cytoplasm and a small nucleus, with a normal overlying epithelium. The granular cytoplasmic changes have been described in many different neoplasm and non-neoplastic tissues, thus the granular cells is to be considered as the result of a metabolic change due to still unknown condition.[4] CE shows positive staining for vimentin either in the intercellular spaces or in the cytoplasm of granular cells, which can be explained by the abundance of collagen and its precursors. Positive reaction for desmin, a 53 kD fibrillar protein, is a specific marker of muscle cells. Ultra structural signs of smooth muscle differentiation were first detected in CE in 1983 and recently confirmed by other authors.[4]

Congenital epulis of the newborn is the widely accepted term and is preferred over congenital granular cell tumor (CGCT), which suggest a neoplastic origin not applicable to CE. The absence of local recurrence even after incomplete excision, the possibility of spontaneous regression and the lack of a malignant counterpart all favors that CE as a non-neoplastic lesion.[2],[4],[7] CE can be differentiated from congenital granular cell tumor (CGCT) by features shown in [Table - 1].

Sonographic prenatal diagnosis of congenital abnormalities is an invaluable tool in the management of pregnancy, delivery and postnatal treatment of the affected newborn. Prenatal diagnosis of CE is possible with ultrasonography after 25th week of intrauterine life when the images of the fetal face reveal a homogenous well-circumscribed, non-septate oral mass with doppler finding inconsistent with hemangioma.[5],[1],[2] Electron microscopic study of CE showed granular cells containing heterogeneous electron dense granules, lysosomes and cytoplasmic lipid droplets. The cells had irregular cytoplasmic borders with small extensions.[5] The clinical differential diagnosis includes cephalocele, dermoid, rhabdomyosarcoma, lymphatic malformations hemangioma, fibroma, granuloma, schwannoma.[3],[6]

Removal of CE by surgery under local[7] or general anesthesia,[5],[6],[8] electrocautery[8] and carbon dioxide laser set at 15 watts continuous wave has been reported.[5] Recurrence of the lesion and damage to the future dentition has not been reported, suggesting that radical excision is not warranted.[5]

1. Congenital Epulis is a reactive or degenerative lesion

2. Cases of spontaneous regression, lack of recurrence even after incomplete removal and lack of malignant counterpart suggest CE is a non-neoplastic lesion and needs to be differentiated from CGCT.

3. Prenatal Ultrasonographic diagnosis of CE is possible after 25th week of intra uterine life.

 
  References Top

1.Yvonne Nam, RT. Prenatal sonographic diagnosis of congenital Epulis. J Diagn Med Sonogr 2003;19:255-7.  Back to cited text no. 1    
2.Shaw L, Al-Matt A, Carlan SJ, Plumley D, Greenbaum L, Kosko J. Congenital Epulis. Three dimensional ultrasonographic findings and clinical implications. J Ultrasound Med 2004:23:1121-4.  Back to cited text no. 2    
3.Koch BL, Myeriii C, Egelhoff JC. Congenital Epulis. AJNR 1997;18:739-41.  Back to cited text no. 3    
4.Leocata P, Bifaretti G, Saltarelli S, Corbacelli A, Ventura L. Congenital (Granular Cell) Epulis of the newborn: A case report with immunohistochemical study on the histiogenesis. Ann Saudi Med 1999;19:527-9.  Back to cited text no. 4    
5.Lapid O, Shaco-Levy R, Krieger Y, Kachko L, Sagi A. Congenital Epulis. Pediatrics 2001;107:1-3.  Back to cited text no. 5    
6.Wittebole A, Bayet B, Veyckemans F, Gosseye S, Vanwijck R. Congenital Epulis of newborn. Acta Chir Belg 2003;103:235-7.  Back to cited text no. 6  [PUBMED]  
7.Dash JK, Sahoo PK, Das SN. Congenital Granular Cell Lesion "Congenital Epulis"- Report of a case. J Indian Soc Prev Dent 2004;22:63-7.  Back to cited text no. 7  [PUBMED]  
8.0nan M, YalVin O, Pul M. Congenital fibrous Epulis in the Infant. Yonsei Med J 2002;43:675-7.  Back to cited text no. 8    


    Figures

[Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5]

    Tables

[Table - 1]


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   Abstract
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