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CASE REPORT
Year : 2015  |  Volume : 33  |  Issue : 4  |  Page : 351-354
 

Congenital peripheral ameloblastic fibroma with intraosseous involvement in a 2-week-old infant: A case report with review of literature


1 Department of Pathology, Sir Ganga Ram Hospital, New Delhi, India
2 Department of Pathology, Lady Hardinge Medical College, New Delhi, India
3 Department of Pathology, UP Rural Institute of Medical Sciences and Research, Saifai, Etawah, Uttar Pradesh, India
4 Department of Dental and Oral Surgery, Lady Hardinge Medical College, New Delhi, India

Date of Web Publication18-Sep-2015

Correspondence Address:
Dr. Savita Agarwal
Department of Pathology, UP Rural Institute of Medical Sciences and Research, Saifai, Etawah, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.165721

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   Abstract 

Ameloblastic fibroma is a rare, slow-growing benign mixed odontogenic tumor. It constitutes 2% of odontogenic tumors and is reported to occur at an age ranging from 6 months to 42 years. The youngest being a 7-week-old infant. We report a case of peripheral ameloblastic fibroma in a 2-week-old infant. The lesion presented since birth. It involved the maxilla with an extraosseous component involving the gingiva. A more or less conservative surgical approach of enucleation and curettage of the lesion was done under general anesthesia, trying to conserve the adjacent tooth buds. Only a few cases of congenital peripheral ameloblastic fibroma have been reported so far.


Keywords: Ameloblastic fibroma, odontogenic tumors, peripheral


How to cite this article:
Langer S, Choudhury M, Agarwal S, Mehra P. Congenital peripheral ameloblastic fibroma with intraosseous involvement in a 2-week-old infant: A case report with review of literature. J Indian Soc Pedod Prev Dent 2015;33:351-4

How to cite this URL:
Langer S, Choudhury M, Agarwal S, Mehra P. Congenital peripheral ameloblastic fibroma with intraosseous involvement in a 2-week-old infant: A case report with review of literature. J Indian Soc Pedod Prev Dent [serial online] 2015 [cited 2019 Jul 20];33:351-4. Available from: http://www.jisppd.com/text.asp?2015/33/4/351/165721



   Introduction Top


Ameloblastic fibroma is a rare, slow-growing benign mixed odontogenic tumor. [1],[2] It is reported to constitute 2% of odontogenic tumors. [1],[2],[3] It is generally found in the mandible and usually remain asymptomatic, except for the expansion of the cortical plates of the jaws.

We present a case of a 2-week-old infant girl with congenital ameloblastic fibroma of the maxilla with soft tissue involvement. A more or less conservative surgical approach of enucleation and curettage of the lesion was done under general anesthesia, trying to conserve the adjacent tooth buds. A brief review of literature in relation to ameloblastic fibroma is also presented.


   Case Report Top


A 2-week-old infant girl presented to the Department of Dental and Oral Surgery, with soft tissue swelling of the gingiva in the region of the maxillary alveolar process since birth. The swelling gradually increased in size with difficulty in breastfeeding.

The medical history of the mother and the infant were unremarkable. Physical examination of the infant revealed no abnormalities except for the growth present intraorally. The growth was reddish, soft to firm, 2 × 2 cm in size involving the right canine region of the alveolar crest [Figure 1] with virtually no expansion of the buccal and palatal cortical plates on X-ray.
Figure 1: Preoperative photographs of the infant at 7 weeks of age showing intraoral soft tissue growth


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A provisional diagnosis of congenital epulis was made. An excisional biopsy of the growth was done and sent for histopathological evaluation. A soft to firm mass measuring 2 × 1.5 cm was received. External surface was gray-white with focal areas of hemorrhage. Microscopically sections showed tumor comprising of strands and groups of cuboidal to columnar epithelial cells arranged in irregular branching strands with cellular intervening connective tissue stroma [Figure 2] and [Figure 3]. Based on these findings diagnosis of ameloblastic fibroma was made. On follow-up, at 3 months of age, the swelling had increased in size, with the expansion of buccal cortical plate and superior displacement of the upper lip.
Figure 2: Photomicrograph of primary lesion-ameloblastic fibroma showing several irregular branching strands of epithelial cells with surrounding cellular connective tissue stroma (H and E, ×100)


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Figure 3: Higher magnification of same lesion showing irregular branching strands of cuboidal to columnar epithelial cells and cellular connective tissue stroma (H and E, ×400)


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An occipitomental view radiograph revealed a multilocular, radiolucent lesion with scalloped, sclerotic margins. It was 2 cm in size and was associated with the crown of a deciduous tooth. Computed tomography scan [Figure 4] showed a large 2 × 2 cm lesion involving the right anterior maxilla that was well circumscribed and associated with the crown of a deciduous tooth. The floor of orbit and the nose were free. Excision of the lesion from the maxilla was performed. The surrounding bone was curetted, and a (1 cm) soft tissue clearance margin of buccal and palatal mucoperiosteum was made. Specimen was sent for histopathological evaluation. The lesion encased in bone measured 3 × 4 × 4 cm was whitish-yellow and soft to firm in consistency. Histopathologically, the jaw swelling showed similar morphology as that of previous excisional biopsy and was again reported as peripheral ameloblastic fibroma, and all margins were reported free from tumor.
Figure 4: Axial computed tomography scan showing a 2 × 2 cm, well-circumscribed growth associated with the crown of a deciduous tooth in right maxilla


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The postoperative course was uneventful, and the patient was discharged for further follow-up in the open patient department. Healing was uneventful, and there was no sign of recurrence 1 year after surgery [Figure 5]. She will continue to be evaluated every 6 months.
Figure 5: Postoperative image of the same case showing no recurrence


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   Discussion Top


Ameloblastic fibroma is a relatively rare, benign neoplasm belonging to the group of mixed odontogenic tumors in which both the epithelial and ectomesenchymal elements are neoplastic. It is characterized by the simultaneous proliferation of epithelial and mesenchymal tissues, without the formation of dentin or enamel. [3]

The precise etiology of ameloblastic fibroma is unknown so far. However, it is believed to arise denovo during odontogenesis probably as a result of over-zealous, elaboration of the basal lamina without further odontogenic differentiation. [4]

Ameloblastic fibroma is reported to occur at an age ranging from 6 months to 42 years with an average of 14.6-15.5 years. [1] The youngest being reported in a 7-week-old infant. [5] The reported sex predilection varies from no preference to males being more frequently affected than females. [1] Over 80% of tumors occur in the mandible the usual site being the canine molar region. Only four cases of tumors in the anterior maxillary region have been reported. [1]

Trodahl reported that in 58% of cases the presenting symptom was a swelling. [1] Thus, it is infrequently associated with complaints and has been discovered accidentally during the roentgenographic examination. In the present case, multilocular osteolytic images were observed radiographically. In the study of 24 cases by Trodahl, most of the lesions were associated with unerupted teeth. [1] It was similar in our case, as unerupted deciduous canine and molar teeth were associated with the lesion.

Very few cases of peripheral ameloblastic fibroma have been reported in the literature. [6],[7],[8] Nakamura and Tamai described the tumor arising from the mandibular posterior alveolus of a 2-year-old boy as an epulis like mass. [6] Harada et al. reported a similar case in the mandibular molar gingiva of a 1-year-old girl. [7] The only difference being that the tumor reported by Harada et al. was congenital. In our case too, the tumor was initially limited to the anterior maxillary gingiva and was present congenitally. Kusama et al. also reported a case of peripheral ameloblastic fibroma found in the lower right premolar region of a 40-year-old woman. [8]

The peripheral ameloblastic fibroma needs to be distinguished histologically from the peripheral odontogenic fibroma; that shows features varying from myxoid, fibroblastic, to densely hyalinized. Odontogenic epithelium may be absent, scanty and inactive or forming cords and nests, with or without osteoid. The peripheral ameloblastic fibroma exhibits prominent cords and islands of epithelium with central areas resembling stellate reticulum, against a pale staining myxoid stroma that resembles dental papilla, as opposed to the tissue morphology seen in odontogenic fibromas. The epithelial islands are reminiscent of the early stages of enamel organ development. [9]

Gupta et al. have clearly mentioned that these patients must be followed up for a long period to detect the early recurrence or development of ameloblastic fibrosarcoma, which is the malignant counterpart of ameloblastic fibroma. [10]


   Conclusion Top


Ameloblastic fibroma though a rare tumor may be present congenitally. In our case, it involved the maxilla and presented as an extraosseous growth, besides the main tumor being intraosseous. Ameloblastic fibroma should be considered in the differential diagnosis of growths involving the gingiva.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Trodahl JN. Ameloblastic fibroma. A survey of cases from the Armed Forces Institute of Pathology. Oral Surg Oral Med Oral Pathol 1972;33:547-58.  Back to cited text no. 1
    
2.
Slootweg PJ. An analysis of the interrelationship of the mixed odontogenic tumors - Ameloblastic fibroma, ameloblastic fibro-odontoma, and the odontomas. Oral Surg Oral Med Oral Pathol 1981;51:266-76.  Back to cited text no. 2
    
3.
Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors: Analysis of 706 cases. J Oral Surg 1978;36:771-8.  Back to cited text no. 3
    
4.
Eversole LR, Tomich CE, Cherrick HM. Histogenesis of odontogenic tumors. Oral Surg Oral Med Oral Pathol 1971;32:569-81.  Back to cited text no. 4
    
5.
Mosby EL, Russell D, Noren S, Barker BF. Ameloblastic fibroma in a 7-week-old infant: A case report and review of the literature. J Oral Maxillofac Surg 1998;56:368-72.  Back to cited text no. 5
    
6.
Nakamura M, Tamai K. A case of ameloblastic fibroma in an infant (2 year old). J Jpn Stomatol Soc 1980;29:99-103.  Back to cited text no. 6
    
7.
Harada H, Kusukawa J, Oh-uchida M, Tanaka S, Okina T, Kameyama T. Ameloblastic fibroma in an infant - A case report. J Jpn Soc Oral Tumors 1992;4:286-93.  Back to cited text no. 7
    
8.
Kusama K, Miyake M, Moro I. Peripheral ameloblastic fibroma of the mandible: Report of a case. J Oral Maxillofac Surg 1998;56:399-401.  Back to cited text no. 8
    
9.
Darling MR, Daley TD. Peripheral ameloblastic fibroma. J Oral Pathol Med 2006;35:190-2.  Back to cited text no. 9
    
10.
Gupta A, Sood R, Bansal P. Bilateral ameloblastic fibroma in a 6 year old child: A unique case report. J Oral Health Community Dent 2010;4:88-91.  Back to cited text no. 10
    


    Figures

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



 

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