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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 42  |  Issue : 2  |  Page : 91-94

Peripheral complex odontoma: A case report and review of differences between central and peripheral types


1 Department of Oral and Maxillofacial Pathology, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran
2 Department of Periodontology, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran
3 Research Center, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran
4 Education Development Office, School of Dentistry, Tehran University of Medical Sciences, Tehran, Iran

Date of Submission03-Jan-2021
Date of Decision22-Apr-2021
Date of Acceptance25-May-2021
Date of Web Publication20-Aug-2021

Correspondence Address:
Dr. Sepideh Mokhtari
School of Dentistry, Tehran University of Medical Sciences, North Kargar Ave., Tehran
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jmedsci.jmedsci_360_20

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  Abstract 


Peripheral odontogenic neoplasms have similarities with other peripheral tumoral and nontumoral lesions. It is difficult to obtain proper diagnosis clinically and it is mostly based on the histopathologic features. Moreover, differential diagnosis of oral lesions differs between children and adults, and as a result, reports of such cases help better diagnose the lesions in this age group. Peripheral odontoma (PO), unlike central types, is uncommon. However, they have the same features as their intraosseous counterparts histopathologically. Here, we report the case of PO in a pediatric patient which was present from his infancy.

Keywords: Peripheral, odontogenic, neoplasm, odontoma


How to cite this article:
Atarbashi-Moghadam S, Lotfi A, Atarbashi-Moghadam F, Shabanipoor M, Mokhtari S. Peripheral complex odontoma: A case report and review of differences between central and peripheral types. J Med Sci 2022;42:91-4

How to cite this URL:
Atarbashi-Moghadam S, Lotfi A, Atarbashi-Moghadam F, Shabanipoor M, Mokhtari S. Peripheral complex odontoma: A case report and review of differences between central and peripheral types. J Med Sci [serial online] 2022 [cited 2022 May 23];42:91-4. Available from: https://www.jmedscindmc.com/text.asp?2022/42/2/91/324156




  Introduction Top


Odontoma is the most common odontogenic tumor or hamartoma of the jaws with central (intraosseous) and peripheral variations.[1] Peripheral odontoma (PO), unlike central types, is uncommon.[2] Histopathologically, both intraosseous and peripheral counterparts have the same features.[2] They are typically divided into compound and complex subgroups.[3] Some authors also name PO as peripheral developing odontoma.[3] Its pathogenesis is unknown, and the role of trauma is doubtful. However, a locally uncontrolled hyperactivity of rests of Serres and a hereditary basis is also suggested.[4] Custódio et al.[3] in a literature review presented 17 cases of PO during 25 years.

POs are found in children with no sex predilection.[2] It appears as a firm, slow-growing gingival mass covered by normal mucosa; therefore, the differential diagnosis is challenging and PO may be misdiagnosed clinically as common oral reactive soft-tissue lesions such as pyogenic granuloma, focal fibrous hyperplasia (irritation fibroma), peripheral giant cell granuloma, and peripheral ossifying fibroma.[5] The treatment of choice is conservative surgery, and no recurrence has been reported.[2] This article presents a slow-growing maxillary gingival mass which has been present for 2 months of age, and finally, when the patient was 6 years old, his parents sought a treatment.


  Case Report Top


A 6-year-old boy was referred to a private oral pathology center for the evaluation of a painless mass in the gingiva of left permanent maxillary central incisor which has existed from 2 months of age. The lesion gradually enlarged over this period and reached to 1 cm in diameter. The patient had no previous medical problems. Extra-oral examination was normal with no lymphadenopathy. Intra-oral examination showed a firm submucosal sessile nodule in the area of the left maxillary central incisor [Figure 1] with intact overlying mucosa. The panoramic radiograph revealed no evidence of intra-osseous changes, but soft-tissue mass had some radio-opaque structures [Figure 2]. Considering clinical and radiographic features and age of the patient, provisional diagnosis of hamartoma/coristoma, peripheral ossifying fibroma, and other peripheral odontogenic neoplasms (PONs) with hard-tissue formation such as PO and peripheral adenomatoid odontogenic tumor were made. Due to the small size of the lesion, its long duration, and its peripheral nature, an excisional biopsy was performed under local anesthesia. The underlying bone was completely intact after the removal of the lesion [Figure 3]. Grossly, the specimen was a well-circumscribed creamy soft-tissue mass measuring 1 cm × 0.8 cm. The cut surface was solid, creamy-white and nonhomogeneous and contained hard-tissue structures [Figure 4]. Histopathologic sections showed a nodular mass covered by oral epithelium. The fibrous connective tissue demonstrated a disorganized mass, without any connection to underlying bone or superficial epithelium and was composed of tubular dentin intermixed with pulpal tissues, odontogenic epithelium, ghost cells, and few basophilic areas interpreted as enamel matrix [Figure 5] and [Figure 6]. These microscopic features were consistent with the diagnosis of PO. The site of surgery was healed uneventfully, and the patient has remained free of the disease for 3 years.
Figure 1: The patient presented with a firm submucosal sessile nodule on the gingiva of left primary maxillary central incisor (black arrow)

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Figure 2: Periapical radiography revealed evidence of some radio-opaque structures in soft tissue between primary central incisors without intra-osseous changes

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Figure 3: The underlying bone was completely intact after excision of the lesion

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Figure 4: Gross section showed solid, creamy-white, and nonhomogeneous cut surface contained hard-tissue structures

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Figure 5: Histopathological section demonstrated a disorganized mass which had dentinoid material (black arrowhead) and was covered by the oral mucosa (black arrow) (H and E ×40)

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Figure 6: (a) Nodular mass had no connection to superficial epithelium and contained ghost cells (white arrow) (H and E ×100). (b) Residue of enamel organ and dental papilla was seen (H and E ×100). (c) Residue of enamel organ and dentin were present (H and E ×400). (d) A disorganized mass of dentin was seen intermixed with small particles resembling enamel matrix (H and E ×400)

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  Discussion Top


Although central odontoma is the most common odontogenic neoplasm, the most common peripheral odontogenic tumors are peripheral odontogenic fibroma and peripheral ameloblastoma.[4] PO typically occurs in young children. Congenital form is also reported.[1] Custódio et al.[3] reported a wide age involvement ranging from 5 months to 39 years. The mean age of 6.6 years old is reported for PO which is 10 years younger than the mean age of patients with central odontoma.[1] This is probably related to the intraosseous nature of central odontoma; thus, clinical signs appear later in life or are never displayed. In our patient, the lesion has developed for 2 months of age. Due to previous reports, maxilla is the most common location.[3] The source of PO is the remnants of dental lamina which is located in the gingiva (rests Serres). PO grows lingually or palatally to those of the primary teeth,[2],[5] and it is also named gingival odontoma.[3] Unlike several case reports,[1],[4] in the present case, buccal aspect of the anterior maxilla was involved.

Intraosseous odontoma may erupt mostly as the result of the eruptive force of nonerupted, underlying teeth. PO rarely erupts, and this process can be an exfoliation rather than a real eruption. It is suggested that this exfoliation may be because of the growth of lesion over time.[6] The present case was not erupted or exfoliated.

PONs are uncommon and usually neglected in differential diagnosis. Thus, understanding clinicopathological features of these tumors is necessary.[5] The clinical differential diagnosis of PON is most often reactive soft-tissue lesions and benign soft-tissue neoplasms.[1],[5],[7] The clinical presentation of our case was similar to irritation fibroma, although peripheral-ossifying fibroma and PON were indicated because of the gingival location and hard-tissue formation in radiography.

Histopathologically, peripheral variant of ameloblastic fibro-odontoma (PAFO) and peripheral dentinogenic ghost cell tumor (PDGT) may be confused with PO.[2] PDGT shows islands of ameloblastic epithelium with peripheral palisaded columnar cells, central stellate reticulum, and eosinophilic ghost cells in a fibrous connective tissue with variable amounts of dentinoid material.[8] Ghost cell differentiation is reported in some odontogenic tumors such as ameloblastoma, odontoma, and adenomatoid odontogenic tumor, but its amount is much less than dentinogenic ghost cell tumor.[9] Ghost cell changes were also seen in the current case, but the reason for their presence remained unclear. Although some authors suggested that ghost cells be also present in inner enamel epithelium of a normal developing human tooth,[10] moreover, dental papilla-like structures are not evident in PDGT.[8] Ameloblastic fibro-odontoma is not now considered as a distinct entity and it is mentioned as a part of the spectrum of histopathologic changes seen in a developing odontoma.[11] In this case, the dental tissues were well circumscribed, and epithelial component was scarce. Consequently, we preferred the diagnosis of PO to PAFO. PO shows a limited growth potential, and its treatment of choice is simple excision and no recurrence has been reported.[2] Although malignant transformation in PONs such as peripheral ameloblastoma has been documented,[5] there is no evidence or previous report of malignant PO.


  Conclusion Top


Due to the clinical similarity of PONs with gingival tumoral and nontumoral lesions, it is difficult to obtain proper diagnosis clinically and it is mostly based on the histopathologic features. Moreover, differential diagnosis of oral lesions differs between children and adults and as a result, reports of such cases help better diagnose the lesions in this age group.

Declaration of patient consent

All appropriate patient consent forms were obtained from the patient's parents. They give his consent for his images and other clinical information to be reported in the journal. They understand that his names and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Mikami T, Yagi M, Mizuki H, Takeda Y. Congenital peripheral developing odontoma accompanied by congenital teratomatous fibroma in a 9-month-old boy: A case report. J Oral Sci 2013;55:89-91.  Back to cited text no. 1
    
2.
Atarbashi Moghadam S, Mokhtari S. Peripheral developing odontoma or peripheral ameloblastic fibroodontoma: A rare challenging case. Case Rep Dent 2016;2016:9379017.  Back to cited text no. 2
    
3.
Custódio M, Araujo JP, Gallo CB, Trierveiler M. Gingival complex odontoma: A rare case report with a review of the literature. Autops Case Rep 2018;8:e2018009.  Back to cited text no. 3
    
4.
Ide F, Obara K, Mishima K, Saito I, Horie N, Shimoyama T, et al. Peripheral odontogenic tumor: A clinicopathologic study of 30 cases. General features and hamartomatous lesions. J Oral Pathol Med 2005;34:552-7.  Back to cited text no. 4
    
5.
Atarbashi-Moghadam S, Lotfi A, Atarbashi-Moghadam F, Ghaedsharafi Y. Peripheral ameloblastoma in an adolescent girl: Differential diagnosis with other gingival lesions. J Clin Diagn Res 2018;12:ZD01-3.  Back to cited text no. 5
    
6.
Reibel J, Grønbaek AB, Poulsen S. Peripheral ameloblastic fibro-odontoma or peripheral developing complex odontoma: Report of a case. Int J Paediatr Dent 2011;21:468-70.  Back to cited text no. 6
    
7.
Saghravanian N, Jafarzadeh H, Bashardoost N, Pahlavan N, Shirinbak I. Odontogenic tumors in an Iranian population: A 30-year evaluation. J Oral Sci 2010;52:391-6.  Back to cited text no. 7
    
8.
Candido GA, Viana KA, Watanabe S, Vencio EF. Peripheral dentinogenic ghost cell tumor: A case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;108:e86-90.  Back to cited text no. 8
    
9.
Singhaniya SB, Barpande SR, Bhavthankar JD. Dentinogenic ghost cell tumor. J Oral Maxillofac Pathol 2009;13:97-100.  Back to cited text no. 9
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10.
Mehendiratta M, Bishen KA, Boaz K, Mathias Y. Ghost cells: A journey in the dark.Dent Res J (Isfahan) 2012;9 (Suppl 1):S1-8.  Back to cited text no. 10
    
11.
Atarbashi-Moghadam S, Ghomayshi M, Sijanivandi S. Unusual microscopic changes of ameloblastic fibroma and ameloblastic fibro-odontoma: A systematic review. J Clin Exp Dent 2019;11:e476-81.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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