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Table of Contents   
CASE REPORT  
Year : 2012  |  Volume : 23  |  Issue : 3  |  Page : 434-435
Concomitant occurrence of cemento-ossifying fibroma and adenomatoid odontogenic tumor with bilateral impacted permanent canines in the mandible


1 Department of Oral Maxillofacial Pathology, G. Pulla Reddy Dental College & Hospital, Kurnool, India
2 Department of Oral Maxillofacial surgery, G. Pulla Reddy Dental College & Hospital, Kurnool, India
3 Department of Oral Maxillofacial surgery, Krishnadevarya College of Dental Sciences, Bangalore, India

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Date of Submission20-Feb-2011
Date of Decision01-Jun-2011
Date of Acceptance20-Mar-2012
Date of Web Publication11-Oct-2012
 

   Abstract 

Adenomatoid odontogenic tumor (AOT) is an uncommon, benign and slow growing odontogenic tumor, which is usually located in an anterior region of the maxilla without pain. Cemento-ossifying fibroma (COF) is a relatively rare benign tumor of the jaw. Here we present 2 lesions presenting in unusual forms, follicular variant of AOT in the mandible and COF associated with impacted canine in the mandible, occurring concomitantly in the same patient. Both lesions presented classic histopathologic features.

Keywords: Calcifications, impacted teeth, mandible, tumor

How to cite this article:
Prakash A R, Reddy P S, Rajanikanth, Bavle RM. Concomitant occurrence of cemento-ossifying fibroma and adenomatoid odontogenic tumor with bilateral impacted permanent canines in the mandible. Indian J Dent Res 2012;23:434-5

How to cite this URL:
Prakash A R, Reddy P S, Rajanikanth, Bavle RM. Concomitant occurrence of cemento-ossifying fibroma and adenomatoid odontogenic tumor with bilateral impacted permanent canines in the mandible. Indian J Dent Res [serial online] 2012 [cited 2019 Aug 23];23:434-5. Available from: http://www.ijdr.in/text.asp?2012/23/3/434/102255
Cemento-ossifying fibroma (COF) is a bone producing, slow growing, asymptomatic, well demarcated, benign lesion of the jaw. [1] It typically affects the females in the age group of 20-40 years, with a predilection for the mandibular premolar-molar region, causing a painless swelling with slow expansile growth. [2] Its presumed origin from periodontal membrane may explain the production of bone or cementum. Cemento-ossifying fibroma and ossifying fibroma have been discussed and termed synonymously. However, the term cementifying fibroma is reserved for an occurrence of cementum-like calcifications alone. When the lesion presents with both cementum-like calcifications and bony trabaculae, it is then termed cemento-ossifying fibroma. These lesions are restricted to only the jaw bones; however, separating these entities is difficult and always discussed together in the literature. Most of the cemento ossifying fibroma in the jaws occur unilaterally in the mandible. [3]

In the early stages, the cemento-ossifying fibroma appears as a radiolucent lesion with no evidence of radiopacities. As the tumor matures, there is an increase in the calcification, so that the radiolucent area becomes flecked with opacities until ultimately the lesion appears as an extremely radiopaque mass. Displacement of adjacent teeth is common. One additional important diagnostic feature is that, there is a centrifugal growth pattern rather than a linear one, and therefore, the lesions grow by expansion equally in all the directions and present as a round tumor mass. [4]

Adenomatoid odontogenic tumour (AOT), first described by Steensland in 1905, is a relatively an uncommon benign, slow growing tumor that is often clinically diagnosed as an odontogenic cyst. AOT accounts for 3% of all odontogenic tumors. It is predominantly found in young female patients, located more often in an anterior maxilla in association with an unerupted permanent tooth, usually lateral incisor and canine. It often causes an expansion of the surrounding bone and displacement of the adjacent teeth. [5]

A rare case of bilaterally impacted canine, associated with cemento-ossifying fibroma and adenomatoid odontogenic tumor, occurring concomitantly in the mandible of the same patient is presented herewith.


   Case Report Top


A 30-year-old female patient presented with a slow growing swelling of 4 months duration on the right side of an anterior mandible. The patient had no systemic disease or used any medication. There was no history of trauma, pain, discharge or any other symptoms related to the lesion [Figure 1].
Figure 1: Clinical pic showing slight swelling on the right mandible

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Intra-orally, a swelling was noted with mild labiolingual expansion in the region of 42-45, with an extension lingually till the lingual frenum [Figure 2]. The swelling was hard with well defined margins and normal overlying mucosa. There was retained deciduous canine in region of 43. Laboratory findings including routine hemogram, urine analysis and serum calcium, serum phosphorus, serum alkaline phosphates were all within the normal limits.
Figure 2: Intra-oral pic showing lesion extending lingually to the lingual frenum

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The radiograph revealed a mixed radiolucent-radiopaque lesion of approximately 2x3 cm, extending from 42 to 44 region with well-defined sclerotic periphery. An impacted permanent canine was noted between divergent roots of 42 and 44 region. An inferior border of the mandible was thinned down, but intact [Figure 3]. Depending on the clinical and the radiographic examination and considering the impacted canine, the differential diagnosis of odontogenic tumors like calcifying epithelial odontogenic tumor, ghost cell odontogenic tumor, cementoblastoma and fibro-osseous lesions were considered.
Figure 3: OPG showing right and left lesions in the mandible with bilateral impacted permanent canines

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The OPG also revealed the presence of another separate and clinically asymptomatic lesion in relation to the mandibular left canine, that showed a unilocular radiolucency with sclerotic margin and an impacted left canine with diverging roots of 32 and 34 region [Figure 3]. The differential diagnoses considered, were dentigerous cyst and adenomatoid odontogenic tumor. An aspiration of the lesion on the left side of the mandible did not yield fluid, thereby suggesting a provisional diagnosis of adenomatoid odontogenic tumor.

An incisional biopsy was taken from the lesion on the right side of the mandible, while the contralateral lesion was enucleated along with an impacted 33 region. Gross examination of the enucleated specimen showed a single reddish-white soft tissue, measuring 1.5cmx1.5cm, was attached to the cervical portion of the extracted left mandibular permanent canine. [Figure 4].
Figure 4: Excised specimen of AOT with extracted canine attached

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Histologically, the lesion on the left side showed spindle-shaped cells that formed sheets, strands and whorled masses with scanty fibrous stroma [Figure 5]. The odontogenic epithelial cells showed rosette-like arrangement with central spaces [Figure 6]. Small foci of calcifications were scattered throughout the tumor. Based on the classic picture, a diagnosis of follicular variant of adenomatoid odontogenic tumor was made [Figure 7].
Figure 5: Odontogenic epithelial cells arranged in sheets and islands with scanty connective tissue (H and E, ×10)

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Figure 6: Odontogenic epithelium in the form of rosettes with induction (H and E, ×20)

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Figure 7: Odontogenic epithelial islands with foci of calcifications (H and E, ×10)

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Histopathological examination of the cellular lesion on the right side of anterior mandible showed dense bundles of collagen fibers along with spicules of lamellated bone [Figure 8]. The periphery of the lesion showed discrete round masses of cementum-like material [Figure 9]. A diagnosis of central cemento-ossifying fibroma was thus made. The lesion was surgically excised and microscopic examination of the rest of the specimen confirmed the diagnosis.
Figure 8: Bundles of collagen fibers with moderate cellularity and few calcifications (H and E, ×10)

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Figure 9: Calcifications of both lamellar bone and cementoid areas (H and E, ×10)

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   Discussion Top


Adenomatoid odontogenic tumor and cemento-ossifying fibroma are well-recognized, yet rather uncommon lesions presenting in the jaw bones. However, these lesions presenting concomitantly is extremely rare.

The AOT is an uncommon cause of jaw swelling. It shows a slightly higher predilection for occurrence in females, and appears most often in the second decade of life. [5] We described that the sex and the age of the patient are consistent with that mentioned in the literature. The lesions are typically asymptomatic, but may cause cortical expansion and displacement of the adjacent teeth, as in the case reported here. While the origin of the AOT is controversial, because of its predilection for tooth-bearing regions of bone, it is thought to arise from odontogenic epithelium. [6]

The AOT has 3 clinico-pathologic variants, namely intraosseous follicular, intraosseous extra-follicular, and peripheral. The follicular type (in 73% of all AOT cases) is associated with an unerupted tooth as in the case we presented here, whereas extra-follicular type (24%) has no relation with an impacted tooth, and the peripheral variant (3%) is attached to the gingival structures. Follicular and extra-follicular types are common in the maxilla than in the mandible, and most of the tumors involve an anterior aspect of the jaws. [6] In this case, the tumor was a follicular intraosseous type, found in an anterior region of the mandible. Histologically, the present case showed classic microscopic features of AOT with odontogenic epithelium in the form of rosettes and showing induction with calcifications in few areas.

Cemento-ossifying fibromas are a distinct form of benign fibro osseous lesions of the jaws. They are thought to arise from the periodontal ligament and are composed of varying amounts of cementum, bone, and fibrous tissue. The hybrid name cemento-ossifying fibroma encompasses a spectrum of fibro-osseous lesions that arise from the periodontal ligament, ranging from those with only deposition of cementum, to those with only deposition of bone. They arise in the mandible in 62% to 89% of patients, 77% occurring in the premolar region. Most cases are diagnosed between 20 and 40 years of age. [7] The findings in the present case of a 30-year-old female patient with the lesion in an anterior mandible are, therefore, consistent with those mentioned in the literature. Central ossifying fibromas are asymptomatic, until they cause an expansion and are generally not diagnosed, until the tumor has progressed with time to produce the calcifications. Central ossifying fibromas are typically well-defined, solitary radiolucencies with scattered radiopaque foci. They maintain a spherical shape, expand the surrounding cortical bone without cortical perforation, and may cause tooth divergence.

The common differential diagnosis includes other lesions that contain radiopacities within a well-defined radiolucent mass in the jaws such as pagets disease, fibrous dysplasia, osseous dysplasia, cementoblastoma, odontogenic cysts, calcifying odontogenic cyst (gorlin cyst), and calcifying epithelial odontogenic tumor (pindborg tumor). Fibrous dysplasia has a characteristic "ground glass" appearance, not seen in the central ossifying fibroma. The radiographic differentiation of central ossifying fibroma from gorlin cysts- and pindborg tumors- is difficult, and the final diagnosis is based on the microscopic appearance.

Pathologic examination of the cemento-ossifying fibroma shows a proliferation of irregularly-shaped calcifications within a hypercellular fibrous connective tissue stroma. The calcifications are extremely variable in an appearance and represent various stages of bone and cementum deposition. Histologic differentiation between osteoid and cementum is difficult. In some cases, most of the calcified fragments are immature cementum, with basophilic coloration on hematoxylin and eosin-stained sections. These tumors have been named central cementifying fibroma. In other cases, the calcified fragments are osteoid, with typical eosinophilic coloration on hematoxylin and eosin-stained sections. These tumors have been named central ossifying fibromas. However, central ossifying fibromas also can be basophilic, causing difficulties when differentiating from central cementifying fibromas. The hybrid term cemento-ossifying fibroma has evolved to indicate the likely presence of both types of tissue within the same lesion because of the difficulty in reliably distinguishing immature bone from immature cementum, and because of the presence of both of these substances in many of the lesions. [8]

Thus, cemento-ossifying fibroma is the most preferred histologic term, but it can be interchanged with either central ossifying fibroma or central cementifying fibroma. There is no apparent clinical or radiologic difference between the two. In this case, both types of calcifications were found, i.e. rounded cementum-like structures found more at the periphery and irregular bony trabaculae scattered throughout the lesion.

In the present case, histopathology of both the lesions were classical and made the diagnosis simple, but their association with impacted teeth made the preliminary clinical and radiographic differential diagnosis difficult. Other than the association of impacted teeth and presence of calcifications in both the lesions, no other features were common. The association of an impacted tooth to AOT is well documented, but the association between COF and an impacted tooth might be a coincidental finding. There was only one case reported by Yoshida M et al in 2006 of COF, associated with multiple impacted teeth in the maxillary bone. [9] In 2003, A Baberi et al reported bilateral COF of the maxillary sinus, [10] but there is no reported case of AOT and COF occurring concomitantly. The laboratory investigations (that included routine hemogram, urine analysis and serum calcium, serum phosphorus, serum alkaline phosphates) that followed the radiologic identification of calcifications in AOT, eliminated an idea of the lesion having any association with systemic stimulation. AOT associated with dentigerous cyst of the maxilla has been reported, but not AOT and COF occurring concomitantly. However, the present case is unique in its concomitant presentation of follicular variant of AOT and central COF, associated with an impacted tooth occurring in the mandible.


   Conclusion Top


The association of impacted teeth and calcifications in both the lesions initially suggested the bilateral presence of a calcifying lesion at different stages of the same spectrum, i.e. earlier stage (radiolucent) on the right side, and mixed to radiopaque (matured) on the left side of the mandible. However, biopsy revealed 2 histologically distinct lesions. An occurrence of follicular AOT in an anterior mandible is rare. Calcifications were noted in both the lesions with greater amounts found in the COF. The exact cause of calcifications, and if there is any common stimulation for this, could not be elicited. The simultaneous occurrence of a central ossifying fibroma and a follicular variant of the adenomatoid odontogenic tumor in the mandible, in association with impacted teeth, are extremely rare.

 
   References Top

1.Chang CC, Hung HY, u-fongchang J, Yu CH, Wang YP, Liu BY, et al. Central ossifying fibroma: A Clinicopathologic study of 128 cases. J Formos Med Assoc 2008;107:288-94.  Back to cited text no. 1
    
2.Jayachandran S, Meenakshi R. Cemento-ossifying Fibroma. Indian J Dent Res 2004;15:35-9.  Back to cited text no. 2
    
3.Jayachandran S, Sachdeva S. Cemento-ossifying Fibroma of Mandible: Report of Two Cases. J Indian Acad Oral Med Radiol 2010;22:53-6.  Back to cited text no. 3
    
4.Sarwar HG, Jindal MK, Ahmad SS. Cemento-ossifying fibroma - a rare case. J Indian Soc Pedod Prev Dent 2008;26:128-31.   Back to cited text no. 4
    
5.Handschel JG, Depprich RA, Zimmermann AC, Braunstein S, Kübler NR. Adenomatoid odontogenic tumor of the mandible: review of the literature and report of a rare case. Head Face Med 2005;24:3.  Back to cited text no. 5
    
6.Yilmaz N, Acikgoz A, Celebi N, Zengin AZ, Gunhan O. Extrafollicular Adenomatoid Odontogenic Tumor of the Mandible: Report of a Case. Eur J Dent 2009;3:71-4.  Back to cited text no. 6
[PUBMED]    
7.Kuta AJ, Worley CM, Kaugars GE. Central Cementoossifying fibroma of the maxillary sinus: A review of six cases. AJNR Am J Neuroradiol 1995;16:1282-86.   Back to cited text no. 7
[PUBMED]    
8.Tamiolakis D, Thomaidis V, Tsamis I . Cementoossifying Fibroma of the Maxilla: a Case Report. Acta Stomat Croat 2005;319-21.  Back to cited text no. 8
    
9.Yoshida Michiko, Suzuki Masayuki, Kimura Shunsuke et al. A case of a cemento ossifying fibroma associated with multiple impacted teeth arising in the right maxillary bone. Japanese J Oral Maxillofac Surg 2006;52:588-91.  Back to cited text no. 9
    
10.Baberi A, Cappabianca S, Colella G. Bilateral cemento-ossifying fibroma of the maxillary sinus. Br J Radiol 2003;76:279-80.  Back to cited text no. 10
    

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Correspondence Address:
A Ravi Prakash
Department of Oral Maxillofacial Pathology, G. Pulla Reddy Dental College & Hospital, Kurnool
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.102255

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  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9]



 

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