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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 2  |  Page : 204-208

Peripheral cemento-ossifying fibroma: Report of a case in an elderly patient


Department of Oral Medicine and Radiology, Saraswati Dental College and Hospital, Lucknow, Uttar Pradesh, India

Date of Submission24-Jul-2014
Date of Acceptance17-Oct-2014
Date of Web Publication30-Oct-2014

Correspondence Address:
Shruti Sinha
Department of Oral Medicine and Radiology, Saraswati Dental College and Hospital, Faizabad Road, Tiwari Ganj, Lucknow, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.143703

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   Abstract 

The peripheral cemento-ossifying fibroma (PCOF) is an osteogenic neoplasm and is believed to arise from the periodontal ligament. It accounts for 3.1% of all oral tumors and 9.6% of all gingival lesions. It frequently occurs in the maxillary anterior region in teenagers and young adults, and has a female predilection. Trauma, local irritation due to calculus, ill-fitting dentures and faulty restorations, and hormonal disturbances are the known predisposing factors. An early diagnosis and surgical excision is required to prevent its recurrence. Here we present a case of PCOF with a history of just two months, occurring in an elderly female involving the edentulous mandibular posterior region.

Keywords: Cementifying fibroma, gingival overgrowth, ossifying fibroma, osteogenic tumor, peripheral cemento-ossifying fibroma


How to cite this article:
Sinha S, Reddy Enja SP, Chandra S, Nethan S. Peripheral cemento-ossifying fibroma: Report of a case in an elderly patient . J Indian Acad Oral Med Radiol 2014;26:204-8

How to cite this URL:
Sinha S, Reddy Enja SP, Chandra S, Nethan S. Peripheral cemento-ossifying fibroma: Report of a case in an elderly patient . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2020 Jul 10];26:204-8. Available from: http://www.jiaomr.in/text.asp?2014/26/2/204/143703


   Introduction Top


Fibro-osseous lesions are a diverse group of processes that are characterized by replacement of normal bone by fibrous tissue containing a newly formed mineralized product. Fibro-osseous lesions of the jaw include developmental (hamartomatous) lesions, reactive or dysplastic processes, and neoplasms. [1] According to the 1992 World Health Organization (WHO) classification of fibro-osseous lesions, cemento-ossifying fibroma (cementifying fibroma, ossifying fibroma) is considered to be an osteogenic neoplasm, with a significant growth potential. [2] It is usually associated with irritant agents such as calculus or bacterial plaque on teeth, orthodontic appliances, ill-adapted crowns, and irregular restorations. It accounts for 3.1% of all oral tumors and 9.6% of all gingival lesions. It is commonly seen in female adolescents and young adults (peak age range is between 10 and 19 years) as a common gingival growth in the vicinity of the interdental papilla of the maxillary incisors or canines. It appears as a nodular mass, either pedunculated or sessile. The color ranges from red to pink and the surface is frequently, but not always ulcerated. [3],[4] The lesion is usually present for many months to years before it is diagnosed. The diagnosis is difficult on the basis of clinical features; therefore, a histopathological examination is mandatory for its confirmation. [5]


   Case Report Top


A 60-year-old female patient reported with a complaint of a rapidly enlarging growth in the right back region of the lower jaw, since two months. The growth developed spontaneously from a small asymptomatic peanut-sized swelling, following exfoliation of periodontally compromised teeth from the same region, on its own. The patient did not give any other dental or medical history. No significant findings were noted on general examination and systemic evaluation of the patient. Also, the extraoral examination was noncontributory, except for right submandibular lymphadenopathy. Intraoral examination revealed a single sessile growth, approximately 1.5 × 1 cm in diameter, roughly ovoid in shape, and of normal mucosal color, in the 45 and 46 region. The overlying mucosa was smooth buccally and showed indentations of the maxillary teeth on its lingual aspect. There was no visible pulsation or discharge. There was mild tenderness on palpation and the swelling was compressible and firm in consistency [Figure 1]. Tooth numbers 45 and 46 were missing in the mandibular right posterior region. There was generalized bone loss, suggestive of generalized periodontitis. A provisional diagnosis of irritation fibroma was made and pyogenic granuloma, peripheral giant cell granuloma, and peripheral odontogenic fibroma were considered as the differential diagnoses.

Radiographic examination (intraoral periapical radiograph and orthopantomograph) revealed a well-defined mixed radiolucent - radiopaque mass, with a prominent radiopaque area of calcified material in the center of the mass [Figure 2]. A radiographic diagnosis of peripheral ossifying fibroma was made (even though a history of short duration and age of the patient were not in favor).
Figure 1: Intraoral photograph showing growth

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Figure 2: Radiographs (OPG and IOPA) showing a well-defi ned, mixed, radiolucent — radiopaque mass, with centrally located and relatively dense radiopaque areas

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The lesion was completely excised [Figure 3] under local anesthesia and was radiographed again, in order to obtain a clearer radiographical picture of the mass, without any interference from the overlapping shadows of the adjacent soft and hard tissues of the oral cavity. The radiograph of the excised specimen revealed a relatively radiopaque mass with a dense calcified material at the center [Figure 4]. Subsequently, a histopathological examination of the specimen was performed.
Figure 3: Excised specimen measuring about 1.5 × 1 cm in diameter

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Figure 4: Radiograph of the excised specimen showing a relatively radiopaque mass (dense calcified material) at the center

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The histopathological examination of the lesion revealed hyperkeratinized, stratified squamous epithelium overlying the moderately dense to dense cellular connective tissue stroma, which consisted of plump to spindle-shaped fibroblasts, arranged in fascicles, at focal areas. Interconnected trabeculae of lamellar bone surrounded by osteoid and areas of woven bone, along with endothelial lined blood vessels were appreciated [Figure 5]. Based on the clinical, radiographical, and histopathological findings, a final diagnosis of peripheral cemento-ossifying fibroma (PCOF) was made. The patient is on periodic postoperative follow-up since eight months. Healing was uneventful and no recurrence has been noted [Figure 6].
Figure 5: Photomicrograph showing cellular connective tissue stroma
consisting of plump to spindle-shaped fi broblasts arranged in fascicles
with interconnected trabeculae of lamellar bone surrounded by osteoid
areas of woven bone (H and E, 10x)


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Figure 6: Postoperative photograph

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   Discussion Top


The term 'peripheral cemento-ossifying fibroma' was coined by Montgomery in 1927. [4] However, a lot of confusion regarding the terminology has persisted, as a result of which numerous synonyms have been used in the past, namely, peripheral cementifying fibroma, ossifying fibroepithelial polyp, peripheral fibroma with osteogenesis, peripheral fibroma with cementogenesis, peripheral fibroma with calcification, calcifying or ossifying fibroma epulis, and calcifying fibroblastic granuloma. Also, the term cemento-ossifying has been considered inappropriate, because the clinical presentation and histopathology of the cemento-ossifying fibroma are the same in areas where there is no cementum, such as, the skull, femur, and tibia, which are all ossifying fibromas. Hence, those occurring in the jaws should not be termed cemento-ossifying fibromas merely based on their presence in the vicinity of teeth. The term cemento-ossifying fibroma is given mainly due to the presence of dysmorphic round basophilic bone particles within the ossifying fibroma, called cementicles, which in reality are not from the cementum, but instead represent a dysmorphic product of this tumor, similar to the keratin pearls seen in squamous cell carcinoma. [3],[6]

Peripheral cemento-ossifying fibroma is considered as an osteogenic neoplasm (WHO 1992 classification), frequently associated with irritant agents, such as, calculus, bacterial plaque, orthodontic appliances, ill-adapted crowns and irregular restorations, and hormonal disturbances. [3],[4],[5] Although its origin is uncertain, it is said to originate from cells of the periodontal ligament due to the following reasons: Exclusive occurrence in the gingiva (interdental papilla), the proximity of the gingiva to the periodontal ligament and the presence of oxytalan fibers within the mineralized matrix of some lesions. [3],[7] It is assumed that local factors such as plaque and calculus played an etiological role in this particular case.

It accounts for 3.1% of all oral tumors and for 9.6% of all gingival lesions. It commonly occurs in young adults and adolescents, and has a female predilection, due to hormonal influences, within the peak age range of 10-19 years. [3] Peripheral cemento-ossifying fibromas show increasing occurrences in the second decade and declining incidence after the third decade, which was contradictory in our case, as the patient was aged 60 years at the time of reporting. Very few cases in the elderly have been reported to date, for example, studies by Bhasin et al., [4] Dalghous et al., [5] and Mohiuddin et al. [8]

The site of occurrence is the interdental papilla of the maxillary incisor - canine region, [3] whereas, in the present case, the lesion was observed in the right mandibular second premolar - first molar edentulous alveolar ridge region, which is a less common site. However, similar cases with lesions in the mandibular region have been reported by Yadav et al. [9] and Passos et al. [10] and those in edentulous regions have been reported by Kumar et al. [7] and Yokoyama et al. [11]

The peripheral cemento-ossifying fibroma clinically presents as a pedunculated or sessile exophytic mass, about <2 cm in diameter (occasionally >10 cm), with a color similar to that of the mucosa, unless the surface is ulcerated. It may be associated with migration of the adjacent teeth. [12] It is usually a slow growing mass that takes many months and years to reach a diagnosable stage, as opposed to the history of a short duration, of merely two months, given by our patient and her family, which is quite unexpected. [13] Cases with such short histories of approximately three months of duration of the tumor have been reported by Satish et al., [14] Sudhakar et al. [15] and Trasad et al. [16]

Radiographically, PCOF exhibits a well-defined, mixed, radiolucent - radiopaque lesion, along with calcified material. In some instances it may appear almost totally radiolucent with just a hint of calcified material. Others may show a wispy or flocculent pattern. Radiographs of the present case revealed a well-defined, mixed, radiolucent - radiopaque mass, with a prominent radiopaque area of calcified material at the center of the mass.

When presented clinically with a gingival lesion, it is important to establish a differential diagnosis, which in this case can be irritation fibroma, pyogenic granuloma or peripheral giant cell granuloma. As the clinical appearance of these various lesions can be remarkably similar, classification is based on their distinct histological differences. [3] The PCOF must be differentiated from the peripheral odontogenic fibroma (PODF) described by the WHO. [3],[17],[18] Histologically, the PODF has been defined as a fibroblastic neoplasm containing an odontogenic epithelium. [3],[19] Despite a preponderance of literature supporting the differentiation, some authors continue to argue that the PCOF is the peripheral counterpart of the central cemento-ossifying fibroma. [3],[20]

In PCOF, histopathologically, either an intact or ulcerated stratified squamous epithelium is seen overlying the highly cellular connective tissue, comprising of fibroblasts, with a central area of calcification, which may consist of bone, cementum-like material, dystrophic calcification or a combination of all these. Histopathological analysis of the present case showed moderately dense to dense cellular connective tissue stroma consisting of plump to spindle-shaped fibroblasts arranged in fascicles at focal areas, with interconnected trabeculae of lamellar bone surrounded by osteoid and areas of woven bone.

The management consists of an elimination of the associated irritating factors, oral prophylaxis, and total aggressive surgical excision, along with the involved periodontal ligament, and periosteum, to minimize the possibility of recurrence, as it has a high recurrence rate of approximately 8-20%. Hence, long-term postoperative follow-up is also essential. [3] The patient reported here, is on periodic postoperative follow-up since eight months and has shown no recurrence till the date of reporting.


   Conclusion Top


The cemento-ossifying fibroma is a relatively rare lesion and is considered as an osteogenic tumor (nonodontogenic) with variable expressiveness. It is defined as a well-demarcated and occasionally encapsulated lesion consisting of fibrous tissue containing variable amounts of mineralized material resembling bone (ossifying fibroma), cementum (cementifying fibroma), or both. [3],[5]

The PCOF shows increasing occurrences in the second decade and declining incidence after the third decade, which is contradictory in our case, as the patient was aged 60 years at the time of reporting. It is usually a slow-growing mass, which takes many months and years to reach a diagnosable stage, as opposed to the history of a short duration of merely two months given by our patient, which is quite unexpected. The common site of occurrence is the interdental papilla of the maxillary incisor - canine region, [1] whereas, in the present case, the lesion has been observed in the right mandibular second premolar - first molar edentulous alveolar ridge region, which is a less common site. Radiographically, a well-defined, mixed, radiolucent - radiopaque mass, with a prominent radiopaque area of calcified material in the center of the mass was evident, which was rare in cases with such a short history (two months, in the present case). Thus, in the present case report, we have described a unique case of PCOF in a 60-year-old female, with uncommon features.


   Acknowledgment Top


We thank the Department of Oral Pathology and Microbiology, Saraswati Dental College and Hospital, Lucknow, for lending us the histopathological images.

 
   References Top

1.
Neville BW, Damm DD, Allen CM, Bouquot J. Oral and maxillofacial pathology. 3 rd ed. St. Louis, Missouri: Saunders Elsevier; 2009. p. 521-2.  Back to cited text no. 1
    
2.
Rajpal K, Agarwal R, Chhabra R, Bhattacharya M. Updated classification schemes for fibro-osseous lesions of the oral and maxillofacial region: A review. IOSR J Dent Med Sci 2014;13:99-103.  Back to cited text no. 2
    
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Verma E, Chakki AB, Nagaral SC, Ganji KK. Peripheral cemento-ossifying fibroma: Case series literature review. Case Rep Dent 2013;2013:930870.  Back to cited text no. 3
    
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Bhasin M, Bhasin V, Bhasin A. Peripheral ossifying fibroma. Case Rep Dent 2013;2013:497234.  Back to cited text no. 4
    
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Dalghous A, Alkhabuli JO. Cemento-ossifying fibroma occurring in an elderly patient. A case report and a review of literature. Libyan J Med 2007;2:95-8.  Back to cited text no. 5
    
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Marx RE, Stern D. Oral and maxillofacial pathology: A rationale for diagnosis and treatment. 2 nd ed. Illinois: Quintessence; 2012. p. 879.  Back to cited text no. 6
    
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Kumar SK, Ram S, Jorgensen MG, Shuler CF, Sedghizadeh PP. Multicentric peripheral ossifying fibroma. J Oral Sci 2006;48:239-43.  Back to cited text no. 7
    
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Mohiuddin K, Priya NS, Ravindra S, Murthy S. Peripheral ossifying fibroma. J Indian Soc Periodontol 2013;17:507-9.  Back to cited text no. 8
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Yadav A, Mishra MB. Peripheral cemento-ossifying fibroma of mandible: A case report. Indian J Stomatol 2011;2:193-6.  Back to cited text no. 9
    
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Passos M, Azevedo R, Janini ME, Maia LC. Peripheral cemento-ossifying fibroma in a child: A case report. J Clin Pediatr Dent 2007;32:57-9.  Back to cited text no. 10
    
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Yokoyama Y, Matsui Y, Nagumo M, Irie T. Peripheral cemento-ossifying fibroma. Asian J Oral Maxillofac Surg 2007;19:160-2.  Back to cited text no. 11
    
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Orkin DA, Amaidas VD. Ossifying fibrous epulis. An abbreviated case report. Oral Surg Oral Med Oral Pathol 1984;57:147-8.  Back to cited text no. 12
    
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Sah K, Kale AD, Hallikerimath S, Chandra S. Peripheral cemento-ossifying fibroma: Report of a recurrence case. Peripheral cemento-ossifying fibroma: Report of a recurrence case. Contemp Clin Dent 2012;3(Suppl 1):S23-5.  Back to cited text no. 13
    
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Satish BN, Kumar P. Peripheral ossifying fibroma of hard palate - A case report. Int J Dent Clin 2010;2:30-4.  Back to cited text no. 14
    
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Sudhakar S, Praveen Kumar B, Prabhat MP. Peripheral ossifying fibroma. Online J Health Allied Sci 2009;8:17.  Back to cited text no. 15
    
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Trasad VA, Devarsa GM, Subba Reddy VV, Shashikiran ND. Peripheral ossifying fibroma in the maxillary arch. J Indian Soc Pedod Prev Dent 2011;29:255-9.  Back to cited text no. 16
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Gardner DG. The peripheral odontogenic fibroma: An attempt at clarification. Oral Surg Oral Med Oral Pathol 1982;54:40-8.  Back to cited text no. 17
    
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Kenney JN, Kaugars GE, Abbey LM. Comparison between the peripheral ossifying fibroma and peripheral odontogenic fibroma. J Oral Maxillofac Surg 1989;47:378-82.  Back to cited text no. 19
    
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Feller L, Buskin A, Raubenheimer EJ. Cemento-ossifying fibroma: Case report and review of the literature. J Int Acad Periodontol 2004;6:131-5.  Back to cited text no. 20
    


    Figures

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



 

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