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CASE REPORT
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Peripheral cemento-ossifying fibroma – A rare case report


 Department of Pediatric and Preventive Dentistry, Subharti Dental College and Hospital, Swami Vivekanand Subharti University, Meerut, Uttar Pradesh, India

Date of Submission22-Jun-2021
Date of Acceptance06-Sep-2021
Date of Web Publication06-Jan-2022

Correspondence Address:
Noopur Kaushik,
Room No. 3, Subharti Dental College and Hospital, Swami Vivekanand Subharti University, Subhartipuram, NH-58, Delhi-Haridwar Bypass Road, Meerut - 250 002, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jcrt.jcrt_1001_21

 > Abstract 


Peripheral cemento-ossifying fibroma (PCOF) is a relatively rare tumor classified between fibro-osseous lesions. PCOF is an osteogenic neoplasm that ordinarily presents as an epulis-like growth with uncertain pathogenesis. It predominantly affects young adults, with peak prevalence between the first and second decades of life. We present here the case report of a 15-year-old girl, which was previously surgically excised twice and had recurred both the times. The disease duration was 2 years and a follow-up of 1-year was done. Clinical, radiographic, and histological characteristics are discussed with recommendations regarding the treatment is provided.

Keywords: Cemento-ossifying fibroma, fibroma, gingival overgrowth, PCOF, pediatric dentistry, pedodontics



How to cite this URL:
Kaushik N, Srivastava N, Rana V, Suhane C. Peripheral cemento-ossifying fibroma – A rare case report. J Can Res Ther [Epub ahead of print] [cited 2022 Dec 4]. Available from: https://www.cancerjournal.net/preprintarticle.asp?id=335104




 > Introduction Top


Several localized reactive lesions may occur on the gingiva of an individual, including pyogenic granuloma, focal fibrous hyperplasia, peripheral giant cell granuloma, and peripheral cemento-ossifying fibroma (PCOF). Out of these, PCOF is thought to be a common growth on the gingiva which can be neoplastic or reactive in nature.[1] The fibro-osseous neoplasms, according to Waldron CA (1993), can be defined as “a demarcated and occasionally encapsulated lesion consisting of fibrous tissue containing variable amounts of mineralized material resembling bone and/or cementum.”[2]

Some PCOFs are thought to develop as pyogenic granuloma because of their clinical and histopathological similarities.[3] Trauma and various irritants such as bacterial plaque, calculus, ill-adapted crowns, irregular restorations, and orthodontic appliance are frequently associated with PCOF. The mineralized product probably originates from periosteal cells or from the periodontal ligament (PDL).[4]

PCOF accounts for approximately 3.1% of all oral tumors, and 9.6% of all gingival lesions.[5] Both the genders are affected by PCOF, but higher female predilection is seen, which can be due to the hormonal influence. Higher predilection is reported in Light skinned (71%) compared to Dark skinned (36%) in terms of racial occurrence.[6] With respect to age, PCOF exhibits peak incidence during the second and third decades of life. However, Neville et al.(1995)[7] stated that PCOF affects adolescents and young adults predominantly, with peak prevalence between 10 and 19 years of age.

Clinical features demonstrate sessile or pediculate nodular mass, originating usually in the interdental papilla.[3] The appearance in terms of color and consistency is similar to surrounding gingiva unless ulcerated. PCOF generally measures <2 cm in diameter, although lesions >10 cm have also been reported. In maxilla, about 60% of the tumor occurs and >50% of all cases affect incisors and canine region.[3] Tooth migration is also reported due to the existence of PCOF. Radiographically, underlying bone involvement is rare in majority of PCOF cases, but occasionally, superficial bone appears erosive.

Presented here is a rare case of PCOF in a 15-year-old female patient and emphasizes the importance of early diagnosis and recurrence of this tumor.


 > Case Report Top


A 15-year-old girl child was presented to the Department of Pediatric and Preventive Dentistry, with the chief complaint of a tooth-like tissue overgrowth in the upper left front tooth region for 2 years. Detailed dental history revealed that tooth no. 22 was extracted 2.5 years back because of carious involvement and malformed tooth after which the tissue growth was noticed. The tissue was excised twice by the local dentist, but recurrence occurred both the times. The intraoral examination revealed a well-circumscribed, erythematous sessile growth on the maxillary left lateral incisor region, measuring about 0.8 mm × 0.5 mm [Figure 1] and [Figure 2], which was firm and nontender on palpation. The overlying mucosa appeared erythematous. Intraoral periapical radiograph revealed a radio-opaque mass of tissue in that region which was less dense than dentin.
Figure 1: Preoperative; frontal view

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Figure 2: Preoperative; occlusal view

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The patient underwent complete blood investigations which revealed that all the readings were within the normal limits. Since the growth was localized, surgical excision of the tissue using electrocautery was planned.

The procedure was then explained to the patient as well as her mother in their own language and informed consent was obtained. Scaling was done before the surgical excision. Under local anesthesia, the tissue was secured and tied using suture material. A needle electrode was used on the electrosurgical unit (Unicorn Denmart) to excise the tissue. The surrounding PDL and periosteum were then aggressively curettaged to prevent recurrence. The charred surgical site acts as a surgical bandage postoperatively. The patient was recalled after 1 week for follow-up.

The excised tissue was sent for histopathological examination. The histopathological report showed ulcerated parakeratinized stratified squamous epithelium with an underlying cellular connective tissue stroma. The stroma showed dense, plump fibroblasts, blood capillaries, and few calcified basophilic masses suggestive of cementum-like tissue and few bone trabeculae in the deeper areas [Figure 3]. Focal chronic inflammatory cells and dense collagen fiber bundles were also seen which confirmed the diagnosis of PCOF.
Figure 3: Histopathology revealed parakeratinized stratified squamous epithelium with an underlying cellular connective tissue stroma

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After 1 month, a removable partial denture was delivered to the patient to meet the esthetic requirement. The patient was asymptomatic at 1-year follow-up with no evidence of recurrence [Figure 4] and [Figure 5]. The patient has been kept at regular follow-up of every 6 month for any recurrence.
Figure 4: One-year follow-up; frontal view

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Figure 5: One-year follow-up; occlusal view

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 > Discussion Top


Since the 1940s, PCOF has been described in the literature. Many names such as epulis, peripheral cementifying fibroma, peripheral fibroma with calcifications, calcifying fibroblastic granuloma, peripheral ossifying fibroma, peripheral fibroma with cementogenesis, and PCOF have been described in the literature.

Benign fibroosseous lesions of the jaws present problems in diagnosis and classification. The 1992 WHO classification groups Benign fibroosseous lesions of the jaws under a single designation as cemento-ossifying fibroma with two histologic types - cementifying fibroma and ossifying fibroma, that may be clinically and radiographically undistinguishable.[8]

Although the etiopathogenesis of PCOF remains uncertain, there exist two schools of thoughts among the investigators, some consider it as a neoplastic process, while others consider it as a reactive process. In either case, PCOF is believed to originate from the cells of the PDL.[1]

Exclusive occurrence in the gingiva, its proximity to PDL, and the occurrence of oxytalan fibers in the mineralized matrix can be the possible reason for PCOFs periodontal origin. The reason could further be attributed As a response to gingival irritation, or gingival injury, Presence of subgingival calculus, in which the mature fibrous connective tissue proliferates rapidly. Chronic irritation to the periosteum and PDL membrane results in the formation of bone or dystrophic calcification due to metaplasia of connective tissue. Consequently, it has been suggested that PCOF can be caused by fibrosis of granulation tissue.[9]

Gardner (1982)[10] suggested the term PCOF which on histological examination shows parakeratinized and hyperplastic epithelium and well-cellularized connective tissue comprising mineralized elements extending from bone to cementum and dystrophic calcifications to a lesser extent.[3] PCOF may be misdiagnosed as pyogenic granuloma, PCOF, epulis granulomatosum, peripheral giant cell lesion, etc. [Table 1].
Table 1: Differential diagnosis of peripheral cemento-ossifying fibroma

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Pyogenic granuloma bleeds on slight manipulation and presents with red soft friable nodule, but it neither causes displacement of tooth nor the resorption of alveolar bone. On the other hand, peripheral giant cell granuloma presents with similar features to that of PCOF, but PCOF lacks blue discoloration and shows flakes of calcification.[11] Eversole and Rovin[12] mentioned in their article about similar sex and site predilection of pyogenic granuloma. Gardner stated that peripheral ossifying fibroma of cellular connective tissue is so characteristic that a histologic diagnosis can be made with confidence, regardless of the presence or absence of calcification. Therefore, the diagnosis of PCOF based only on clinical or radiographic presentation is difficult. The importance of histopathological examination to confirm the diagnosis of PCOF was highlighted by Orkin and Aimadas (1984),[4] which clinically is relatable to pregnancy tumor, inflammatory hyperplasia, epulis fibrosa, or peripheral and central giant cell granuloma. The PCOF must be differentiated from the peripheral odontogenic fibroma (PODF) described by the World Health Organization. Histologically, the PODF has been defined as a fibroblastic neoplasm containing odontogenic epithelium.[13]

A histopathologic evaluation leads to the confirmatory diagnosis of PCOF of the biopsy specimens. The microscopic examination observes the following features:

  1. Stratified squamous surface epithelium ulcerated
  2. Varying numbers of fibroblasts and benign fibrous connective tissue
  3. Endothelial proliferation (sparse to profuse)
  4. Mineralized material consisting of mature, lamellar, or woven osteoid, cementum-like material, or dystrophic calcifications
  5. Inflammatory cells in lesion (acute or chronic).


Treatment of PCOF includes a variety of options such as removal of etiological factors, scaling, and surgical excision of the lesion. Many authors have further suggested excision of involved PDL and periosteum to minimize the possibility of recurrence of incompletely removed lesions[14] and can be considered as a definitive treatment modality.[15]

If correctly managed, excellent prognosis and rare recurrence are seen.[2] However, the recurrence rate of PCOF has been reported from 8% to 20%,[16] due to the partial removal of the lesion, repeated injury, or persistence of local irritants. Follow-up is recommended to check possible recurrences.[17] Any irritant factors such as calculus, an ill-fitting orthodontic appliance, or faulty restorations should be removed.

Peripheral cemento-ossifying fibroma PCOF is a slowly progressing lesion. Histopathological examination is essential for accurate diagnosis. Due to the high recurrence rate of the lesion, postoperative follow-up is necessary. In addition, planned surgery of these lesions results not only in eliminating etiological factors but also results in good esthetics without any deformity.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 > References Top

1.
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. 1
    
2.
Waldron CA. Fibro-osseous lesions of the jaws. J Oral Maxillofac Surg 1993;51:828-35.  Back to cited text no. 2
    
3.
Delbem AC, Cunha RF, Silva JZ, Soubhia AM. Peripheral cemento-ossifying fibroma in child. A follow-up of 4 years. Report of a case. Eur J Dent 2008;2:134-7.  Back to cited text no. 3
    
4.
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. 4
    
5.
Walters JD, Will JK, Hatfield RD, Cacchillo DA, Raabe DA. Excision and repair of the peripheral ossifying fibroma: A report of 3 cases. J Periodontol 2001;72:939-44.  Back to cited text no. 5
    
6.
Sachdeva SK, Mehta S, Sabir H, Rout P. Peripheral cemento-ossifying fibroma with uncommon clinical presentation: A case report. Odovtos-Int J Dent Sc 2018;20:17-23.  Back to cited text no. 6
    
7.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. Philadelphia: W.B. Saunders; 1995.  Back to cited text no. 7
    
8.
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. 8
    
9.
Kendrick F, Waggoner WF. Managing a peripheral ossifying fibroma. ASDC J Dent Child 1996;63:135-8.  Back to cited text no. 9
    
10.
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. 10
    
11.
Mishra AK, Maru R, Dhodapkar SV, Jaiswal G, Kumar R, Punjabi H. Peripheral cemento-ossifying fibroma: A case report with review of literature. World J Clin Cases 2013;1:128-33.  Back to cited text no. 11
    
12.
Eversole LR, Rovin S. Reactive lesions of the gingiva. J Oral Pathol 1972;1:30-8.  Back to cited text no. 12
    
13.
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. 13
    
14.
Guru SR, Singh SS, Guru RC. Peripheral cemento-ossifying fibroma: A report of two cases. J Health Sci Res 2016;7:71-5.  Back to cited text no. 14
    
15.
Kaur T, Dhawan A, Bhullar RS, Gupta S. Cemento-ossifying fibroma in maxillofacial region: A series of 16 cases. J Maxillofac Oral Surg 2019;20:240-5.  Back to cited text no. 15
    
16.
Cuisia ZE, Brannon RB. Peripheral ossifying fibroma--a clinical evaluation of 134 pediatric cases. Pediatr Dent 2001;23:245-8.  Back to cited text no. 16
    
17.
Aburas S, Bandura P, Al-Ibraheem A, Berger S, Meier M, Turhani D. A large maxillary cemento-ossifying fibroma superimposed with solitary bone cyst documented over 18 years: A case report. Int J Surg Case Rep 2020;68:257-62.  Back to cited text no. 17
    


    Figures

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

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