Recurrent peripheral cemento-ossifying fibroma.

Peripheral cement-ossifying fibroma (PCOF) is a rare osteogenic neoplasm that ordinarily presents as an epulis-like growth. It frequently occurs in maxillary anterior region in teenagers and young adults. We report a case of PCOF in a 42-year-old male, which was previously surgically excised and recurred after a period of 2 years. PCOF should be considered in the differential diagnosis of reactive hyperplastic lesions originating from gingiva. Hence, early diagnosis with proper surgical excision and aggressive curettage of the adjacent tissues is essential for prevention of recurrence.

INTRODUCTION

Many types of localized reactive lesions may occur on gingiva, including focal fibrous hyperplasia, pyogenic granuloma, peripheral giant cell granuloma and peripheral cement-ossifying fibroma (PCOF)[1]

Peripheral cement-ossifying fibroma being a common gingival growth, it is thought to be either reactive or neoplastic in nature.[2] It is widely accepted that this lesion may originate from the cells of periodontal ligament,[23456] and is often associated with irritants such as calculus, dental appliances, poor quality dental restorations, plaque or may occur as a result of trauma.[34567] The close proximity to the periodontal tissues has made the term periodontoma a colloquial term used.[8] PCOFs are sessile or pedunculated, usually ulcerated, erythematous or may exhibit a color similar to the surrounding gingiva.[9] Most of these lesions are <2 cm in size, although lesions >10 cm are occasionally observed. Female predilection as high as 4.3:1 has been reported with a peak incidence in the second decade and a declining incidence in later years.[3]

Diagnosis of PCOF based only on the clinical history is difficult and histopathological evaluation of the surgical specimen is necessary for an accurate diagnosis. The recurrence rate of PCOF is considered to be high and is reported at 9%,[7] 14%[4] and 20%[2] and hence regular follow-up is mandatory. We present here a recurrent case of PCOF and also highlight the importance of early diagnosis with proper surgical excision and aggressive curettage of the adjacent tissues to prevent its recurrence.

CASE REPORT

A 42-year-old male patient reported to our dental clinic at Airoli, Navi Mumbai with complaint of a growth on his maxillary gingiva since 8 months. The patient gave a history of a similar growth 2 years back, which was then excised by a dentist but was not histopathologically evaluated. His past medical history was noncontributory. He gave a history of chewing tobacco 8–10 times/day. Occasionally, bleeding occurred when he brushed his teeth. The patient was concerned about the pathogenesis of the lesion since it had recurred once again and had discussed the possibility of the lesion being a malignancy. This had raised the patient's anxiety level considerably.

Intra-oral examination revealed a well circumscribed erythematous growth between maxillary right premolars, measuring approximately 1.2 cm × 1 cm in diameter and originating from the interdental papilla [Figure 1]. It was firm in consistency and nontender. Oral hygiene was poor. No other marked deformity was noted extraorally or intraorally. Intra-oral periapical radiograph in relation to the maxillary right premolars no showed no abnormality.

Figure 1

Intra-oral photograph showing growth between the maxillary right bicuspids

Clinical differential diagnosis included PCOF, peripheral giant cell granuloma, traumatic fibroma and pyogenic granuloma. The patient underwent complete blood investigation prior to surgery. Under local anesthesia, the lesion was completely excised with aggressive curettage of surrounding tissue. The excisional biopsy specimen was sent for histopathological examination.

Histopathological examination of the lesion revealed a parakeratinized stratified squamous epithelium with long and slender rete ridges. Underlying connective tissue was fibrocellular with calcifications. Cellular areas comprised of proliferating plump fibroblasts with trabecular bone lined by osteoblasts. Many round to oval hematoxoyophilic calcified matrix were seen resembling cementum like material. A final diagnosis of PCOF was made [Figure 2]. An 8 months postsurgical follow-up showed no evidence of recurrence [Figure 3].

Figure 2

Photomicrograph showing bony trabeculae along with cementum like material below the surface epithelium. (H and E, ×10)

Figure 3

Eight months postoperative intra-oral photograph

DISCUSSION

Intra-oral ossifying fibromas have been discussed in the literature since the 1940s. Epulis, peripheral fibroma with calcifications, calcifying fibroblastic granuloma, peripheral cementifying fibroma, peripheral ossifying fibroma, peripheral fibroma with cementogenesis and PCOF are few of the many names given to similar lesions.[23] The number of names used for fibroblastic gingival lesions indicates that there is a lot of controversy surrounding the classification of these lesions.[2]

Although the etiopathogenesis of PCOF is uncertain, some investigators consider it as a neoplastic process while others call it a reactive process. Anyway, the lesion is thought to arise from the cells of the periodontal ligament due to trauma or local irritational factors such as plaque or ill-fitting or poor quality dentures.[234] In the present case the etiology could be local irritational factors such as plaque or calculus.

Hormonal influences may play a role too, as these lesions have shown a female predilection, with an increased occurrence in the second decade and a declining incidence after the third decade. In a case published by Kumar et al., the presence of multicentric PCOF at an edentulous site of a 49-year-old woman raises a question regarding the pathogenesis of this lesion.[2]

Peripheral cement-ossifying fibroma is considered to arise from the cells of the periodontal ligament. As suggested by Kumar et al. in 2006, this may be due to the proximity of the gingiva to the periodontal ligament and the presence of oxytalon fibers within the mineralized matrix of the same lesions.[2] Excessive proliferation of the mature matrix, connective tissue is a response to gingival injury, subgingival plaque or calculus or a foreign body in the gingival sulcus. Furthermore, chronic irritation of the periosteal and periodontal membrane causes metaplasia of the connective tissue, which could initiate the formation of bone or dystrophic calcifications. It is therefore suggested that this lesion may be caused by fibrosis of granulation tissue.[10]

With respect to race, there is a predominance in whites (71%), as compared to blacks (36%).[11] It may occur at any age but a peak incidence is seen in the second and third decade. Approximately, 60% of the PCOFs occur in the maxilla, and they occur more often in the anterior than in the posterior area with 55–60% presenting in the incisor-cuspid region.[347]

In the present case, the patient was a 42-year-old male patient with the lesion seen in the maxillary bicuspid region.

Radiographically, PCOF may follow different patterns depending upon the amount of mineralized tissue. They may show radio opaque foci of calcification, but not all lesions exhibit radiographic characteristics. Radiological findings were noncontributory in the present case.

A diagnosis of PCOF is made by histopathological evaluation of the biopsy specimen. The features observed during microscopic evaluation are a benign fibrous connective tissue stroma with a varying content of fibroblasts, myofibroblasts, and collagen. There is sparse to profuse endothelial cell proliferation. Mineralized material varies in the form of lamellar or woven osteoid to cementum like material or dystrophic calcification.[2] Most of these features were present in the present case.

Treatment consists of conservative surgical excision and scaling of adjacent teeth.[1] The recurrence rate of PCOF is high and is probably due to incomplete removal of the lesion, repeated injury or persistence of local irritants.[4] The rate of recurrence has been reported to vary from 8.9% to 20% (Bhaskar and Jacoway 1966; Kenny et al. 1989; Eversole and Rovin 1972).

Recurrence in the present case was probably due to previous incomplete removal of the lesion and the presence of local irritants such as plaque and calculus.

CONCLUSION

PCOF is a slow progressive lesion generally with limited growth. Many cases will progress for long periods before the patient seeks treatment because of lack of symptoms associated with the lesion. It is possible to misdiagnose PCOF from other reactive lesions arising from the gingiva. Therefore, histopathological examination is essential for an accurate diagnosis and for proper management. We describe a case of PCOF in a 42-year-old male, which recurred probably due to incomplete surgical removal along with the presence of local irritating factors. Complete excision with aggressive curettage of the adjacent tissues is essential to prevent any recurrence. Close postoperative follow-up is required because of the growth potential and 8–20% recurrence rate.