Idiopathic peripheral ossifying fibroma in a young adolescent girl: A very rare clinical presentation.

The use of the term "fibroma" for any soft-tissue lesion or gingival lesion by general practitioners has led to inadequate diagnosis of quite a few rare entities. The occurrence of gingival lesions in adolescent female patients is a routine clinical finding. The site of occurrence of such a lesion is of prime significance, as the rarity of these lesions is determined by the site and size of the lesion. On the other hand, the dilemma over the diagnosis of peripheral ossifying fibroma (POF) versus peripheral odontogenic fibroma still continues. Commonly used synonyms for POF include calcifying fibroblastic granuloma, peripheral fibroma with calcification, peripheral cementifying fibroma, and calcifying or ossifying fibrous epulis. The present case report deals with the management of a unique case of POF between two maxillary central incisors in an adolescent female child patient, followed up to 2-year postsurgical excision.

INTRODUCTION

The occurrence of gingival lesions in young female adolescents is a routine clinical finding, but the site of such lesions determines their rarity. The extensive use of the term “fibroma” with regard to the diagnosis of a swelling or lump on the oral mucosa and gingiva has often being misleading for young dentists. The term “epulis” is used for smaller, more vascular and less fibrous gingival lesions, more frequently by specialists in periodontology or experienced general practitioners. However, the precise diagnosis of peripheral ossifying fibroma (POF) versus peripheral odontogenic fibroma in young children is often being debated since a long time. POF is a relatively uncommon, reactive gingival overgrowth usually composed of cellular fibroblastic tissue containing one or more mineralized tissues, namely, bone, cementum-like material, or dystrophic calcification. The origin of the lesion in most of these cases is from the interdental papilla, which is often intimately related to inflammatory fibrous hyperplasia.[1] In the present case report, a very rare case of POF in an adolescent female patient is discussed, which had grown to greater proportions, because of its nonaggressive, symptom-free behavior ultimately leading to an esthetic, unpleasant facial appearance with incompetent lips, and forcing the patient to undergo immediate treatment.

CASE REPORT

A 15-year-old teenage girl had reported to our department with a chief complaint of an unusual swelling in the anterior maxillary region with lip incompetency [Figure 1]. The history revealed that the gingival growth has increased in size rapidly in the past 6-month period with no visible clinical etiologic factor. The teeth around the lesion were vital, and the responses to vitality tests were positive.

Figure 1

The preoperative image of the lesion obliterating the labial frenum causing displacement of incisors

Clinically, the lesion appeared as an elevated, oval-shaped mass of 24 mm × 20 mm in dimension with a smooth, shiny surface without any bleeding or ulceration. The lesion had well-defined margins, as it was originating from labial gingival in relation to maxillary central incisors. The size of the lesion had increased to greater proportions, causing displacement of the incisors. On palpation, the lesion was firm, nodular, nontender, sessile, and noncompressible but resilient and without any surface bleeding, purulence, or ulceration. Radiographically, there was no definite radiolucency in relation to both the central incisors. Orthopantomogram revealed no signs of bony involvement.

The lesion was exposed, in an attempt to preserve the overlying mucosa [Figure 2]. Initially, it was dissected using a scalpel, and the underlying lesion was later excised involving the base, exposing the bony surface [Figure 3]. Finally, after complete debridement, the overlying mucosa was sutured with black silk [Figure 4]. The excised specimen was then sent for histopathological evaluation to the department of pathology [Figure 5]. Histopathological report of the lesion revealed fibrous connective tissue containing bundles of collagen fibers and plump fibroblasts. Few areas revealed calcifications, irregularly-shaped trabeculae, and droplets of basophilic material [Figure 6]. There was no hint of any tissue resembling cementum-like material.

Figure 2

The image shows the thick periosteal layer covering the nodular mass

Figure 3

The image shows the site of the excised lesion after surgical excision exposing bare bone

Figure 4

The immediate postoperative image after suturing with black silk

Figure 5

The specimen of the excised lesion measured with a metal scale

Figure 6

The histopathological picture of the biopsy specimen showing fibrous connective tissue, areas of calcifications and irregularly shaped trabeculae

Based on the clinical and histopathological findings, the case has been diagnosed as POF. After 2 years of follow-up, there was no recurrence of the lesion at the excised site and also in other areas in the oral cavity. The healing of the gingiva and oral mucosa was normal, without any gingival defects.

DISCUSSION

POF was first reported by the Shepherd in 1844 as alveolar exostosis.[2] The term “POF” was coined by Eversol and Rovin in 1972.[3] It occurs exclusively on the gingiva, usually arising from the interdental papilla, supposed to be derived from periodontal ligament.[1] Although the etiopathogenesis of POF is uncertain, an origin from cells of the periodontal ligament has been suggested.[4] The site of the lesion in 50% of cases is usually the incisor-cuspid region.[5] In the present case, the lesion is present in between the two maxillary central incisors, causing slight displacement of both the teeth. The occurrence of the lesion in such a site is quite rare and classical, which makes this case report very rare.

POF has a peak incidence in young and teenaged females. Cundiff reported that the lesion is prevalent between the age of 5 and 25 years with peak incidence at 13 years of age. He also reported a definite female predilection.[6] In the present case, the age of the female child was 14 years. However, the overall prevalence of POF is 3.1% of all oral tumours and 9.6% of all gingival lesions.[78] Clinically, the lesion appears as nodular mass, which may be pedunculated or sessile, pink to red, and surface is rarely ulcerated.[1] In the present case, the lesion is sessile, surface is smooth, and non-ulcerated [Figure 1].

POF is considered to be reactive rather than neoplastic.[9] It is interesting to note that the fibroma, a true neoplasm of connective tissue origin, is microscopically similar to the condition known as inflammatory hyperplasia, an increased bulk of connective tissue which forms as part of an inflammatory reaction. The distinction between the two general processes, hyperplasia and neoplasia, is poorly defined in such cases. Hyperplastic tissues sometimes, but not invariably, regress after removal of the stimulus or the irritant. A neoplastic tissue shows no such regressions. However, processes of hyperplasia and neoplasia may not be as completely disassociated as previously considered and that a true oral neoplasm may result from chronic irritation.[9] However, in the present case, there was no source of any chronic irritation in relation to both the central incisors. POF is characterized by a high degree of cellularity usually exhibiting bone formation, although occasionally cementum-like material or dystrophic calcification may also be formed. Chronic irritation of the gingiva causes localized reactive hyperplastic lesions, which are classified into POF; peripheral giant cell granuloma (PGCG); pyogenic granuloma (PG); and focal fibrous hyperplasia (FFH).[10] Hence, in the present case report, the diagnosis of POF was thought to be appropriate.

Differential diagnosis in such cases is essential, due to the clinical relevance to many other similar lesions and POF's tendency to recur.[11] Histopathological examination is essential for accurate diagnosis.[12] Differential diagnosis includes peripheral odontogenic fibroma, PG, peripheral giant cell granuloma, peripheral ameloblastoma, and calcifying epithelial odontogenic tumor.[13]

The peripheral odontogenic fibroma (WHO type) is a relatively rare, benign, unencapsulated, and exophytic gingival mass of fibrous connective tissue.[14] According to the present concept, cases reported in the literature under the terms “odontogenic gingival epithelial hamartoma” “hamartoma of the dental lamina” and “peripheral ameloblastic fibrodentinoma” are actually examples of peripheral odontogenic fibroma.[15] The peripheral odontogenic fibroma must be differentiated histologically from POF, which is a reactive lesion, and from the peripheral ameloblastoma and the calcifying epithelial odontogenic tumor which are benign odontogenic tumours.[15] In the present case, the lesion is sessile and encapsulated by a thick periosteum. Hence, it is excluded from peripheral odontogenic tumor, PG, and epulis/papilloma. Both PGs and pregnancy epulides may mature and become less vascular and more collagenous, gradually converting to fibrous epulides.[1] There is no doubt that the PG, if left untreated, will undergo eventual healing by sclerosis and will then microscopically resemble the POF.

The management protocol in such cases varies, depending on the availability of options for the clinicians and may include the use of scalpel or laser or electrocautery for successful excision of the lesion. Surgical excision including the involved periodontal ligament and periosteum is the preferred treatment. Complete surgical excision, down to the periosteum is performed in the present case, as the recurrence rate of POF is quite high. According to Eversole LR et al., the recurrence rate of POF is 8%–20%.[16] Close postoperative follow-up is essential for better prognosis.

Gingival and periodontal lesions in children are quite rare. However, in teenage adolescent female patients, the incidence of gingival lesions such as PG and FFH in the second decade of life is quite high due to the hormonal effect on the gingival tissues. POFs showed no gender predilection.[10] Pediatric patients with such lesions have special management considerations as they require early recognition and treatment. POF requires proper treatment protocol with close postoperative follow-up for the better prognosis.

CONCLUSION

POF a reactive lesion of the gingiva, which has grown rapidly to significant proportions, becomes very rare when it arises from the interdental papilla between two central incisors. POF of such magnitude should be dealt cautiously, as it has a high recurrence rate. Surgical dissection of the lesion should be followed by quarterly follow-up to avoid future mucogingival defects.

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Conflicts of interest

There are no conflicts of interest.