An Unusual Presentation of Cemento-ossifying Fibroma in the Anterior Maxilla: A Case Report

1. Postgraduate Student, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College (DMIHER), Sawangi (M), Wardha, Maharashtra, India. 2. Associate Professor, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College (DMIHER), Sawangi (M), Wardha, Maharashtra, India. 3. Professor, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College (DMIHER), Sawangi (M), Wardha, Maharashtra, India. 4. Assistant Professor, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College (DMIHER), Sawangi (M), Wardha, Maharashtra, India. 5. Assistant Professor, Department of Oral Pathology and Microbiology Sharad Pawar Dental College (DMIHER), Sawangi (M), Wardha, Maharashtra, India.

Abstract

Cementifying Fibroma (CF) is a benign fibro-osseous lesion with sharply defined and well-confined edges. It is slow-growing and has a radiolucent peripheral component. It is mostly seen in the mandible in the 20-39 years age group with a female predilection. Hereby, the authors present a case report of a 60-year-old woman who had a painless growth in the upper front region of her jaw for two months and visited Outpatient Department (OPD). Extraoral examination revealed facial asymmetry due to swelling in the upper front region of the jaw. Intraoral examination showed round to oval swelling in the 11 and 21 regions. On radiographic examination, a round radiolucency in the 11 region was appreciated. After excision, the tissue was sent to the laboratory, and histopathologically the diagnosis was confirmed as peripheral CF. The patient was kept in a 12-month follow-up postoperatively, which reported no recurrence.

Central cemento-ossifying fibroma, Fibro-osseous lesions, Ossifying fibroma

Case Report

A 60-year-old female visited the Outpatient Department (OPD) of a dental hospital with a chief complaint of a painless swelling in her upper front region of the jaw for eight months. There was no history provided regarding any trauma or previous occurrence of the same condition. Following a complete evaluation of her history, nothing noteworthy was found in her dental, medical, or family history. On extraoral examination, slight facial asymmetry was observed in the upper anterior region due to a proliferative growth in the anterior maxilla. Upon palpation, the swelling was non tender and felt soft to firm in consistency. Temporomandibular joint movements were normal, with adequate mouth opening and incompetent lips. Intraoral examination revealed a small, well-defined, oval swelling over the upper anterior gingiva that extended from the maxillary central incisors region, measuring approximately 1.8×2.5 cm in size. On inspection, the lesion was smooth, pink in colour, the same as the surrounding normal mucosa, and covered by intact mucosa (Table/Fig 1). Upon palpating the lesion, it was not tender and was soft to firm in consistency. Radiographic examination of the area of concern showed an ill-defined radiolucency which is round to oval in shape and approximately 1.3×1 cm in size. The lesion was well-demarcated radiographically and showed minute foci of calcifications, suggesting a Calcifying Odontogenic Fibroma (COF) in association with the right maxillary first incisor (Table/Fig 2). After a complete blood examination, the lesion was excised under local anaesthesia, and the excised tissue was sent for histopathological examination {Haematoxylin and Eosin (H&E)}. In the laboratory, a single irregular whitish tissue piece measuring 1.4×0.9×0.6 cm was received and sent for further processing (Table/Fig 3). Under microscopy, the scanner view showed hyperplastic parakeratinised stratified squamous epithelium and fibrous connective tissue stroma (Table/Fig 4). At 10X magnification, long and broad rete ridges and bundles of collagen were appreciated, with closely packed growing fibroblasts in cellular fibroblastic tissue (Table/Fig 5). In numerous areas of the connective tissue stroma, small foci of calcifications comprising bony trabeculae and cementum-like tissue with a few osteoid tissues were seen (Table/Fig 6). These calcifications were in the form of single or multiple interconnecting trabeculae of bone or osteoid, suggestive of ‘cemento-ossifying fibroma’. The lesion was initially considered to be a fibroma or traumatic fibroma due to the high concentration of collagen fibres in the connective tissue. However, upon histopathological examination, the presence of calcific foci confirmed the diagnosis as a COF.

After the final histopathological diagnosis, the patient was recalled after 15 days and one month for evaluation (Table/Fig 7)a-c. The healing after one month was good. The patient was then recalled at intervals of six months and 12 months, and she did not report any history of the recurrence of the same condition.

Discussion

Fibro-osseous lesions of the skull are an uncommon category of diverse disorders that can develop either as a localised solitary process or as a symptom of a systemic skeletal disorder. This group includes benign mesenchymal tumours like CFs. CF, which develops from the mesodermal germ layer, is an uncommon, non odontogenic disease similar to the ossifying fibroma of the periodontal ligament (1). The present locally damaging, non cancerous tumour affects the petromastoid region less frequently than the jaw bones, zygoma, paranasal air sinuses, and orbits. Since CF is a benign, slow-growing tumour, it is relatively less seen in younger children, and so this subtype is known as benign of a juvenile type (1). Four different forms of cementum-containing lesions were identified by the World Health Organisation (WHO) in 1971: fibrous dysplasia, ossifying fibroma, CF, and cemento-ossifying fibroma (2). The most widely accepted theory for its aetiopathogenesis holds that it is a tumour of periodontal origin. However, other theories have also been proposed. It is believed that it originates from multipotent mesenchymal blast cells, which can produce cementum, alveolar bone, and fibrous tissue and are found in the periodontal membrane (3).

In the aetiopathological aspects, chromosome translocations have been observed in a small number of cemento-ossifying fibroma cases. Originating from mesenchymal blast cells found in the periodontal ligament, this entity has the capability to develop into fibrous tissue, cement, bone, or a blend of these components (4). Due to microscopic commonalities with fibrous dysplasia and cemento-osseous dysplasia, researchers suggest that the lesion is an example of a localised dysplastic process in which bone metabolism has been altered (5). Its origin may also be triggered by an irritant stimulus such as tooth extraction or, in some cases, trauma (6).

Early diagnosis is necessary for appropriate management to achieve local control and prevent spread; it can also be quickly obtained using minimally invasive Fine Needle Aspiration Cytology (FNAC) (7). Histopathologically, the lesion is mostly made up of hyperplastic epithelium with numerous finely interlacing collagen fibres, along with significant areas of active fibroblasts interspersed in the connective tissue stroma. This connective tissue typically contains several tiny foci of atypical bone trabeculae (8). This finding raises two areas of research that deserve more attention: first, whether instances of extremely aggressive CF affirm a more aggressive surgical approach, specifically when they recur; and second, whether the histologic manifestation may aid in predicting the nature of this tumour (9). COF presents with different radiographic prototypes depending on the degree of mineralisation. Initially, it appears radiolucent; as it matures, calcific specks become more prominent until it eventually becomes a fully radiopaque mass (10). Depending on the size and location of the specific lesion, conservative enucleation, curettage, or aggressive surgery has historically been used to treat CFs. They are distinguished by their effortless ability to absorb the environment. Mandibular central cemento-ossifying fibromas are easier to remove completely than maxillary central CFs. This difference might be due to the maxillary sinus’s capacity for expansion and the differential in bone density between the mandible and maxilla. The prognosis is good, although recurrence is possible if, not treated with complete excision (11). In the literature, various reported cases of CF are listed in (Table/Fig 8) (12),(13),(14),(15),(16),(17).

Conclusion

In summary, the case of cemento-ossifying fibroma in the maxillofacial region discussed in present report highlights its rarity within a timeframe, as well as the considerable challenges exposed in terms of differential diagnosis. Benign fibro-osseous lesions present a diagnostic conundrum due to their overlapping clinical, radiological, and histopathological features, which can perplex surgeons, pathologists, and radiologists. The divergence in the treatment plan and prognosis further complicates matters. Therefore, a comprehensive approach involving meticulous correlation of clinical, radiological, and histopathological findings is imperative for reaching an accurate diagnosis. Subsequently, a tailored treatment protocol can be devised to address the specific characteristics of each case effectively.

DOI and Others

DOI: 10.7860/JCDR/2024/68935.19746

Date of Submission: Dec 06, 2023
Date of Peer Review: Feb 17, 2024
Date of Acceptance: May 10, 2024
Date of Publishing: Aug 01, 2024

AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. Yes

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ETYMOLOGY: Author Origin

EMENDATIONS: 8