Exudative Benign Cementoblastoma with Radiographic Rarities- A Case Report with Review of Literature
Correspondence Address :
Dr. Karthikeya Patil,
Professor and Head, Department of Oral Medicine and Radiology, JSS Dental College and Hospital, JSS Academy of Higher Education and Research, Bannimantapa, Mysuru, Karnataka, India.
E-mail: dr.karthikeyapatil@jssuni.edu.in
Cementoblastoma is a rare benign odontogenic neoplasm of mesenchymal origin. The benign cementoblastoma was previously classified as one of the cementoma neoplasias by the World Health Organisation’s (WHO) classification of odontogenic tumours. The benign cementoblastoma in 2005 has been added to the list of odontogenic tumours that involve “mesenchyme and/or odontogenic ectomesenchyme, with or without odontogenic epithelium”. It accounts for around 0.69-8% of all odontogenic tumours. The lesion usually is asymptomatic and slow growing causing cortical plate expansion of both buccal and lingual plates. Cementoblastoma has male predilection, commonly seen involving mandibular first molar diagnosed in early age. There are very few incidents of an infected cementoblastoma in the literature. Radiological examinations can reveal a blend of radiolucent and radiopaque mass presenting a wheel spoke pattern. The histopathological results for both cementoblastoma and osteoblastoma are comparable. The relationship of the lesion with the tooth is the distinguishing element. Hereby, authors present a case of infected cementoblastoma in a 51-year-old female patient involving impacted mandibular second molar, clinically aggressive with unique radiologic features. Cementoblastoma is usually an incidental finding while the present case was associated with an impacted tooth and was symptomatic with infection. The uniqueness of the present case was the complete involvement of the root structure and the cyst like expansion of the lesion instead of the usual thin radiolucent rim. Also, the lesion was infected with no dental caries associated with the tooth.
Dental cementum, Impacted, Odontogenic tumour
A 51-year-old female patient reported to the Dental Hospital with the chief complaint of swelling and pain on the left side of the face which she had noticed two months ago. The pain was moderate in intensity, intermittent in nature with no aggravating factors and relived on taking analgesics. The swelling was initially small which gradually grew to attain the present size. The patient had visited a dental practitioner with the same complaint two months ago where she got her tooth left mandibular first molar (36) extracted which was misdiagnosed as the cause for swelling and pain without prior diagnostic radiographs. The patient reported to our dental hospital since there was no relief form pain and swelling. The patient’s medical and family history were unremarkable.
Clinically, patient presented with an extraoral swelling in the left lower third of the face measuring around 5×6 cm in diameter with no remarkable surface changes (Table/Fig 1)a. On palpation, the swelling was well defined and hard in consistency and no signs of paraesthesia noted. Intraoral examination revealed obliteration of the left buccal vestibule in the region of 36 and 37, associated buccal cortical plate expansion with intact lingual cortical plate. Pus discharge was noted in the region of 37 which was clinically missing (Table/Fig 1)b.
Considering the above history and examination a provisional diagnosis of dentigerous cyst with respect to impacted 37 was given and differential diagnoses of infected residual cyst (in relation to 36) and odontogenic tumour with impacted teeth were considered. Orthopantomograph (OPG) revealed an impacted 37 with a radiopaque mass involving the entire root. Loss of complete root morphology and well circumscribed wide spacious concentric ring around the root which had fibrous root appearance was evident (Table/Fig 1)c which is rarely seen as compared to common spoke wheel appearance. Cone Beam Computed Tomography (CBCT) images of different sections, showed a well-defined radiolucent band inside the cortical border with a radiopaque mass at the centre accompanied by minor areas of radiolucency (Table/Fig 2)a,(Table/Fig 2)b,(Table/Fig 2)c.
There was no invasion of the lesion to the mandibular canal in all three sections- axial, coronal and sagittal. There was evidence of buccal cortical plate expansion and perforation of buccal cortical plate, giving more evidence of an aggressive matured benign cementoblastoma (Table/Fig 2)d.
Since, the lesion was infected, the patient was put on broad spectrum antibiotic coverage and routine haematological examination before surgical excision showed normal blood parameters. Excisional biopsy of the lesion along with the extraction of the associated 37 was done under general anaesthesia. The gross specimen consisted of a tooth along with bony tissue mass measuring 2.5×2×2 cm (Table/Fig 3)a. There was loss of the root morphology while the crown appeared normal. Microscopically, the sections showed basophilic masses with inflammatory infiltrate, fibrovascular connective tissue, areas of fibrosis and bony trabeculae lined by osteoblasts and association of the lesion with the root of the tooth, confirmed the diagnosis of cementoblastoma (Table/Fig 3)b.
The patient was advised periodic follow-up upto six months. The patient showed remarkable improvement in terms of healing and pain control in two months. Postoperative follow-up OPG taken at the end of six months showed complete resolution of the lesion with no recurrence (Table/Fig 3)c. Patient was advised for replacement of missing teeth. Schematic representation of the proposed staging of cementoblastoma based on root involvement with varied radiographic features (Table/Fig 3)d.
Cementoblastoma is a relatively rare and uncommon benign neoplasm of mesenchymal origin (1). Cementoblastoma accounts for around 0.69-8% of all odontogenic tumours (2). It is formed by the haphasard deposition of cementum like material by the proliferating cementoblast cells. The neoplasm usually presents as a slow growing bony hard swelling which is predominantly asymptomatic (3). Paraesthesia was also observed when the lesion involves the mandibular canal (4). The lesion has a slight male predilection usually occurring in the 2nd and 3rd decade of life. While there are cases reported in the literature in patients ranging between 10-72 years of both genders (4),(5). Few literature points to their equal gender predilection (6). A fully erupted mandibular permanent 1st molar is most commonly associated with the lesion. Nevertheless, the incidence of occurrence in the maxilla and primary dentition cannot be denied (7). The present case under discussion is unique in its presentation which is highlighted in (Table/Fig 4). Based on the literature survey very few cases have been reported involving impacted mandibular second molar (Table/Fig 5) (1),(2),(3), (5),(6),(7),(8),(9),(10),(11),(12),(13),(14),(15),(16),(17),(18),(19),(20),(21),(22),(23),(24),(25).
The lesion usually is asymptomatic and slow growing causing cortical plate expansion of both buccal and lingual plates (5). There are very few incidents of an infected cementoblastoma in the literature (5),(8),(9). The clinical examination in the present case revealed pus discharge in relation to unerupted 37 with intraoral sinus opening and 36 was missing. The present case seemed aggressive and more matured (considering the age at diagnosis) with the expansion of the buccal cortical plate and an extraoral swelling which the patient had noticed for the past two months.
Chrcanovic BR and Gomez RS, analysed 258 cases of cementoblastoma deriving the following result i.e, equal gender prevalence and higher prevalence was evident in the 2nd/3rd decades 9of life. The common site of occurrence is mandibular 1st molars. Lesions were frequently related with bone expansion (74.9%), clinical symptoms (70.2%), vital teeth (78%), and root resorption (59.8%). Less frequent observations included perforation of cortical bone (16.3%), inferior displacement of the mandibular canal (23.6%) (10).
The results of a radiological examination can present three stages i.e, initial, intermediate and mature stages. In the initial stage, the lesion is usually radiolucent. The intermediate stage manifests a blend of radiolucent and radiopaque mass. While a completely mature lesion displays a dense radiopaque mass surrounded by a radiolucent halo (26). Going through the radiographic data of case reports, it was observed that almost all the lesions were in the maturing stage and few were found to be in the intermediate stage. Therefore, lesions in their initial stage, seldom show any symptoms. Also, the case reports of cementoblastoma associated with infection or impacted tooth, are only a few (Table/Fig 5) (1),(2),(3), (5),(6),(7),(8),(9),(10),(11),(12),(13),(14),(15),(16),(17),(18),(19),(20),(21),(22),(23),(24),(25).
In the present case, the radiographic examination via orthopantomogram and CBCT images revealed an impacted 37 with a radiodense mass involving the entire root component from the cervical region to its apex showing a fibrous root appearance which was different from the regular wheel spoke pattern. The density of the cemental mass was obscuring the entire outline of the enveloped root resulting in loss of entire root morphology. The mass had invaded the root canal and had obliterated the periodontal ligament space. The tumour was considered to be in the matured stage since the central radiopaque mass involving the entire root was surrounded by a radiolucent band from the center to the periphery. The lesion was extensive enough to cause the expansion of mandible perforating the buccal cortical plate without any periosteal reaction. In the literature review most of the reported cases involved either apical 3rd or extended up to middle third of root which was different in the present case with the involvement of the entire root. Considering the unique radiographic features in the present case, a new classification for staging can be proposed based on the level of root involvement (Table/Fig 3)d.
1. Initial stage-Tumour involving apical third.
2. Intermediate stage-Tumour involving middle third of root.
3. Mature stage-Tumour involving entire root.
Cementoblastoma in its initial stage can be treated endodontically. While the intermediate and mature stages require extraction of the involved tooth (27).
Histopathological investigation gives similar findings for both cementoblastoma and osteoblastoma. The differentiating factor is through the association of the lesion to the tooth. In Cementoblastoma, the radiopaque mass is attached to the root of the associated tooth while in osteoblastoma the radiopaque mass is usually not in contact with the root of any tooth (13).
The present case of infected cementoblastoma associated with impacted tooth, had a history of extraction of the adjacent tooth. The chances of infection of cementoblastoma associated with an unerupted tooth, is highly unlikely (5). The lesion may have incurred the infection from an infected periapical lesion of the adjacent tooth or could be idiopathic. The probable cause of infection of cementoblastoma associated with an erupted tooth with no dental caries may be from the per coronal abscess or periodontal infection of the associated tooth or idiopathic.
The present case was a case of cementoblastoma with distinct clinical and radiological manifestations. The present case mandates the inclusion of cementoblastoma in the differential diagnosis of impacted/clinically missing tooth. Also, highlights the importance of radiographic investigation prior to arriving at a specific diagnosis and intervening with any surgical intervention. Radiographic investigation should be considered mandatory by all practitioners to avoid misdiagnosis and inaccurate treatment planning.
DOI: 10.7860/JCDR/2022/56579.16741
Date of Submission: Mar 23, 2022
Date of Peer Review: Apr 18, 2022
Date of Acceptance: May 30, 2022
Date of Publishing: Aug 01, 2022
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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