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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 28  |  Issue : 4  |  Page : 445-448

A rare case of benign aggressive cementoblastoma


1 Department of Oral Medicine and Radiology, Sree Anjaneya Institute of Dental Science, Calicut, Kerala, India
2 Department of Oral Medicine and Radiology, Mar Baselios Dental College, Kothamangalam, Kerala, India
3 Department of Oral and Maxillofacial Pathology, Mar Baselios Dental College, Kothamangalam, Kerala, India

Date of Submission17-Dec-2015
Date of Acceptance31-Jan-2017
Date of Web Publication21-Feb-2017

Correspondence Address:
Dr. Joseph Johny
Department of Oral Medicine and Radiology Sree Anjaneya Institute of Dental Science, Calicut, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.JIAOMR_255_15

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   Abstract 

Cementoblastoma is considered to be a rare odontogenic neoplasm of mesenchymal origin. The World Health Organization classified benign cementoblastoma and cementifying fibroma as the only true cemental neoplasms. This is a case of cementoblastoma which has shown an unusual local aggressiveness causing lingual cortical plate perforation, which is a rare presentation of this lesion. Radiographic and histologic findings along with the treatment plan are described in detail.

Keywords: Cementoblastoma, locally aggressive, radiographic findings, treatment plan


How to cite this article:
Johny J, Kunjumon R, George GB, Sebastian J. A rare case of benign aggressive cementoblastoma. J Indian Acad Oral Med Radiol 2016;28:445-8

How to cite this URL:
Johny J, Kunjumon R, George GB, Sebastian J. A rare case of benign aggressive cementoblastoma. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2020 Aug 14];28:445-8. Available from: http://www.jiaomr.in/text.asp?2016/28/4/445/200638


   Introduction Top


Cementoblastoma is a rare neoplasm, which was previously known as “benign cementoblastoma.” It is considered to be an odontogenic ectomesenchymal tumor as well as a fibro-cement-osseous lesion. It is also considered to be a true neoplasm of cementoblasts.[1] It is characterized by a vital tooth which has a mass of cementum or cementum-like tissue fused to its roots.[2] It was first described in 1927 by Dewey.[3] The tumor is often seen in young adults and is commonly associated with a mandibular molar or premolar, which comprises less than 1–6.2% of all odontogenic tumors.[4] Commonly known to be asymptomatic, these tumors can present with pain and swelling. Mostly, the final diagnosis of cementoblastoma is based on histopathology, however, certain typical radiographic features are sufficient to diagnose cementoblastoma.[5],[6],[7] Removal of both tooth and attached cementoblastoma is the treatment of choice. Although the lesion is of a benign nature, a rare case of aggressive behavior is reported here.[8]


   Case Report Top


A 14-year-old male patient reported to the department of Oral Medicine and Radiology with a chief complaint of pain and swelling in the lower right jaw. Swelling was noticed since 2 months which was initially small in size and gradually increased to the present size. His medical and family histories were noncontributory. Extraorally, there was a gross facial asymmetry on the right side of the face, with a solitary diffuse swelling on the right side of the mandible, measuring 15 × 10 mm, which on palpation was tender and bony hard in consistency [Figure 1]. Intraorally, the lesion was extending from the mesial aspect of 47 to distal aspect of 48 with mild obliteration of the buccal vestibule [Figure 2]. On palpation, the lesion was tender and bony hard in consistency. Electric pulp testing was performed, and all the teeth associated with the lesion were vital in nature. With these clinical findings and histories, we made a provisional diagnosis of benign tumor of the jaw, with cementoblastoma being the first differential diagnosis because of the age, location and vitality of the teeth. Juvenile ossifying fibroma, keratocystic odontogenic tumor, ameloblastoma, and osteoblastoma were our other differential diagnoses.
Figure 1: Extraoral view showing a diffuse solitary swelling on the right side of the mandible

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Figure 2: Intraoral view showing expansion of the buccal cortical plates with obliteration of buccal vestibule

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Intraoral periapical radiograph, mandibular occlusal radiograph, orthopantomogram and computed tomography (CT) were obtained. Intraoral periapical radiograph showed an ill-defined radiopaque mass roughly spherical in shape with a diameter of 15 mm surrounded by a diffuse radiolucent halo attached to the apex of 47 [Figure 3]. Mandibular occlusal radiograph showed buccal and lingual cortical plate expansion with discontinuity of the lingual cortical plate between 46 and 47 [Figure 4]. Orthopantomogram revealed an ill-defined radiopaque mass roughly spherical in shape with a diameter of 15 mm surrounded by a diffuse radiolucent halo attached to the apex of 47 extending to the mesial root of 46 [Figure 5]. CT revealed a radiodense mass measuring 25 × 23 mm, at the apex of 47 with buccal cortical plate expansion and lingual cortical plate perforation [Figure 6] and [Figure 7], which suggested an aggressive nature of the lesion. The radiopaque mass surrounded by ill-defined radiolucency attached to the tooth and the vital nature of the tooth were pointing towards a radiographic diagnosis of cementoblastoma. Expansion of the jaw and perforation of the lingual cortical plate were suggesting an aggressive nature.
Figure 3: Intraoral periapical radiograph of 46, 47 showing an ill-defined radiopaque mass roughly spherical in shape with a diameter of 15 mm surrounded by a diffuse radiolucent halo attached to the apex of 47

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Figure 4: Mandibular occlusal radiograph showing buccal and lingual cortical plate expansion with discontinuity of lingual cortical plate between 46 and 47

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Figure 5: Orthopantomogram showing an ill-defined radiopaque mass roughly spherical in shape with a diameter of 15 mm surrounded by a diffuse radiolucent halo attached to the apex of 47 extending to the mesial root of 46

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Figure 6: Three-dimensional CT reconstruction showing a radiodense mass measuring 25 × 23 mm at the apex of 47 with lingual cortical plate perforation

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Figure 7: CT (axial section) showing a radiodense mass with buccal cortical plate expansion and lingual cortical plate perforation

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A compete routine blood examination was done before incisional biopsy and the specimen was sent for histopathological examination. Histologic specimen [Figure 8] revealed trabeculae of the calcified tissue with numerous lacunar spaces filled with cementoblasts. Trabeculae spaces were filled with fibrovascular connective tissue. Mineralized areas in many foci showed reversal lines and resting lines. Tumor mass was seen merging with the cementum of the root of the associated tooth. The section also exhibited root dentin and associated pulp canal. Clinical, radiological and histopathologic features were suggestive of a locally aggressive cementoblastoma. The lesion was surgically managed by a mandibular marginal resection, preserving the neurovascular bundle and stabilized using a reconstruction plate in the lower border of the mandible [Figure 9].
Figure 8: Histologic specimen showing trabeculae of calcified tissue with numerous lacunar spaces filled with cementoblasts

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Figure 9: Postsurgical orthopantomogram showing marginally resected right side the mandible stabilized with reconstruction plate

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   Discussion Top


A rare case of benign cementoblastoma with an aggressive nature is presented here. They can be defined as a true neoplasm of the cementum or cementum-like tissues fused to the roots of the involved teeth.[1],[2] It was first described by Dewy in the year 1927,[3] and was described by Norberg in 1930 as a slow growing, benign odontogenic tumor arising from cementoblasts.[9],[10] Cementoblastoma comprises less than 1–6.2% of all odontogenic tumors.[4] Majority of the cases have been reported among Caucasians.[11]

Most patients complain of mild pain and bony swelling in the initial stage. It is seen across a wide range of age groups but often seen among young adults, mostly under 20 years of age (50%), and is mostly associated with a mandibular molar or premolar.[5] Males are affected more frequently than females. Though this lesion is usually asymptomatic, facial asymmetry and cortical expansion have also been reported.[6],[7],[12],[13] In our case, the patient presented with the complaints of swelling and pain.

The cementoblastoma has a typical pathognomonic radiographic appearance. It appears with a well-defined radiopacity surrounded by a radiolucent halo. The lesion is fused to the partly resorbed root(s) of the associated tooth. The internal structure of the mass may show a mixed radiolucent–radiopaque nature, which often has a radiating pattern.[14] In our case, we could identify the radiopacity attached to the tooth surrounded by radiolucent halo. Though the lesion is considered to be benign with a relatively minor tendency to recur, there are reports which show local aggressive nature. These aggressive signs include bony expansion, erosion of cortical plates, displacement of adjacent teeth, invasion of pulp chamber and root canals, and extension to adjacent teeth.[15] In our case, buccal cortical plate expansion and lingual cortical plate perforation was seen, indicating its aggressive nature. In a study conducted by Brannon et al.,[1] in the year 2002, it was found that the incidence of aggressive nature was more in recurrent tumors than in nonrecurring tumors; it was also found that, out of 44 cases, there was recurrence in 13 cases (37.1%). This is the second case to be reported in literature of a nonrecurring cementoblastoma with an aggressive nature; the first case was reported by Collins et al. in the year 2011 in a 19-year-old female in the mandibular molar region.[8]

Cementoblastoma and osteoblastoma are lesions that have histologically similar features.[16] The cementoblastoma is distinguished from osteoblastoma by its location and intimate association with a tooth root. The osteoblastoma normally arises in the medullary cavity of many bones, which includes the long bones, vertebrae, and jaws.[1] The appropriate treatment normally considered is the removal of the lesion along with the affected tooth or teeth, followed by thorough curettage or peripheral ostectomy along with regular follow up.[17] A more conservative technique to retain the involved tooth and remove the lesion using a surgical endodontic approach has also been reported.[17]


   Conclusion Top


Although cementoblastoma has a benign nature, local aggressiveness and recurrence may occur. Hence, despite its rarity, it is very important to improve the awareness of this type of lesion among practitioners. Through this article we aim to provide an insight to dental practitioners regarding the clinical features, radiographic findings, histologic findings and treatment options of an aggressive benign cementoblastoma.

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 initials 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.
Brannon RB, Fowler CB, Carpenter WM, Corio RL. Cementoblastoma: An innocuous neoplasm? A clinicopathologic study of 44 cases and review of the literature with special emphasis on recurrence. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;93:311-20.  Back to cited text no. 1
    
2.
Sankari LS, Ramakrishnan K. Benign cementoblastoma. J Oral Maxillofac Pathol 2011;15:358-60.  Back to cited text no. 2
  Medknow Journal  
3.
Dewey KW. Osteoma of a molar. Dent Cosmos 1927;69:1143-9.  Back to cited text no. 3
    
4.
Lu Y, Xaun M, Takata T, Wang C, He Z, Zhou Z, et al. Odontogenic tumours. A demographic study of 759 cases in a Chinese population. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;86:707-14.  Back to cited text no. 4
    
5.
Ulmansky M, Hjorting-Hansen E, Praetorius F, Haque MF. Benign cementoblastoma; a review and five new cases. Oral Surg Oral Med Oral Pathol 1994;77:48-55.  Back to cited text no. 5
    
6.
Piattelli A, Di Alberti L, Scarano A, Piattelli M. Benign cementoblastoma associated with an unerupted third molar. Oral Oncol 1998;34:229-31.  Back to cited text no. 6
    
7.
Piatelli A, D'addona A, Piatelli M. Benign cemento blastoma: Review of the literature and report of a case at unusual location. Acta Stomatol Belg 1990;87:209-15.  Back to cited text no. 7
    
8.
Collins RE, Asturias RF. An aggressive benign cementoblastoma. US Army Med Dep J 2011 Jan-Mar: 91-4.  Back to cited text no. 8
    
9.
Norberg O. Zur Kenntnis der dysontogenetischen Geschwulste der Kieferknochen. Vrtljsschr f Zahnh 1930;46:321-55.  Back to cited text no. 9
    
10.
Schafer TE, Sing B, Myers DR. Cementoblastoma associated with a primary tooth: A rare pediatric lesion. Pediatr Dent 2001;23:351-3.  Back to cited text no. 10
    
11.
Papageorge MB, Cataldo E, Nghiem FT. Cementoblastoma involving multiple deciduous teeth. Oral Surg Oral Med Oral Pathol 1987;63:602-5.  Back to cited text no. 11
    
12.
Cundiff J. Developing cementoblastoma: Case report and update of differential diagnosis. Quintessence Int 2000;31:191-5.  Back to cited text no. 12
    
13.
Matteson SR. Benign tumors of the jaws. In: White SC, Pharoah MJ, editors. Oral Radiology: Principles and Interpretation. 4th ed. Toronto: Mosby; 2000. p. 401-2.  Back to cited text no. 13
    
14.
Zachariades N, Skordalaki A, Papanicolaou S, Androulakakis E, Bournias M. Cementoblastoma: Review of the literature and report of a case in a 7-year-old girl. Br J Oral Maxillofac Surg 1985;23:456-61.  Back to cited text no. 14
    
15.
Slootweg PJ. Cementoblastoma and osteoblastoma: A comparison of histologic features. J Oral Pathol Med 1992;21:385-9.  Back to cited text no. 15
    
16.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 1st ed. Philadelphia: Saunders; 1995. p. 476.  Back to cited text no. 16
    
17.
Goerig AC, Fay JT, King E. Endodontic treatment of a cementoblastoma. J Oral Surg 1984;58:133-6.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9]



 

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