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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 28  |  Issue : 4  |  Page : 449-452

Osteoid osteoma in anterior border of ramus of mandible: A rare entity


1 Department of Oral Medicine and Radiology, Church of South India College of Dental Sciences and Research, Madurai, Tamil Nadu, India
2 Department of Oral and Maxillofacial Surgery, Church of South India College of Dental Sciences and Research, Madurai, Tamil Nadu, India
3 Department of Oral and Maxillofacial Pathology, Church of South India College of Dental Sciences and Research, Madurai, Tamil Nadu, India

Date of Submission31-Dec-2015
Date of Acceptance05-Dec-2016
Date of Web Publication21-Feb-2017

Correspondence Address:
Dr. Thomas Jones Raja Devathambi
Department of Oral Medicine and Radiology, Church of South India College of Dental Sciences and Research, Madurai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.200640

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   Abstract 

Osteoid osteoma is a benign lesion accounting for 3% of all primary bone tumors and approximately 10% of benign bone tumors. Approximately 80% of osteoid osteomas occur in the long bones, whereas less than 1% occur in jaws. Osteoid osteoma is most frequently observed in the second and third decades of life, more commonly in males than females, in a ratio of approximately 2:1. In this case report, we discuss the case of a 13-year-old female patient having a painful bony lesion in the anterior border of the ramus of the mandible, which is a rare site of occurrence.

Keywords: Osteoid osteoma, ramus of the mandible, tumors


How to cite this article:
Devathambi TJ, Ambrose WC, Niazi KZ, Raja S. Osteoid osteoma in anterior border of ramus of mandible: A rare entity. J Indian Acad Oral Med Radiol 2016;28:449-52

How to cite this URL:
Devathambi TJ, Ambrose WC, Niazi KZ, Raja S. Osteoid osteoma in anterior border of ramus of mandible: A rare entity. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2021 Jan 28];28:449-52. Available from: https://www.jiaomr.in/text.asp?2016/28/4/449/200640


   Introduction Top


Osteoid osteoma was first described as a distinct clinical entity by Jaffe in 1935.[1] Jaffe and Lichtenstein have suggested that the lesion is a true neoplasm of osteoblastic derivation.[2] Jaffe refers to the osteoid osteoma as “sui genris,” denoting the lesion's small, self-limiting nature.[3] The exquisite pain produced by a very small lesion, never greater than 1 cm in diameter, is the most interesting clinical feature of osteoid osteoma.[4] Osteoma can be central, peripheral, or extraskeletal.[5]


   Case Report Top


A 13-year-old female patient reported to the Department of Oral medicine and Radiology, C.S.I. College of Dental Science and Research, Madurai, with complaint of pain and swelling in left side of the face since 3 months. Pain was of gradual onset, dull type, intermittent in nature, which increased on mastication and relieved with medication. The patient was conscious of the swelling due to its slow increase in size causing a mild disfigurement of the face on the left side of the face [Figure 1]. No significant medical and dental history was present.
Figure 1: Extra oral photograph showing facial asymmetry

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On extraoral examination, a solitary diffuse ill-defined swelling of approximately 3 × 3 cm was present in left side of the face [Figure 2]. It was spherical in shape extending superiorly up to left zygomatic arch, inferiorly to 1 cm above the left lower border of the mandible, anteriorly to 1 cm away from the left ala of the nose and posteriorly to 1 cm away from the left anterior border of the mandible. Skin and color over the swelling were normal. On palpation, swelling was found to be bony hard in consistency and tender on palpation. It was immobile and attached to underlying tissue; but, the swelling was moving with jaw movements. Mouth opening was restricted to 24 mm [Figure 3]. On intraoral examination, there was blanching of tissue present in the left buccal mucosa along the anterior border of the ramus of mandible [Figure 4]. On palpation, a bony mass was palpable along the left anterior border of the ramus of the mandible, which measured approximately 1 × 1 cm in size, spherical in shape, tender on palpation, stony hard in consistency. On the basis of clinical findings, a provisional diagnosis of osteoma was made and differential diagnoses viz., fibrous dysplasia, bony exostoses, and coronoid hyperplasia were considered.
Figure 2: Swelling on the left side of the face

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Figure 3: Restricted Mouth opening of 24 mm

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Figure 4: Intraoral view showing blanching on buccal mucosa

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Orthopantomograph (OPG) revealed a well-defined radiopaque mass measuring 1 × 1 cm in size with intermingled radiolucencies was present on the left side anterior border of the ramus of the mandible [Figure 5]. Furthermore, on computed tomography (CT) examination, in axial, coronal, and sagittal sections, there was a well-defined radiopaque mass present on the left side anterior border of the ramus of mandible, which measured 1.4 × 1.5 cm in size [Figure 6]a,[Figure 6]b,[Figure 6]c. CT with three-dimensional reconstruction shows projected bony mass from the left anterior border of the ramus [Figure 6]d. Based on radiological investigations, we arrived at a radiographic diagnosis of osteoma.
Figure 5: Orthopantomograph showing radiopaque mass present on the left anterior border of the ramus of the mandible

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Figure 6: Computed tomography showing radiopaque mass in anterior border of the ramus in the (a) axial view, (b) coronal view, (c) sagittal view, and (d) 3D reconstruction

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Routine blood investigations such as complete hemogram were in normal limits, and serological investigation such as human immunodeficiency virus (HIV) and hepatitis were negative. Under general anesthesia through intraoral ramus approach, osteotomy was done along with anterior border of the ramus extending till the sigmoid notch. The bony mass was removed along with coronoid process [Figure 7]a and [Figure 7]b. Histopathological examination of the lesional tissue revealed irregular bony trabeculae lined with plump osteoblasts. The bony trabeculae showed varying degrees of calcification. The trabeculae comprised lacunae with osteocytes within them [Figure 8]. Patient was reviewed after 1 week, periodic follow-ups were done, and a postoperative extraoral photograph [Figure 9]a and OPG [Figure 9]b were taken.
Figure 7: The excised bony mass: (a) lateral aspect, and (b) medial aspect

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Figure 8: Photomicrograph showing irregular bony trabeculae lined by plump osteoblasts. Many dilated blood vessels are seen in the stroma (hematoxylin and eosin, 40×)

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Figure 9: Post-operative (a) extraoral photograph, and (b) orthopantomograph of the patient

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   Discussion Top


Osteoid Osteoma was described as a specific entity by Jaffe in 1935.[1] Approximately 80% of osteoid osteomas occur in the long bones, while less than 1% occur in jaws.[6],[7],[8],[9] Till date only less than 20 cases of osteoid osteoma occurring in mandible has been reported. No other case has been reported in the anterior border of the ramus of the mandible, and thus this case is being reported for its rarity.[10] Osteoid osteoma is most frequently observed in the second and third decades of life, more commonly in males than females, in a ratio of approximately 2:1.[6],[7],[8],[9] Our patient is also in the second decade, but is a female and does not fit into the gender predilection for this disease. Osteomas appears as unilateral, pedunculated, or sessile mushroom-like mass, well-marginated, and varying in diameter from 10–40 mm.[11] Osteomas can arise from the surface of the bone as a polypoidal or sessile mass or may be located within medullary bone.[12] Our patient also had a similar presentation.

Jaffe described a type of nidus which appeared as a hard osseous core composed of densely set trabeculae of newly formed bone which was atypical. Jaffe perceived the initial notable changes in this lesion as an increased vascularization and destruction with replacement by new atypical bone following resorption of the destroyed tissue.[13] Radiographically, this typical nidus is a round or ovoid radiolucency less than 1 cm in diameter, having no, or a small radiopacity in the center, located at the intracortex or subperiosteal site, which is surrounded by reactive sclerosis of the bone and sometimes with thick homogenous periosteal reaction.[8],[9] However, a radiolucent nidus of osteoid osteoma is not always easily recognized on a radiograph because of superimposition of other bony structures or because it is often obscured by extremely dense sclerosis around it,[14] as seen in our case. Histologically osteomas consist of mature, lamellar bone, or cancellous bone with abundant fibrofatty marrow between bony trabeculae.

According to the original description of the lesion by Jaffe, complete removal of the tumor with the nidus is the treatment of choice. Though some cases have regressed without any treatment, our patient had pain due to which surgical removal of the lesion was done. Osteomas usually do not recur, however osteomas may occur in Gardner's syndrome which is an autosomal dominant disease characterized by gastrointestinal polyps, multiple osteomas, skin and soft tissue tumors, and multiple impacted or supernumerary teeth. Because intestinal polyps are predominantly adenomas and may progress to malignancy in almost 100% of the patients, an endoscopy may be advised to rule out intestinal polyps.[15],[16] We referred the patient to a medical gastroenterologist to rule out intestinal polyps, however, the patient refused due to financial constraints.


   Conclusion Top


The dentist should always be aware of the unusual positions and presentation of osteomas in the maxillofacial region. Good clinical knowledge and proper radiographic investigations are essential for correct diagnosis of these lesions. Although recurrence is very rare, periodic follow-up should be done after surgical excision of osteomas.

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.
Jaffe HL. Osteoid-osteoma: A benign osteoblastic tumor composed of osteoid and atypical bone. Arch Surg 1935;31:709-28.  Back to cited text no. 1
    
2.
ShaferWG, Hine MK, Levy BM. A Text Book of Oral Pathology. 5th ed. New Delhi: Elseiver; 2006. p. 213-5.  Back to cited text no. 2
    
3.
Jaffe HL, Lichtenstein L. Osteoid osteoma: Further experience with the benign tumour. J Bone Joint Surg 1945;22:645.  Back to cited text no. 3
    
4.
Schulman L, Dorfman HD. Nerve fibres in osteoid osteoma. J Bone Joint Surgery Am 1970;52:1351-6.  Back to cited text no. 4
    
5.
Li G, Wu YT, Chen Y, Li TJ, Gao Y, Zhang J, et al. Soft-tissue osteoma in the pterygomandibular space: Report of a rare case. Dentomaxillofacial Radiol 2009;38:59-62.  Back to cited text no. 5
    
6.
Mirra JM, Gold RH, Picci P. Osteoid Osteoma. In: Mirra JM, editor. Bone Tumors. Philadelphia, PA: Lea Febiger; 1989. p. 226-48.  Back to cited text no. 6
    
7.
Schajowicz F. Bone-forming tumours 1.1.2 osteoid osteoma and osteoblastoma. In: Schajowicz F, ed. Histological Typing of Bone Tumours. 2nd ed. Berlin: Springer-Verlag; 1993. p. 7-9.  Back to cited text no. 7
    
8.
Resnick DL, Kyriakos M, Greenway G. Tumors and tumor-like lesions of bone: Imaging and pathology of specific lesions. In: Resnick DL, editor. Diagnosis of Bone and Joint Disorders. 3rd ed. Philadelphia, PA: Saunders; 1995. p. 3629-47.  Back to cited text no. 8
    
9.
Ayala AG, Ro JY, Raymond AK. Bone tumors. In: Damjanov I, Linder J, editors. Anderson's Pathology. 10th ed. St Louis: Mosby; 1996. p. 2531-73.  Back to cited text no. 9
    
10.
SinghA, Solomon MC. Osteoid osteoma of the mandible: A case report with review of the literature. J Dent Sci 2012. doi: 10.1016/j.jds. 2012.10.002.  Back to cited text no. 10
    
11.
Sayan NB, Uçok C, Karasu HA, Günhan O. Peripheral osteoma of oral and maxillofacial region: A study of 35 new cases. J Oral Maxillofacial Surg 2000;60:1299-301.  Back to cited text no. 11
    
12.
Neville BW, Damm DD, Allen CM, Bouquot JE, editors. Oral and maxillofacial pathology. Philadelphia: WB Saunders; 1995. p. 472-3.  Back to cited text no. 12
    
13.
Jaffe HL. Tumors and Tumorous Conditions of the Bones and Joints. Philadelphia, PA: Lea and Febiger; 1958. p. 92-105.  Back to cited text no. 13
    
14.
Sim FH, Dahlin DC, Beabout JW. Osteoid osteoma: Diagnostic problems. J Bone Joint Surg Am 1975;57A:154-9.  Back to cited text no. 14
    
15.
Bilkay U, Erdem O, Ozek C, Helvaci E, Kilic K, Ertan Y, et al. Benign osteoma with Gardner syndrome: Review of the literature and report of a case. J Craniofac Surg 2004;15:506-9.  Back to cited text no. 15
    
16.
Do Lee B, Lee W, Oh SH, Min SK, Kim EC. A case report of Gardner syndrome with hereditary widespread osteomatous jaw lesions. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107:e68-72.  Back to cited text no. 16
    


    Figures

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



 

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