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E-JCRT CORRESPONDENCE
Year : 2015  |  Volume : 11  |  Issue : 3  |  Page : 651

Primary intraosseous carcinoma of the mandible: A clinicoradiographic view


Department of Oral Medicine and Radiology, Kamineni Institute of Dental Sciences, Narketpally, Andhra Pradesh, India

Date of Web Publication9-Oct-2015

Correspondence Address:
M L Avinash Tejasvi
Department of Oral Medicine and Radiology, Kamineni Institute of Dental Sciences, Narketpally - 508 254, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.140814

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 > Abstract 

Primary intraosseous carcinoma (PIOC) or Primary intraosseous squamous cell carcinoma (PIOSCC) is a rare carcinoma, which arises within the jaws without connection to the oral mucosa and presumably develops from a remnant of odontogenic epithelium. It is a rare case which arises by direct transformation of odontogenic epithelial rests in the jawbone, including the epithelial rests found within the periodontal ligament and alveolar bone, remnants of the dental lamina, and the reduced enamel epithelium surrounding an unerupted/impacted tooth. Herein, we report a case of a 76-year-old male patient who came with a complaint of deviation of his jaws to one side which revealed resorption of total part of the condyle along with the condylar head and coronoid process on radiographic evaluation, which was histopathologically diagnosed as primary intraosseous carcinoma which is a very rare clinical entity.

Keywords: Denovo carcinoma, intra alveolar epidermoid carcinoma, primary intraosseous carcinoma, primary intraosseous squamous cell carcinoma, squamous cell carcinoma


How to cite this article:
Geetha P, Avinash Tejasvi M L, Babu B B, Bhayya H, Pavani D. Primary intraosseous carcinoma of the mandible: A clinicoradiographic view. J Can Res Ther 2015;11:651

How to cite this URL:
Geetha P, Avinash Tejasvi M L, Babu B B, Bhayya H, Pavani D. Primary intraosseous carcinoma of the mandible: A clinicoradiographic view. J Can Res Ther [serial online] 2015 [cited 2019 Dec 11];11:651. Available from: http://www.cancerjournal.net/text.asp?2015/11/3/651/140814


 > Introduction Top


Primary intraosseous carcinoma (PIOC) is a rare squamous cell carcinoma arising within the bone with no initial connection with the oral mucosa either from a previous odontogenic cyst or de novo rather than from a pre-existing epithelial lesion. [1] It was first described by Loos in 1913 as a central epidermoid carcinoma of the jaw. [2] The etiology of this tumor is still not clear, it is thought be arising from the remnants of odontogenic epithelium, epithelial rests of Malassez. The present report describes a case of PIOC of mandible arising de novo in a 76-year-old-man, which was initially treated by a general dentist, who misdiagnosed as a periodontal lesion.


 > Case report Top


A 76-yr-old-male patient reported with a complaint of pain and swelling in his right lower back teeth region of his jaw since 15 days. Presenting illness revealed that patient was asymptomatic 6months back, later developed pain in the right lower back tooth region of his jaw followed by swelling in the same region, which gradually increased to present size. Patient noted gradual deviation of jaw to right side since 15 days.

On extra oral clinical examination, there was gross facial asymmetry with jaw deviated to the right side [Figure 1] and [Figure 2]. On temporomandibular joint (TMJ) palpation, Condyle was not palpable on right side and condylar movements were not felt. Palpable right submandibular lymph node, one in number, firm, freely mobile and nontender. Intraoral examination revealed a diffuse swelling on right buccal vestibule [Figure 3], which was slight tender and soft in consistency on palpation. Associated tooth was fractured with grade II mobility irt 46. Thus, it was provisionally diagnosed as dento-alveolar abscess with respect to 46. For further evaluation, following set of radiographic investigations were done.
Figure 1: Extra oral clinical photograph showing deviation of jaw to right side

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Figure 2: Extra oral clinical photograph showing diffuse swelling on right lower third of face

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Figure 3: Intraoral clinical photograph showing diffuse swelling in retro molar region with an intact alveolar mucosa with areas of pigmentation and depigmentation

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Intraoral periapical (IOPA) radiograph revealed [Figure 4] a gross destruction of interdental and interradicular bones with radiopaque flecks of remnant bone seen with respect to 46 giving an appearance of floating tooth. Occlusal radiograph showed a destruction of buccal and lingual cortical plates with remnants of bone on the buccal aspect [Figure 5]. Orthopantomogram [Figure 6] showed a complete destruction of bone anterio-posteriorly involving symphysis, mandibular ramus, Condyle and coronoid process on right side with radiopaque flecks of remnant bone. A screening chest radiograph was made to rule out any primary lesion or any metastatic lesions in the lungs however, chest radiograph appeared to be normal.
Figure 4: IOPA radiograph of 46 region showing severe bone destruction with flecks of bone remnants appearing as floating tooth

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Figure 5: Occlusal radiograph showing destruction of buccal and lingual cortex

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Figure 6: Panoramic radiograph showing destruction of mandible on right side involving body, ramus and condylar region

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Multiple detector computed tomography (MDCT) in axial section [Figure 7]a showed diffuse swelling and bulky masseter and pterygoid muscle suggestive of infiltration. Extension of the lesion till the midline of the mandible and buccal and lingual cortical break can be seen [Figure 7]b MDCT in sagital section showed destruction of the complete condyle on right side [Figure 8]a and normal condyle on left side [Figure 8]b. MDCT in coronal section [Figure 9]a showed presence of condyle on left side and absence of condyle on the right side. MDCT in coronal section [Figure 9]b showed presence of coronoid and ramus on left side and resorption of coronoid and ramus on the right side. 3D MDCT [Figure 10] section showed gross destruction of mandible on right side with flecks of remaining bone seen. A radiographic differential diagnosis of primary intraosseous carcinoma and oral squamous cell carcinoma was arrived.
Figure 7: (a) Axial CT section - destruction of mandible on right side involving body, angle and ramus of the mandible (b) Axial CT section - extension of the lesion within the mandible till midline with break in buccal and lingual cortex

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Figure 8: (a) R- Sagital CT section of right side with destruction of condyle (b) L- Sagital CT section of left side with normal condyle

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Figure 9: (a) Coronal CT section - Shows presence of condyle on left side and absence of condyle on right side (b) Coronal CT section - Shows presence of coronoid and ramus of mandible on left side and absence of coronoid and ramus of mandible on right side

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Figure 10: 3D reconstructed CT image shows gross destruction on mandible on right side with flecks of remaining bone

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Furthermore, histopathological report of incised biopsy specimen revealed epithelial cells proliferating in the connective tissue stroma in the form of sheets, nests and islands. Tumor cells with hyperchromatic nuclei and abundant keratin formation is seen with some areas of keratin pearl formation [Figure 11], [Figure 12] and [Figure 13]. Hence based on clinical, radiological and histopathological features, the present case was diagnosed as primary intraosseous carcinoma.
Figure 11: Scanner view showing epithelial cells proliferating in the connective tissue stroma in the form of sheets (blue arrow) and islands (yellow arrow)

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Figure 12: x10 view showing tumor cells with hyperchromatic nuclei (yellow arrow) and abundant keratin formation (blue arrow)

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Figure 13: x10 view showing epithelial cells proliferating in connective tissue stroma in the form of sheets, nests and islands with hyperchromatic nuclei (yellow arrow) and with some areas of keratin pearl formation (green arrow)

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 > Discussion Top


According to WHO, PIOC is defined as a squamous cell carcinoma arising within the jawbone without connection to the oral mucosa, probably from odontogenic epithelial residues. [3] Pindborg in 1971coined the term PIOC. [4] Waldron and Mustoe suggested adding intraosseous mucoepidermoid carcinoma to the previous classification [Table 1]. [5]
Table 1: Classification of PIOC according to walderon and mustoe


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According to Narcisse Zwetyenga et al., patients ranged between the age group of 4 to 76 years (mean, 54 years). [6] It occurs more frequently in the mandible (especially the posterior section) than in the maxilla and have a male to female ratio of 2.5:1. [7] Suei et al., suggested following criteria for diagnosis of PIOC as: (a) intact oral mucosa before diagnosis; (b) microscopic evidence of squamous cell carcinoma without a cystic component or other odontogenic tumor cells; and (c) absence of another primary tumor on chest radiographs obtained at the time of diagnosis and during a follow-up period of more than 6 months. [8] The finding of an intact mucosa in the present case made the possibility of direct extension of squamous cell lesions from the oral mucosa appear unlikely. Hence, the tumor described in this paper completely fulfilled the above mentioned strict criteria and our case represented the feature of type 3a according to the classification given by Waldron and Mustoe.

According to Thomas et al., pain was the most common presenting feature followed by swelling of the jaw and sensory disturbances, which were similar to the one reported by Zwetyenga et al. [6],[7] Paresthesia as seen in present case along with accelerated growth, trismus and deviation of jaw because of muscle infiltration are also common symptoms.

According to Thomas et al., PIOCs have varied radiographic presentations like cup- or dish-shaped patterns, well-defined lesions, small radiolucent loculations and poorly defined mouth eaten appearance. [7] Zwetyenga et al., noted osteolytic bone changes with irregular or regular margins and pathological fractures. [6] The internal structure is usually radiolucent without any new bone production and sometimes residual bone can be seen within the radiolucency. Root resorption is unusual. Teeth that lose both lamina dura and the supporting bone appear to be 'floating' in space as seen in our case. [2]

The disease is rare; therefore, the prognosis is hard to estimate and the importance should be given to early diagnosis so that suitable treatment can be given at the earliest. To et al., reported a 46% of survival for periods ranging from 6 months to 5 years. [9] PIOCs originating from odontogenic cysts have a better prognosis than the de novo lesions. [10]

 
 > References Top

1.
Sivapathasundharam B, Sivakumar G. Primary de novo intraosseous carcinoma of the mandible--report of a case and review of literature. Indian J Dent Res 2004;15:103-6.  Back to cited text no. 1
[PUBMED]    
2.
Huang JW, Luo HY, Li Q, Li TJ. Primary intraosseous squamous cell carcinoma of the jaws clinicopathologic presentation and prognostic factors. Arch Pathol Lab Med 2009;133:1834-40.  Back to cited text no. 2
    
3.
Kramer IR, Pindborg JJ, Shear M. Histologic typing of odontogenic tumors. 2 nd ed. Berlin: World Health Organization, Springer-Verlag; 1992. p. 112-6.  Back to cited text no. 3
    
4.
Pindborg JJ, Kramer IR, Torloni H. Histologic typing of odontogenic tumors, jaw cysts and allied lesions. Geneva: World Health Organization 1971. p. 35-6.  Back to cited text no. 4
    
5.
Lin YJ, Chen CH, Wang WC, Chen YK, Lin LM. Primary intraosseous carcinoma of the mandible. Dentomaxillofac Radiol 2005;34:112-6.  Back to cited text no. 5
    
6.
Zwetyenga N, Pinsolle J, Rivel J, Majoufre-Lefebvre C, Faucher A, Pinsolle V. Primary intraosseous carcinoma of the jaws. Arch Otolaryngol Head Neck Surg 2001;127:794-7.  Back to cited text no. 6
    
7.
Thomas G, Pandey M, Mathew A, Abraham EK, Francis A, Somanathan T, et al. Primary intraosseou carcinoma of the jaw: pooled analysis of world literature and report of two new cases. Int J Oral Maxillofac Surg 2001;30:349-55.  Back to cited text no. 7
    
8.
Suei Y, Tanimoto K, Taguchi A, Wada T. Primary intraosseous carcinoma: Review of the literature and diagnostic criteria. J Oral Maxillofac Surg 1994;52:580-3.  Back to cited text no. 8
    
9.
To EH, Brown JS, Avery BS, Ward-Booth RP. Primary intraosseous carcinoma of the jaws: Three new cases and a review of the literature. Br J Oral Maxillofac Surg 1991;29:19-25.  Back to cited text no. 9
    
10.
Tiwari M. Primary intraosseous carcinoma of the mandible: A case report with literature review. J Oral Maxillofac Pathol 2011;15:205-10.  Back to cited text no. 10
[PUBMED]  Medknow Journal  


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