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CORRESPONDENCE
Year : 2019  |  Volume : 15  |  Issue : 3  |  Page : 700-703

A mixed radiopaque – radiolucent lesion in the anterior mandible associated with multiple impacted teeth: A radiodiagnostic challenge?


1 Department of Oral Medicine and Radiology, A.J. Institute of Dental Sciences, Mangalore, Karnataka, India
2 Department of Pedodontia, A.J. Institute of Dental Sciences, Mangalore, Karnataka, India
3 Department of Oral Pathology, A.J. Institute of Dental Sciences, Mangalore, Karnataka, India

Date of Web Publication29-May-2019

Correspondence Address:
Dr. Gowri Pandarinath Bhandarkar
Department of Oral Medicine and Radiology, A.J. Institute of Dental Sciences, Kuntikan, Mangalore - 575 004, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcrt.JCRT_721_16

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 > Abstract 


Desmoplastic ameloblastoma (DA) exhibits important differences in gender, anatomic distribution, radiographic findings, and histologic appearance compared to other types of ameloblastoma. Radiologically, DA is seen either as ill-defined mass containing osteolytic and sclerotic areas or as multifocal radiodense flecks within radiolucent background resembling a honeycomb. The radiographic differential diagnosis includes fibro-osseous lesions such as cemento-ossifying fibroma, fibrous dysplasia, calcifying odontogenic cyst, and chronic sclerosing osteomyelitis. Thus, DA should primarily be included in the differential diagnosis of a mixed radiopaque-radiolucent lesion with diffuse borders in the anterior premolar region of the jaws. This report adds to the literature of mixed radiolucent-radiopaque lesions which may not always be histopathologically diagnosed as a fibro-osseous lesion but could turn out to be a DA. This report also benefits the dental community by cautioning them to be aware of DA that can be associated with multiple unerupted teeth which is quite a rare finding.

Keywords: Anterior jaw, desmoplastic ameloblastoma with osteoplasia variant, desmoplastic ameloblastoma, diagnosis, mixed radiolucent, radiopaque lesion, unerupted teeth


How to cite this article:
Bhandarkar GP, Shetty KV, Desai D. A mixed radiopaque – radiolucent lesion in the anterior mandible associated with multiple impacted teeth: A radiodiagnostic challenge?. J Can Res Ther 2019;15:700-3

How to cite this URL:
Bhandarkar GP, Shetty KV, Desai D. A mixed radiopaque – radiolucent lesion in the anterior mandible associated with multiple impacted teeth: A radiodiagnostic challenge?. J Can Res Ther [serial online] 2019 [cited 2019 Jun 16];15:700-3. Available from: http://www.cancerjournal.net/text.asp?2019/15/3/700/244232




 > Introduction Top


Ameloblastoma is the most common benign odontogenic tumor of epithelial tissue origin. Desmoplastic ameloblastoma (DA) is a variant. Eversole et al.[1] gave the first detailed report in the English literature in 1984. He described three cases and called it “ameloblastoma with pronounced desmoplasia.”

The growing knowledge regarding the clinical and radiographic presentation and pathology of DAs has led to inclusion as a distinct variant of ameloblastoma in the World Health Organization classification of odontogenic tumors.[2]


 > Case Report Top


A 35-year-old female reported to our department with a swelling in the lower anterior region of the jaw for 6 months.

History dates back to 6 months when she noticed a small swelling that spontaneously increased to the present size associated with mobility of anterior teeth and without bleeding or sensory changes. There was no history of trauma, and the past medical and dental histories were unremarkable and noncontributory. On general examination, no lymphadenopathy or fistulae were present.

Extraoral examination revealed a well-defined solitary swelling in the region of the symphysis that was oval in shape measuring about 6 cm × 4 cm in size. The skin over the swelling appeared smooth, and borders were well defined with obliteration of the mental fold and with no signs of paresthesia. On intraoral examination [Figure 1], the swelling was nontender. The consistency of the swelling was bony hard. 31, 32, 41, and 42 were of Grade 2 mobility and showed a negative response to vitality test. 23, 33, and 43 were missing without any history of extraction.
Figure 1: Swelling of the anterior mandible extending from 45 to 34 with marked obliteration of the labial vestibule. Expansion of both labial and lingual cortical plates was evident. The mucosa over the swelling was smooth, intact and of normal mucosal color. 33 and 43 were missing

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A provisional diagnosis of the adenomatoid odontogenic tumor was given. The differential diagnosis had been given as DA, calcifying odontogenic cyst, and fibro-osseous lesion such as cemento-ossifying fibroma.

Mandibular occlusal radiograph of the lesion revealed the presence of a single, ill-defined, mixed radiopaque-radiolucent lesion [Figure 2]. It was extending from the mesial aspect of 34 to the distal aspect of 45 with the marked expansion of the labial cortical plate and mild expansion of the lingual cortical plate. There were small flecks of radio-opacities seen against radiolucent background within the lesion. There was displacement of 41 and 42. Unerupted 33 was seen placed lingually. A panoramic radiograph revealed a complete absence of normal trabeculae with the presence of a diffuse, ill-defined, mixed radiolucent-radiopaque lesion in the mandible [Figure 3]. It had poorly defined borders extending from 34 crossing the midline to 46 region. It showed unerupted 33 and 43 lying almost close to the inferior border of the mandible in the symphyseal and parasymphyseal region, respectively. The lesion caused divergence of roots of 41 and 42, and slight resorption of apical roots of 31, 32, 41, and 42 with loss of lamina dura. Impacted 23 and mesiodens were also seen. The patient was referred for routine blood investigations. Hematological and biochemical values were within normal range.
Figure 2: Mandibular occlusal radiograph revealing the expansion of the labial and lingual cortical plates. There were small flecks of radio-opacities seen against radiolucent background within the lesion. There was displacement of 41 and 42. Unerupted 33 was seen placed lingually. Lesion was seen extending from 34 to 45

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Figure 3: Panoramic radiograph revealed a complete absence of normal trabeculae with the presence of a mixed radiolucent-radiopaque lesion in the mandible. It had poorly defined borders extending from 34 crossing the midline to 46 region. It showed unerupted 33 and 43 lying almost close to the inferior border of the mandible in the symphyseal and parasymphyseal region, respectively, and 23 in the maxilla. The lesion caused displacement of roots of 41 and 42 and resorption of roots of 31, 32, 41, and 42 with loss of lamina dura

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With an informed consent, an incisional biopsy was performed, and the histopathological report was suggestive of DA with new bone formation [Figure 4].
Figure 4: Showing ameloblastic epithelium surrounded by dense collagenous fibrous stroma with abundant new bone formation (low power)

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Under G A and proper antibiotic coverage, the tumor was operated, anterior mandible resected and reconstructed using a rib graft. The gross surgical specimen on histopathological examination confirmed the diagnosis of desmoplastic variant of ameloblastoma. The patient's postoperative period was uneventful, and postoperative radiograph [Figure 5] and clinical follow-up examination of the patient for 2 years disclosed no evidence of recurrence or residual tumor. Later, 23 and mesiodens were extracted.
Figure 5: Postoperative radiograph

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All photos were taken with the adequate understanding and informed consent of the patient.


 > Discussion Top


The incidence of DA is 0.9%–12.1% of all the ameloblastomas. Kaffe et al.[3] reported the following findings:

The mean age is 42.3 years (range 17–70 years) and is more common in females (as was seen in our case) and in Asians. DA develops from epithelial rests of Malassez in the periodontal membrane of the related tooth.

Approximately half of the desmoplastic lesions has been located in the maxilla in the anterior or premolar region.[4] In our case, DA was located in the anterior mandible. Higuchi et al. postulated that ameloblastoma in the tooth-bearing area had a greater tendency to have an abundant stroma and to be desmoplastic.

The radiographic appearance of this neoplasm is that of a mixed radiolucent-radiopaque lesion which suggests that the tumor may be more aggressive than other variants of ameloblastoma and expresses the infiltrative nature of the tumor attributing to the ill-defined borders. The above appearance is due to the infiltration of tumor cells into the adjacent marrow spaces with simultaneous vigorous osteoblastic activity.[2] There is involvement of lamina dura. Tooth displacement was a common feature of this variant (92% of the cases). Root resorption had been discovered in only 33% of the cases. All these were noted in our case also.

Islands of ameloblastic appearing columnar cells that surround spindle-shaped cells that resemble stellate reticulum and stroma with marked desmoplasia characterize the histological pattern.[4]

It is speculated that the desmoplasia in DA might act as a limiting barrier for the local spread of the DA tumor cells compared to the situation in conventional ameloblastoma where such barrier is absent.[5] Histologically, the DA cases were grouped into the following two types: (1) simple desmoplastic type which is predominant (88.0%), showing histologic features of extensive desmoplasia of the stroma and (2) desmoplastic with osteoplasia type which is rare (12.0%), showing additional calcific structures.[5] This explains the radiologic appearance.[6] Our case is an example of DA with osteoplasia variant which is a rare type.

Philipsen et al.[7] remarked that DA has a tendency to produce a de novo synthesis of extracellular fibrous protein, which has been attributed to desmoplasia seen in this tumor. Furthermore, the de novo synthesized extracellular protein could serve as a nidus for calcification seen in the DA with osteoplasia.

Compared with the superficial muscular aponeurotic system, Ng and Siar using various immunohistochemical studies have identified DA tumor cells as showing variable expression of S-100 protein and desmin. There was also a high expression of caspase-3 and Fas, decreased expression of CK19, and high expression of p63.[6] The desmoplastic stroma of DA has been reported to show a strong positive reaction to collagen Type VI,[7] immunonegativity for tenascin, and strong immunopositive reaction for fibronectin and Type I collagen.[6] There was also marked immunoexpression of transforming growth factor-beta, one of the most potent local factors for modulating extracellular matrix formation, thus playing a role in the desmoplastic matrix formation.[6]

DA has a potential for recurrence (21.4%) because it fails to form a connective tissue capsule.[3] Curettage often leaves islands of the tumor within the bone, which later manifest as recurrences.[8] Thus, resection is the most sought-after option for DA.


 > Conclusion Top


This report attempts to help the clinicians in developing familiarity with the clinical presentation of DA and recognize such cases and also contributes to the dentists a better understanding of this variation in an odontogenic tumor.

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.
Eversole LR, Leider AS, Hansen LS. Ameloblastomas with pronounced desmoplasia. J Oral Maxillofac Surg 1984;42:735-40.  Back to cited text no. 1
    
2.
Kishino M, Murakami S, Fukuda Y, Ishida T. Pathology of the desmoplastic ameloblastoma. J Oral Pathol Med 2001;30:35-40.  Back to cited text no. 2
    
3.
Kaffe I, Buchner A, Taicher S. Radiological features of desmoplastic variant of ameloblastoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1993;76:525-9.  Back to cited text no. 3
    
4.
Higuchi Y, Nakamura N, Ohishi M, Tashiro H. Unusual ameloblastoma with extensive stromal desmoplasia. J Craniomaxillofac Surg 1991;19:323-7.  Back to cited text no. 4
    
5.
Effiom OA, Odukoya O. Desmoplastic ameloblastoma: Analysis of 17 Nigerian cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;111:e27-31.  Back to cited text no. 5
    
6.
Yazdi I, Seyedmajidi M, Foroughi R. Desmoplastic ameloblastoma (a hybrid variant): Report of a case and review of the literature. Arch Iran Med 2009;12:304-8.  Back to cited text no. 6
    
7.
Philipsen HP, Ormiston IW, Reichart PA. The desmo – And osteoplastic ameloblastoma. Histologic variant or clinicopathologic entity? Case reports. Int J Oral Maxillofac Surg 1992;21:352-7.  Back to cited text no. 7
    
8.
Mintz S, Velez I. Desmoplastic variant of ameloblastoma: Report of two cases and review of the literature. J Am Dent Assoc 2002;133:1072-5.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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