Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Contacts Login 
Home Print this page Email this page Users Online: 70



 
 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 4  |  Issue : 1  |  Page : 18-21

Sunburst appearance in odontogenic myxoma of mandible: A radiological diagnostic challenge


1 Department of Oral Medicine and Radiology, Subharti Dental College and Hospital, Meerut, Uttar Pradesh, India
2 Department of Oral Pathology, Subharti Dental College and Hospital, Meerut, Uttar Pradesh, India

Date of Web Publication19-Feb-2016

Correspondence Address:
Ravi Prakash Sasankoti Mohan
Department of Oral Medicine and Radiology, Subharti Dental College and Hospital, Meerut, Uttar Prades
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2321-3841.177065

Rights and Permissions
  Abstract 

Odontogenic myxoma in the jaws is infrequent, accounting for only 3%-6% of odontogenic tumours. The radiological appearance of this tumor is complicated and there exist controversies regarding its internal structure and locularity. The "sunray" or "sunburst" appearance in Odontogenic myxoma has been reported in the literature although rarely. Here we report a case of odontogenic myxoma in the mandible showing sunburst appearance which posed a diagnostic dilemma for the oral radiologists. We also emphasize the usefulness of Cone Beam Computed Tomography in the diagnosis of such lesions.

Keywords: Cone beam computed tomography, mandible, odontogenic myxoma, sunburst appearance


How to cite this article:
Kidwai SM, Mohan RS, Wadhwan V, Kamarthi N, Goel S, Gupta S. Sunburst appearance in odontogenic myxoma of mandible: A radiological diagnostic challenge. J Oral Maxillofac Radiol 2016;4:18-21

How to cite this URL:
Kidwai SM, Mohan RS, Wadhwan V, Kamarthi N, Goel S, Gupta S. Sunburst appearance in odontogenic myxoma of mandible: A radiological diagnostic challenge. J Oral Maxillofac Radiol [serial online] 2016 [cited 2022 Jan 27];4:18-21. Available from: https://www.joomr.org/text.asp?2016/4/1/18/177065


  Introduction Top


Myxomas of the head and neck are rare tumors. Odontogenic myxoma (OM) is a rare, nonencapsulated benign but locally invasive odontogenic tumor first described by Thoma and Goldman in 1947. [1] It represents 3%-6% of all odontogenic tumors. [1],[2] The evidence for its odontogenic origin arises from its almost exclusive location in the tooth bearing areas of the jaws, its occasional association with missing or unerupted teeth, and the presence of odontogenic epithelium. [2],[3],[4]

Most of the reported cases of OM are in young adults usually in their second and third decade of life and these lesions have marked female predilection. Although intraosseous myxoma has been reported in various anatomical sites, the majority of these tumors occur in the mandible, followed by the maxilla. [1],[3],[4],[5]

In the facial region, OM occurs mostly within the bone and radiologic examination is therefore important. Generally, diagnosis of OM is made by the examination of conventional radiographs (CRs) and confirmed by histopathology. Advanced technologies such as computed tomography (CT), Cone Beam Computed Tomography (CBCT) and magnetic resonance imaging (MRI) may offer diagnostic options, which could overcome some of the limitations of CR. [5],[6],[7]

The radiological appearance of the OM is more complicated than has generally been thought. [5] Indeed, there is considerable debate and controversy regarding the internal structure and locularity of the tumor. Radiologically, the appearance may vary from a unilocular radiolucency to a multicystic lesion with well-defined or diffused margins with fine, bony trabeculae within its interior structure expressing a "honey combed," "soap bubble," or "tennis racket" appearance. [6],[7],[8] The "sunray" or "sunburst" appearance in OM has been reported in the literature although rare. Here we report a case of odontogenic myxoma showing sunray appearance which posed a diagnostic dilemma for the oral radiologists.


  Case Report Top


A 28 year old male of Asian origin reported with pain in the right lower back tooth region since past 6 months. History revealed that he had undergone tooth extraction due to deep caries which was causing the pain in the same region six months back after which he developed a swelling in that region. He visited a local dentist for the same who performed an incisional biopsy in the right mandibular molar area. The biopsy report confirmed the swelling as peripheral fibroma. However, the pain did not subside and even the biopsy site remained unhealed for one month. Patient had no significant medical history. He was non smoker and non alcoholic. On extraoral examination there was no gross facial asymmetry and lymph nodes were non palpable. However, on palpation there was a bony hard swelling in the right lower border of mandible. Intraoral examination revealed a non indurated shallow ulcer on the alveolar ridge with a hard swelling of approx 3 cm × 3 cm in size due to both buccal and lingual cortical expansion [Figure 1]. The mucosa overlying the swelling was pale pink in color with few areas showing keratosis due to secondary trauma from opponent teeth. The area was mildly tender. On hard tissue examination, 37, 47 and 48 were missing. On the basis of history of extraction and clinical presentation of non healing ulcerative area and swelling, a provisional diagnosis of osteomylitis was made. On conventional panoramic examination, a mixed radiolucent and radiopaque lesion was seen with severe bony resorption in the right body of the mandible. Moreover there were radioapaque bony spicules emanating from the inferior aspect of the lesion giving a characteristic sun ray/sunburst appearance. Mandibular lateral occlusal projection revealed buccal and lingual cortical expansion and internal septas [Figure 2]. Cone beam computed tomography views demonstrated the exact extent of the lesion along with clear visualization of internal septas and periosteal reaction [Figure 3]. The 3D reconstruction further helped in visualizing the lesion in three dimensions [Figure 4]. The radiological differential diagnosis made was metastatic lesion, osteosarcoma, odontogenic myxoma, ossifying fibroma or central hemangioma. Consent form was filled by the patient and complete surgical excision was performed and the excised specimen was sent for histopathological examination. The gross excised specimen was gelatinous and slippery. Radiograph of the specimen clearly exhibited internal septae and bony spicules [Figure 5]. Histopathological examination revealed hyperplastic stratified squamous epithelium with stellate, spindle, round cells in loose myxoid stroma. Odontogenic epithelial rests and mixed inflammatory cells were also evident. Moreover, myxoid matrix stained positively with alcian blue [Figure 6]. Thus histopathology confirmed the final diagnosis of odontogenic myxoma. The patient is on regular follow up without any discomfort.
Figure 1: A non indurated shallow ulcer on the alveolar ridge with mild cortical expansion

Click here to view
Figure 2: Mandibular lateral occlusal projection showing buccal and lingual cortical expansion and internal septas

Click here to view
Figure 3: Cone beam computed tomography views showing the extent of the lesion along with clear visualization of internal septas and sunburst appearance

Click here to view
Figure 4: The 3D reconstruction visualizing the lesion in three dimensions showing clearly the bony spicules emanating from the inferior aspect of the lesion giving sunburst appearance

Click here to view
Figure 5: Radiograph of the excised specimen

Click here to view
Figure 6: Histopathological examination showing myxoid matrix staining positively with alcian blue and hyperplastic stratified squamous epithelium with stellate, spindle, round cells in loose myxoid stroma

Click here to view



  Discussion Top


Odontogenic myxoma is a rare aggressive intraosseous lesion derived from embryonic mesenchymal tissue associated with odontogenesis primarily consists myxomatous ground substance with widely scattered undifferentiated spindled mesenchymal cells. [4] Clinically, odontogenic myxoma is a benign, painless, invasive, slowly enlarging mass. Large lesions can cause marked asymmetry of the face. It causes expansion of bony cortices, displacement and loosening of teeth. [1],[2],[6] In the present case, the lesion appeared as a bony hard swelling with expansion of bony cortices along with a non indurated shallow ulcer on the alveolar ridge. However, because there was history of extraction, swelling and presence of a non healing ulcer, we made osteomyelitis as our clinical diagnosis.

Zhang et al., classified radiographic appearances of OM into six types:

Type I: Unilocular well-defined radiolucency,
Type II (multilocular): Two or more compartments with multiple interlaced osseous trabeculae described as honey comb, soap bubble or tennis racquet radiolucency,
Type III: Lesion located in alveolar bone,
Type IV: Lesion involving the maxillary sinus,
Type V (moth eaten appearance): Larger radiolucent area with irregular borders,
Type VI: Combination of bone destruction and bone formation giving sun ray appearance. [1],[4] Our case is thus classified as Type VI.

The sunburst pattern is a classic finding in bone sarcomas such as osteosarcoma and chondrosarcoma. On close examination, these spicules are not truly straight, but instead form irregular, slightly wavy lines. [6] In literature such mimicking pattern is also reported in some odontomas, odontogenic myomas, solitary plasmacytomas, metastasis (especially from sigmoid colon and rectum), mandibular squamous cell carcinoma, haemangioma, tubercular osteomylitis and fibrous dysplasia although rarely. [6],[7] Therefore, in present case, we included osteosarcoma in our radiological differential diagnosis along with odontogenic myxoma.

On panoramic radiographs, expansion of the tumor can be assessed visually, taking into consideration the 2-dimensional (2-D) nature of the images and by degree of displacement of the associated teeth accompanied by distortion, thinning, and/or perforation of the cortex. [5],[8],[9] In some of the maxillary tumors the tumors growns silently inside the maxillary sinus, resulting in opacification, also reported by other investigators. [5],[8] MRI has the ability to demonstrate the tumors vividly with precise determination of the margins. The walls of the tumors and patterns of the growth are clearly depicted in this imaging modality. [5] It is mainly useful in case of maxillary tumors. In the present case, Panoramic radiograph was not very useful in visualization of internal aspect of the lesion. MRI was not performed in our case as it is more useful in maxillary tumors.

Computed tomographic images of odontogenic myxomas may show any osteolytic expansile lesions with mild enhancement of the solid portion of the mass or bony expansion and thinning of cortical plates with strong enhancement of the mass lesion. Cone beam CT of a limited area is very effective in reducing the radiation dose in patients and achieving high spatial resolution in comparison with conventional CT. Moreover, it has wide applications and can be used to determine the extent and condition of the internal structure of lesions, fracture of teeth roots, the relationship between the apex of adjacent teeth and the mandibular canal, the condition of impacted teeth, diagnosis of temporomandibular joint disorders and the condition of alveolar bone when planning implant treatment. [7] An advantage of CT and CBCT is the ability for 3-D modeling which are valuable in assessing the true extent of the tumor, particularly with reference to planning reconstructive procedures as seen with the present case. [5]

The excised mass exhibits a whitish, translucent, mucinous appearance. [7] Histologically the WHO (1992) defined OM as "A locally invasive neoplasm consisting of rounded and angular cells lying in an abundant mucoid stroma". [1],[2],[4] They show loose myxoid stroma containing spindle-shaped, angular or round cells. Polymorphic cells or nuclei are rarely encountered. Stroma shows very few fine fibrillar material and minimum vascularity. Inflammatory infiltration and remnants of odontogenic epithelium are occasionally seen. [1],[2],[4] The ground substance of odontogenic myxomas has been shown to consist of about 80% hyaluronic acid and 20% chondroitin sulfate. The myxoid intercellular matrix stains positively with alcian blue as seen with the present case. [10] Immunohistochemical studies demonstrated positive reaction with antibodies to vimentin and actin, and negative reaction to antibody to S-100 protein. [10]

The aggressive nature of OM is well documented in the literature. The tumor is not radiosensitive, and the surgery is the treatment of choice. Radical treatment of en-block resection is advised by most authors over conservative treatment due to its invasive nature, large size, and recurrence history. [2],[11],[12] The prognosis is usually good. However recurrence ranging from 10%-33% is reported with conservative treatment. [11],[12]


  Conclusion Top


In conclusion, the importance of radiology in the diagnosis of OM cannot be ruled out as this tumor occurs inside the bone and can reach a considerable size with little or no clinical manifestations. In addition, good imaging is essential in visualization of tumor's boundaries before surgical treatment. Conventional radiographs, CT, CBCT and MRI whenever indicated, are essential in the diagnosis of OM to see the extension of the tumors, status of cortication, expansion, locularity, and extension into the surrounding structures. However, definitive diagnosis can only be made through histopathological examination in addition to clinical and radiographic examinations. The present case adds to literature another rare case of odontogenic myxoma showing sunburst appearance.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Zhang J, Wang H, He X, Niu Y, Li X. Radiographic examination of 41 cases of odontogenic myxomas on the basis of conventional radiographs. Dentomaxillofac Radiol 2007;36:160-7.   Back to cited text no. 1
    
2.
Reddy SP, Naag A, Kashyap B. Odontogenic myxoma: Report of two cases. Natl J Maxillofac Surg 2010;1:183-6.   Back to cited text no. 2
[PUBMED]  Medknow Journal  
3.
Simon EN, Merkx MA, Vuahaula E, Ngassapa D, Stoelinga PJ. Odontogenic myxoma: A clinicopathological study of 33 cases. Int J Oral Maxillofac Surg 2004;33:333-7.  Back to cited text no. 3
    
4.
Rani V, Mahaboob Kadar MK, Babu A, Sankari L, Krishnasamy G. Odontogenic myxoma diagnostic dilemma: A case report and review of literature. J Cranio Max Dis 2014;3:163-7.  Back to cited text no. 4
  Medknow Journal  
5.
Kheir E, Stephen L, Nortje C, van Rensburg LJ, Titinchi F. The imaging characteristics of odontogenic myxoma and a comparison of three different imaging modalities. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:492-502.  Back to cited text no. 5
    
6.
White SC, Pharoah MJ. Oral radiology principles and interpretation. 5 th edn. St. Louis, MO: Mosby; 2004. p. 433-4.  Back to cited text no. 6
    
7.
Araki M, Kameoka S, Matsumoto N, Komiyama K. Usefulness of cone beam computed tomography for odontogenic myxoma. Dentomaxillofac Radiol 2007;36:423-7.  Back to cited text no. 7
    
8.
Kaffe I, Naor H, Buchner A. Clinical and radiological features of odontogenic myxoma of the jaws. Dentomaxillofac Radiol 1997;26:299-303.  Back to cited text no. 8
    
9.
Peltola J, Magnusson B, Happonen RP, Borrman H. Odontogenic myxoma - A radiographic study of 21 tumours. Br J Oral Maxillofac Surg 1994;32:298-302.  Back to cited text no. 9
    
10.
Singaraju S, Wanjari SP, Parwani RN. Odontogenic myxoma of the maxilla: A report of a rare case and review of the literature. J Oral Maxillofac Pathol 2010;14:19-23.  Back to cited text no. 10
[PUBMED]  Medknow Journal  
11.
Lo Muzio L, Nocini P, Favia G, Procaccini M, Mignogna MD. Odontogenic myxoma of the jaws: A clinical, radiologic, immunohistochemical, and ultrastructural study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1996;82:426-33.  Back to cited text no. 11
    
12.
Adebayo ET, Ajike SO, Adekeye EO. A review of 318 odontogenic tumors in Kaduna, Nigeria. J Oral Maxillofac Surg 2005;63:811-9.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]


This article has been cited by
1 The Rare Radiographic Sunburst Appearance of Odontogenic Myxomas: A Case Report and Review of the Literature
Jamie A. White,Naomi Ramer,Todd R. Wentland,Molly Cohen
Head and Neck Pathology. 2020;
[Pubmed] | [DOI]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Report
Discussion
Conclusion
References
Article Figures

 Article Access Statistics
    Viewed8154    
    Printed107    
    Emailed0    
    PDF Downloaded646    
    Comments [Add]    
    Cited by others 1    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]