Journal of Pharmacy And Bioallied Sciences
Journal of Pharmacy And Bioallied Sciences Login  | Users Online: 601  Print this pageEmail this pageSmall font sizeDefault font sizeIncrease font size 
    Home | About us | Editorial board | Search | Ahead of print | Current Issue | Past Issues | Instructions | Online submission




 
 Table of Contents  
DENTAL SCIENCE - CASE REPORT
Year : 2015  |  Volume : 7  |  Issue : 6  |  Page : 759-762  

Odontogenic myxoma of maxilla: A rare presentation in an elderly female


1 Department of Oral and Maxillofacial Surgery, Adhiparasakthi Dental College, Melmaruvathur, Tamil Nadu, India
2 Department of Oral and Maxillofacial Pathology, Adhiparasakthi Dental College, Melmaruvathur, Tamil Nadu, India
3 Department of Endodontics, Adhiparasakthi Dental College, Melmaruvathur, Tamil Nadu, India
4 Department of Oral and Maxillofacial Pathology, Chettinad Dental College, Chennai, Tamil Nadu, India

Date of Submission28-Apr-2015
Date of Decision28-Apr-2015
Date of Acceptance22-May-2015
Date of Web Publication1-Sep-2015

Correspondence Address:
C Sreeja
Department of Oral and Maxillofacial Pathology, Adhiparasakthi Dental College, Melmaruvathur, Tamil Nadu
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-7406.163550

Rights and Permissions
   Abstract 

Odontogenic myxomas are rare benign neoplasm of mesenchymal origin, comprising 3-6% of all odontogenic tumors. They are slow growing, non-metastasizing, often asymptomatic with local aggressiveness due to its infiltrative nature and hence high recurrence rate, with a high incidence of occurrence in the mandible. Most frequently occurs in second to third decade of life, seldom occurs beyond these age groups. Hereby, we present a case of odontogenic myxoma occurring in the maxilla in a 65-year-old female managed by partial maxillectomy.

Keywords: Maxilla, odontogenic myxoma, odontogenic tumors


How to cite this article:
Vijayabanu B, Sreeja C, Bharath N, Aesha I, Kannan V S, Devi M. Odontogenic myxoma of maxilla: A rare presentation in an elderly female. J Pharm Bioall Sci 2015;7, Suppl S2:759-62

How to cite this URL:
Vijayabanu B, Sreeja C, Bharath N, Aesha I, Kannan V S, Devi M. Odontogenic myxoma of maxilla: A rare presentation in an elderly female. J Pharm Bioall Sci [serial online] 2015 [cited 2019 Aug 25];7, Suppl S2:759-62. Available from: http://www.jpbsonline.org/text.asp?2015/7/6/759/163550

Odontogenic myxomas are benign tumors, non-metastasizing, nonencapsulated, derived from the primitive mesenchymal tissue of dental follicle, dental papilla or periodontal ligament. [1] WHO defines odontogenic myxoma as a benign, locally invasive neoplasm characterized by rounded and angular cells lying in an abundant mucoid stroma that replaces the cancellous bone expanding the cortex. [2] Clinically present as a slow-growing, locally aggressive lesion, often remains asymptomatic and hence reach considerable size even before patient become aware of it and seek treatment. [3] It more frequently affects the mandible than maxilla in the ratio of 3:1. Commonly occurs in the age group of second to a fourth decade, more often in females than in males. Rarely occurs below 10 years of age and older than 50 years. [4]

Odontogenic maxillary myxoma was first described by Thoma and Goldman in 1947, which is less frequent and behaves more aggressively than mandible, as it spreads through maxillary sinus, causing loosening and displacement of tooth and less frequently root resorption and exophthalmos and nasal obstruction in severe cases. [5],[6]

Radiographically, it presents a varied appearance ranging from unilocular radiolucency to multilocular and mixed radiolucent-radio opaque lesion. [3] Histologically, the mucoid stroma is responsible for its local infiltrative nature, and it may resemble few malignant lesions such as myxoid chondrosarcoma and malignant nerve sheath tumor. [7] Hence, it necessitates accurate clinical, radiological, and histopathological interpretation to arrive at a definitive diagnosis and effective management of the patient.


   Clinical Presentation Top


A 65-year-old female patient presented with a complaint of swelling over the left side of the cheek for the past 6 months. Patient gave a history of the extraction of a left back tooth due to mobility 3 months back, followed by progressive increase in the size of the lesion accompanied by a dull pain and burning sensation with difficulty in swallowing.

Extraoral examination revealed a diffuse, ill-defined, firm swelling of size 5 cm × 4 cm involving the left cheek extending superioinferiorly from infraorbital rim to 2 cm above the inferior border of the mandible. Medially the lesion was found to extend to the lateral wall of the nose with obliteration of the nasolabial groove and laterally to about 1 cm in front of tragus; the skin over the lesion appeared normal and pinchable. The swelling was tender and afebrile.

Intraoral examination revealed an erythematous ulceroproliferative growth on left maxillary alveolus extending anteroposteriorly from 23 to the left maxillary tuberosity. Medially the lesion was found to extend 1 cm lateral to midpalatine raphae and laterally extending buccally with obliteration of the vestibular sulcus. The entire occlusal surface of the left posterior teeth was covered by the lesion with an inferior extension of about 1 cm below the occlusal plane [Figure 1].
Figure 1: Ulceroproliferative growth involving left maxillary alveolus

Click here to view


Radiographically para nasal sinus (PNS) view of the skull shows haziness of the entire left maxillary sinus. Computed tomography (CT) scan of both axial and coronal views showed the lesion was confined to left maxillary sinus [Figure 2] and [Figure 3].
Figure 2: Computed tomography scan of coronal showing the lesion involving the left maxillary sinus

Click here to view
Figure 3: Computed tomography scan of axial views showing the lesion involving the left maxillary sinus

Click here to view


Considering the clinical and radiographic features a differential diagnosis of fibro-osseous lesion, central giant cell granuloma, ameloblastoma, and odontogenic myxoma were made. Patient was sending for a routine blood examination, which revealed normal parameters.

An incisional biopsy was performed, and the tissue was sent for histopathological examination. Microscopic examination of the hematoxylin and eosin stained tissue section showed a stroma composed of myxoid tissue with the spindle to stellate-shaped fibroblasts. Many odontogenic cell rests were present all over the stroma with loose collagen fiber bundles [Figure 4].
Figure 4: Stroma composed of myxoid tissue with spindle to stellate-shaped fibroblasts

Click here to view


On account of these features, the case was finally diagnosed as odontogenic myxoma. Hence, the patient was advised for surgical excision of the lesion under general anesthesia. A left side Weber Ferguson incision was placed, and partial maxillectomy was performed sparing the infraorbital rim, excising the lesion in toto, by keeping a margin of safety of 1 cm [Figure 5]. Reconstruction of the maxillary defect was done using an obturator [Figure 6]. The patient was followed up for 2 years and remained disease free.
Figure 5: Excised lesion in toto

Click here to view
Figure 6: Prosthetic reconstruction of the maxillary defect using obturator

Click here to view



   Discussion Top


Odontogenic myxoma, an uncommon benign tumor represents 3-6% of all odontogenic tumors with a high incidence of occurrence in the mandible (66.4%) than the maxilla (33.6%).

Myxomas can occur in various sites such as skin, subcutaneous tissue, heart, and head and neck. [1] Those arising in head and neck can be either derived from facial bone (osteogenic/odontogenic) or facial soft tissues, [8] with odontogenic origin been more common. They appear to originate from the mesenchymal tissue of dental papilla, follicle or periodontal ligament. Its odontogenic origin being evident by its high incidence of occurrence in tooth-bearing areas of jaws, occurrence in young individuals, association with missing or unerupted teeth and presence of the odontogenic epithelium. [2] Controversies still persists regarding its origin as being either as a myxomatous change of odontogenic fibroma or residual foci of embryonic tissue. [2],[9]

In 1863, Virchow coined the term myxoma for a set of tumors having a histologic resemblance to the mucinous substance of the umbilical cord. [10] In 1948, Stout redefined the histologic criteria for myxomas as true neoplasms that do not metastasize and exclude the presence of recognizable cellular components of other mesenchymal tissues such as chondroblasts, lipoblasts, and rhabdomyoblasts.

In the international histological classification of odontogenic tumors, odontogenic myxoma is defined as a benign odontogenic tumor of mesenchymal origin that is locally invasive and consists of rounded and angular cells that lie in the abundant mucoid stroma. [2]

Odontogenic myxoma commonly occurs in the age group of 10-40 years, and seldom seen below 10 years and above 50 years of age. [10],[11] The most frequent site of location is posterior mandible and premolar-first molar region in case of maxilla. [11],[12] They are often asymptomatic being diagnosed accidentally on a routine dental checkup. [12] However in advanced cases, they present with disturbing symptoms such as pain, paresthesia, mobility of teeth, ulceration, bony perforation, with subsequent invasion into the soft tissues. [11],[13]

Radiographically they present a varied appearance with smaller lesions tends to be unilocular, larger lesions are greater than 4 cm tend to be multilocular with a honeycomb, tennis racket or soap bubble pattern or with mixed radiopaque-radiolucent appearance. [14] CT images may present with the cortical expansion with or without cortical perforation, and a mixture of hypodense or hyperdense regions depending on the amount of calcified areas.

On magnetic resonance imaging, presents with the intermediate signal in the T1-weighted image and heterogeneous high signal on the T2-weighted image. [15] Histologically odontogenic myxoma shows proliferation of a few rounded, fusiform and stellate types of cells in an abundant myxoid stroma with scattered odontogenic epithelium and collagen fibers.

Histologically, other lesions with mucoid stroma such as neurogenic sarcoma, myxoid liposarcoma, rhabdomyosarcoma, neurofibroma, fibroma, lipoma, chondromyxoid fibroma may mimic odontogenic myxoma. [16] Clinically and radiographically odontogenic myxomas must be distinguished from cherubism, ameloblastoma, central giant cell granuloma, aneurysmal bone cyst intraosseous hemangioma, traumatic bony cyst, fibrous dysplasia, and odontogenic cysts such as radicular, lateral periodontal, dentigerous and keratocysti. [5],[17]

On immunohistochemistry, the tumor cells show positivity for smooth muscle actin and vimentin, and negative for neuron-specific enolase, desmin, glial fibrillary acid protein and S-100, suggestive of its myofibroblastic origin. [18]

Odontogenic myxoma being benign and locally aggressive with high recurrence rate, its treatment remains controversial, varying from simple enucleation and curettage to more aggressive en bloc resection. Due to its local infiltration, simple enucleation and curettage have been associated with a high recurrence rate of 10-33% and hence requires radical excision with burring of the cavity borders with maximum preservation of surrounding structures. [12] Due to its high recurrence rate, a minimum of 5 years follow-up period is required before moving to the final reconstruction. [7] Buccal pad of fat or corticocancellous iliac crest bone graft can be used for reconstruction of maxillary bony defects lesser than 5 cm. For larger maxillary defects, greater than 5 cm requires prothetic reconstruction using obturator. For Mandibular defects, reconstruction can be done using either free fibula flap, costochondral graft, iliac crest graft or scapular osteocutaneous free flap. [19]


   Conclusion Top


Odontogenic myxoma with its local infiltrative nature and high recurrence rate, justifies aggressive treatment with surgical en bloc resection and delayed reconstruction requiring a minimal follow-up period of 5 years to ensure disease free status before moving to final reconstruction phase. Its radiologically varied appearance and histologically the mucoid stroma mimicking few malignant lesions impose a diagnostic and operative dilemma; hence, warrants accurate clinical, radiological and histopathological interpretation to arrive at a definitive diagnosis, and effective management of patient.

 
   References Top

1.
Halfpenny W, Verey A, Bardsley V. Myxoma of the mandibular condyle. A case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90:348-53.  Back to cited text no. 1
    
2.
Kramer IRH, Pindborg JJ, Shear M. Histological Typing of Odontogenic Tumor. 2 nd ed. Berlin: Springer; 1992. p. 23.  Back to cited text no. 2
    
3.
Farman AG, Nortje CJ, Wood RE. Oral and Maxillofacial Imaging. St. Louis: Mosby; 1993. p. 257-60.  Back to cited text no. 3
    
4.
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. 4
    
5.
Brannon RB. Central odontogenic fibroma, myxoma (odontogenic myxoma, fibromyxoma), and central odontogenic granular cell tumor. Oral Maxillofac Surg Clin North Am 2004;16:359-74.  Back to cited text no. 5
    
6.
Stout AP. Myxoma, the tumor of primitive mesenchyme. Ann Surg 1948;127:706-19.  Back to cited text no. 6
[PUBMED]    
7.
Leiser Y, Abu-El-Naaj I, Peled M. Odontogenic myxoma - A case series and review of the surgical management. J Craniomaxillofac Surg 2009;37:206-9.  Back to cited text no. 7
    
8.
Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors: Analysis of 706 cases. J Oral Surg 1978;36:771-8.  Back to cited text no. 8
[PUBMED]    
9.
Thoma KH, Goldman HM. Central myxoma of the jaws. Am J Orthop 1947;33:532-40.  Back to cited text no. 9
    
10.
Slootweg PJ, Wittkampf AR. Myxoma of the jaws. An analysis of 15 cases. J Maxillofac Surg 1986;14:46-52.  Back to cited text no. 10
[PUBMED]    
11.
Simon EN, Merkx MA, Vuhahula 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. 11
    
12.
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. 12
    
13.
MacDonald-Jankowski DS, Yeung R, Lee KM, Li TK. Odontogenic myxomas in the Hong Kong Chinese: Clinico-radiological presentation and systematic review. Dentomaxillofac Radiol 2002;31:71-83.  Back to cited text no. 13
    
14.
Aquilino RN, Tuji FM, Nayene LM, Molina EO, Joo HY, Francisco HN. Odontogenic myxoma in the maxilla and characteristics on CT and MR. Oral Oncol Extra 2006;42:133-6.  Back to cited text no. 14
    
15.
Jaswal A, Jana AK, Biswas M, Sikder B, Nandi TK. Recurr ent odontogenic myxoma of maxilla: A diagnostic and operative dilemma. Indian J Otolaryngol Head Neck Surg 2008;60:41-4.  Back to cited text no. 15
    
16.
Kim J, Ellis GL. Dental follicular tissue: Misinterpretation as odontogenic tumors. J Oral Maxillofac Surg 1993;51:762-7.  Back to cited text no. 16
    
17.
Carvalho de Melo AU, de Farias Martorelli SB, Cavalcanti PH, Gueiros LA, Martorelli Fde O. Maxillary odontogenic myxoma involving the maxillary sinus: Case report. Braz J Otorhinolaryngol 2008;74:472-5.  Back to cited text no. 17
    
18.
Li TJ, Sun LS, Luo HY. Odontogenic myxoma: A clinicopathologic study of 25 cases. Arch Pathol Lab Med 2006;130:1799-806.  Back to cited text no. 18
    
19.
Tang YB, Hahn LJ. Major mandibular reconstruction with vascularized bone graft. J Formos Med Assoc 1990;89:34-40.  Back to cited text no. 19
    


    Figures

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



 

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
    Clinical Present...
   Discussion
   Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed1028    
    Printed19    
    Emailed0    
    PDF Downloaded46    
    Comments [Add]    

Recommend this journal