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CASE REPORT Table of Contents   
Year : 2008  |  Volume : 19  |  Issue : 1  |  Page : 62-65
Odontogenic myxoma of maxilla


Department of Oral and Maxillo-Facial Pathology, Meenakshi Ammal Dental College and Hospital, Chennai, Tamil Nadu, India

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Date of Submission05-Jul-2007
Date of Decision04-Nov-2007
Date of Acceptance14-Nov-2007
 

   Abstract 

Odontogenic myxoma (OM) is a rare and locally invasive benign neoplasm found exclusively in the jaws. OM commonly occurs in the second and third decade, and the mandible is involved more commonly than the maxilla. The lesion often grows without symptoms and presents as a painless swelling. The radiographic features are variable, and the diagnosis is therefore not easy. A case of OM of the maxilla with unusual radiographic and histologic features is described in a 30-year-old male. A panoramic radiograph revealed a well-demarcated, multilocular radiolucent lesion with 'tennis racket' appearance involving maxillary antrum. The histopathology showed loosely arranged spindle-shaped stellate cells and few areas of inactive odontogenic epithelium in a mucoid intercellular substance.

Keywords: Benign odontogenic tumor, fibromyxoma, odontogenic myxoma

How to cite this article:
Sivakumar G, Kavitha B, Saraswathi T R, Sivapathasundharam B. Odontogenic myxoma of maxilla. Indian J Dent Res 2008;19:62-5

How to cite this URL:
Sivakumar G, Kavitha B, Saraswathi T R, Sivapathasundharam B. Odontogenic myxoma of maxilla. Indian J Dent Res [serial online] 2008 [cited 2019 Oct 21];19:62-5. Available from: http://www.ijdr.in/text.asp?2008/19/1/62/38934

   Introduction Top


Odontogenic myxomas (OM) are tumors derived from embryonic mesenchymal elements of dental anlage. [1],[2] According to the World Health Organization (WHO), OM is classified as a benign tumor of ectomesenchymal origin with or without odontogenic epithelium. [3] It appears to originate from the dental papilla, follicle or periodontal ligament. [4] 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. [5]

OM is a locally invasive benign neoplasm. The invasiveness is attributed to the biological nature of the tumor. The OM exhibits abundant extracellular production of ground substance and thin fibrils by the delicate spindle-shaped cells. These undifferentiated mesenchymal cells are capable of fibroblastic differentiation also. [6],[7],[8] Depending upon the pattern of differentiation, the histological nature of the tumor varies. It may have complete myxomatous tissue or varying proportions of myxomatous and fibrous tissue. In the latter case it can be designated either as odontogenic fibromyxoma, in which the myxomatous element predominates; or odontogenic myxofibroma, with predominance of fibrous tissue. Some regard OM as a modified form of fibroma in which the myxoid intercellular substance separates the connective tissue. [9],[10]

OM of bone is a rare, benign tumor of unknown etiology. Most commonly, it occurs in the second and third decades. The mandible is involved more frequently than the maxilla, and most reports show a slight predilection for females. [1] OM are usually painless and displacement of teeth and paresthesia are uncommon clinical features. It therefore reaches considerable size before being detected, and perforation of the cortices of the involved bone may be seen. [4],[6],[11],[12],[13]

Radiographically, the tumor presents as a unilocular or multilocular radiolucent lesion with well-defined borders with fine, bony trabeculae within its interior structure expressing a 'honeycombed,' 'soap bubble,' or 'tennis racket' appearance. Unilocular appearance may be seen more commonly in children and in the anterior part of the jaws. [5],[7],[11] Displacement of teeth is a relatively common finding, root resorption is rarely seen and the tumor is often scalloped between the roots. [3],[4],[6],[12]

In this article a rare case of OM of the maxilla in a male patient is reported, and the varied histopathological features are discussed.


   Case Report Top


A 30-year-old male reported to the outpatient department of Meenakshi Ammal Dental College and Hospital, Chennai, on April 2005, complaining of a painless swelling in the posterior maxillary region for a period of 2 years. Initially, the swelling was small in size and showed a gradual increase to its present dimensions. The swelling extended from second premolar region to the maxillary tuberosity posteriorly, and it obliterated the buccal vestibule [Figure - 1]. A biopsy was performed, along with extraction of two teeth elsewhere a year back and the result of the biopsy was not known.

Extraoral examination showed a diffuse swelling in the right infraorbital region, obliterating the naso-labial fold. The skin over the swelling was normal, and there was no local rise of temperature. On palpation the swelling had variable consistency (hard anteriorly and soft posteriorly). Buccal and palatal cortices were expanded, and there was no history of paresthesia.

The radiograph showed a well-demarcated multilocular radiolucent lesion with 'tennis racket' appearance involving maxillary antrum. The CT image showed an expansile mass in the right maxilla, which completely obliterated the maxillary sinus [Figure - 2].

Histopathological examination of incisional biopsy specimen showed a dense and diffuse collection of fibers and fibroblasts and small inconspicuous strands of odontogenic epithelium. It was diagnosed as odontogenic fibroma, and the lesion was surgically excised.

On gross examination, the lesion appeared white in color with soft gelatinous texture [Figure - 3]. Histopathological examination of excised specimen showed tumor mass without encapsulation. It showed spindle- and stellate-shaped cells in loose, abundantly myxoid connective tissue stroma, closely resembling the mesenchymal portion of a developing tooth. Some areas showed moderately dense collagen fibers. Few strands of inactive odontogenic epithelium were seen within the connective tissue stroma. Overall histological appearance of the lesion revealed more amount of myxoid stroma in a less fibrous and acellular background [Figure - 4],[Figure - 5],[Figure - 6]. This histological feature of excisional specimen is different from that of the incisional biopsy, which was highly cellular and fibrous in nature, and it led to the diagnosis of odontogenic fibroma. The presence of myxomatous tissue, along with areas of moderately dense collagen fibers, led to the final diagnosis of OM.


   Discussion Top


OM is a rare aggressive intraosseous lesion derived from embryonic mesenchymal tissue associated with odontogenesis and primarily consisting of a myxomatous ground substance with widely scattered undifferentiated spindled mesenchymal cells. [14] Though it is a benign neoplasm, it may be infiltrative, aggressive and may recur. [5],[15],[16],[17],[18]

OM almost exclusively occurs in the jaw bones, comprising around 3-6% of all odontogenic tumors. The tumor occurs across an age group that varies from 22.7 to 36.9 years. It is rarely seen in patients younger than 10 years of age or older than 50. [12] Our case presented at the age of 30 years, which is in confirmity with that reported in literature. The mandible appears to be more frequently affected than the maxilla, especially the posterior region. In our case posterior region of the maxilla is involved. The majority of myxomas are almost always asymptomatic, although some patients present with progressive pain in lesions involving maxilla and maxillary sinus, with eventual neurological disturbance. OM of the jaw has a tendency for extensive bone destruction, invasion into surrounding structures and a relatively high recurrence rate; however, metastasis is rare. [19] OM of the maxilla is less frequent but behaves more aggressively than that of the mandible, as it spreads through the maxillary sinus as presented in our case. [10],[12],[15],[20],[21]

OMs radiographically appear as multilocular or unilocular radiolucencies. They are more frequently found in the anterior region of the jaws, while multilocular lesions occur mainly in the posterior region. [12],[15] OM should be included in the differential diagnosis of both radiolucent and mixed lesions, in both the jaws, for individuals of all age groups. When unilocular and without trabeculae, the tumor closely resembles periapical, lateral, periodontal and traumatic bone cysts. When multilocular, it must be distinguished from ameloblastoma, central hemangioma and odontogenic keratocyst. [15]

OM is a benign neoplasm without encapsulation. A spectrum of fibrous connective tissue stroma is present: from myxoid to densely hyalinized and from relatively acellular to cellular. [20],[22] Calcification may or may not be present. It is distinguished by the presence of sparse cords and islands of inactive odontogenic epithelium. [18],[23] The variation in the histopathological diagnosis between the initial biopsy as odontogenic fibroma and the final histopathological diagnosis as odontogenic fibromyxoma in our case could be attributed either to the biological spectrum of this lesion or non-inclusion of myxomatous areas in the biopsy. [11],[24] The ultra-structural findings indicate that the odontogenic fibroma and the odontogenic myxoma share many common morphological features. [4],[7]

An immunohistochemical panel of poly- and monoclonal antibodies was used to characterize and distinguish the nature of cells as of fibroblastic, histiocytic, myoblastic and neural origin. Three types of odontogenic myxoma cells were discriminated: spindle cells, stellate cells and hyaline cells. Neoplastic cells of myxomas were positively stained for transferrin, ferritin, alpha-1-antichymotrypsin (alpha 1-ACT), alpha-1-antitrypsin (alpha 1-AT), S-100 protein, vimentin (pan-mesenchymal marker) and actin; however, neuron-specific enolase (NSE), S-100 alpha subunit, S-100 beta subunit, Factor VIII-related antigen (FVIII-AG) and cytokeratin (CK1) were negative. [7],[18] Antibodies directed against vimentin are used to identify mesenchymal cells, and keratin antibodies detect epithelial differentiation. Spindle cells were positive for transferrin, ferritin, alpha 1-ACT, alpha 1-AT, S-100 protein and vimentin. Stellate cells were strongly positive for transferrin, alpha 1-AT, S-100 protein and vimentin. Hyaline cells reacted with alpha 1-ACT and alpha 1-AT. Myxomatous matrix showed negative reaction for all the antibodies used. These results have confirmed that odontogenic myxoma is a tumor of a dual fibroblastic-histiocytic origin and also suggest that the cells comprising odontogenic myxoma are of myofibroblastic origin. [7],[17],[18]

The aggressive nature of OM is well documented in the literature. The tumor is not radiosensitive, and surgery is the treatment of choice. [1],[11],[25] The lack of a capsule and infiltrative growth pattern is responsible for high rate of recurrence when conservative enucleation and curettage are performed. [26],[27] Recurrence is minimized with extensive partial or total resection procedures, and this method of treatment is particularly indicated in the maxilla due to the proximity of vital structures. [16],[22],[26],[28]

Summary

A case of odontogenic myxoma of the maxilla in a 30-year-old male patient is presented, initially diagnosed as odontogenic fibroma and later reported as odontogenic myxoma; it was treated by total surgical excision. The biologic spectrum of the lesion is reflected by the variation in the final excisional histopathological diagnosis

 
   References Top

1.Abiose BO, Ajagbe HA, Thomas O. Fibromyxomas of the jaws: A study of 10 cases. Br J Oral Maxillofac Surg 1987;25:415-21.  Back to cited text no. 1    
2.Lu Y, Xuan M, Takata T, Wang C, He Z, Zhou Z, et al . Odontogenic tumors: A demographic study of 759 cases in a Chinese population. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;86:707-14.  Back to cited text no. 2    
3.Pindborg JJ, Kramer IRH: Histological typing of Odontogenic tumors. Jaw cysts and Allied lesions. International Histological classification of tumors, No.5, World Health Organization: Geneva; 1971.  Back to cited text no. 3    
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12.Gnepp DR. Diagnostic Surgical Pathology of head and neck. WB Saunders Company Ltd: London; 2001. p. 643.  Back to cited text no. 12    
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15.Chuchurru JA, Luberti R, Cornicelli JC, Dominguez FV. Myxoma of the mandible with unusual radiographic appearance. J Oral Maxillofac Surg 1985;43:987-90.  Back to cited text no. 15    
16.Slootweg PJ, Wittkampf RM. Myxoma of the jaws: An analysis of 15 cases. J Maxillofac Surg 1986;14:46-52.  Back to cited text no. 16    
17.Green TL, Leighty SM, Walters R. Immunohistochemical evalution of oral myxoid lesions. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1992;73:407-71.  Back to cited text no. 17    
18.Zhao M, Lu Y, Takata T, Ogawa I, Miyauchi M, Mock D, et al. Immunohistochemical and histochemical characterization of the mucosubstances of odontogenic myxoma: Histogenesis and differential diagnosis. Pathol Res Pract 1999;195:391-7.  Back to cited text no. 18    
19.Hernαndez Vallejo G, Cohn C, Garcνa Peρνn A, Martνnez Lara S, Llanes Menιndez F, Montalvo Moreno JJ. Myxoma of the jaws: Report of three cases. Med Oral 2001;6:106-13.   Back to cited text no. 19    
20.Gosh BC, Huvos AG, Gerold FP, Miller TR. Myxoma of the jaw bones. Cancer 1973;31:237-40.  Back to cited text no. 20    
21.Odukoya O. Odontogenic tumors: An analysis of 289 cases. J Oral Pathol Med 1995;24:454-7.  Back to cited text no. 21    
22.Gormley MB, Mallin RE, Solomon M, Jarrett W, Bromberg B. Odontogenic myxofibroma: Report of two cases. J Oral Surg 1975;33:356-9.  Back to cited text no. 22    
23.OygŁr T, Dolanmaz D, Tokman B, Bayraktar S. Odontogenic myxoma containing osteocement-like spheroid bodies: Report of a case with an unusual histopathological feature. J Oral Pathol Med 2001;30:504-6.  Back to cited text no. 23    
24.Hwang EH, Lee SR. Central odontogenic fibroma of the simple type. Korean J Oral Maxillofac Radiol 2002;32:227-30.  Back to cited text no. 24    
25.Farman AG, Nortje CJ, Grotepass FW, Farman FJ, Van Zyl JA. Myxofibroma of the jaws. Br J Oral Surg 1977;15:3-18.  Back to cited text no. 25    
26.Simon NM, 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. 26    
27.Brannon RB. Oral and maxillofac surg, odontogenic tumors. Clin North Am 2004;16:359-74.  Back to cited text no. 27    
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Correspondence Address:
G Sivakumar
Department of Oral and Maxillo-Facial Pathology, Meenakshi Ammal Dental College and Hospital, Chennai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.38934

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    Figures

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

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