Journal of Indian Society of Periodontology
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   Table of Contents    
CASE REPORT
Year : 2012  |  Volume : 16  |  Issue : 3  |  Page : 461-464  

Odontogenic myxoma presenting as localized inflammatory gingival enlargement: A diagnostic dilemma


Department of Periodontics, Chatrapati Sahuji Maharaj Medical University (Erstwhile KGMC), Lucknow, Uttar Pradesh, India

Date of Submission02-Oct-2010
Date of Acceptance12-Mar-2012
Date of Web Publication12-Sep-2012

Correspondence Address:
Rameshwari Singhal
D 3023, Indira Nagar, Lucknow, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-124X.100932

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   Abstract 

Odontogenic myxoma (OM) is a rare and locally invasive benign neoplasm found exclusively in the maxillofacial region. The radiographic and clinical features are variable, and the diagnosis is therefore not easy. A case of OM of the maxilla is described in a 19-year-old female, previously diagnosed as inflammatory gingival enlargement. Clinical, histological, radiographic, and computed tomographic (CT) scan assessments were done, which were confirmatory for OM. Surgical excision of the lesion was done. The patient was put on periodic recall, as the recurrence rate of the lesion was high. The biological spectrum of OM was highly variable and diagnosis at an early stage was very difficult. Gingival enlargement presentation might not always be an inflammatory reaction. All possible differential diagnosis should be explored and various diagnostic tools utilized, to screen the enlargement.

Keywords: Diagnosis, gingival enlargement, odontogenic myxoma


How to cite this article:
Singhal R, Singh A, Rastogi P, Dixit J. Odontogenic myxoma presenting as localized inflammatory gingival enlargement: A diagnostic dilemma. J Indian Soc Periodontol 2012;16:461-4

How to cite this URL:
Singhal R, Singh A, Rastogi P, Dixit J. Odontogenic myxoma presenting as localized inflammatory gingival enlargement: A diagnostic dilemma. J Indian Soc Periodontol [serial online] 2012 [cited 2019 Aug 25];16:461-4. Available from: http://www.jisponline.com/text.asp?2012/16/3/461/100932


   Introduction Top


According to the World Health Organization (WHO), Odontogenic Myxoma is classified as an odontogenic tumor of the ectomesenchymal origin. It appears to originate from the dental papilla, follicle, or periodontal ligament. [1]

Odontogenic Myxoma of the 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. [2] OM is usually painless and displacement of teeth and paresthesia are uncommon clinical features. Therefore, it reaches considerable size before being detected, and perforation of the cortices of the involved bone may be seen. [3]

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 an odontogenic epithelium.

Radiographically, the tumor presents as a unilocular or multilocular radiolucent lesion with well-defined borders and 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. [4] Displacement of teeth is a relatively common finding, root resorption is rarely seen and the tumor is often scalloped between the roots. [5]

Application of a computed tomography (CT) Scan allows the production of panaromic, radial and axial 2D reconstruction. The extent of the lesion's relationship to teeth, root resorption, internal structure, cortical expansion and erosion, the boundary of the lesion and the presence of multiple lesions can all be evaluated.

Microscopically, the tumor consists of rounded, spindled, and stellate cells arranged in loose, myxoid stroma, with few collagen fibrils. Small islands of apparently inactive epithelial odontogenic rests may be scattered through the myxoid stroma. [6]

The recommended therapy varies from curettage to radical excision. Complete surgical excision can be difficult as the lesion is not encapsulated and the myxomatous tissue infiltrates the adjacent osseous tissue. This also accounts for the high recurrence rate of myxomas, which range from 10 - 30% with an average of 25%. [7]


   Case Report Top


A 19-year-old patient reported to the Outpatient Department of the Department of Periodontics, Faculty of Dental Sciences, Chatrapati Sahuji Maharaj Medical University (Erstwhile King George's Medical College), Lucknow, with a complaint of swelling in the left upper jaw region since one year.

Her history revealed exfoliation of a deciduous tooth in that area at the age of 12 years. Two months after exfoliation, the patient noticed a white mass in the canine premolar area, which resembled a nodule. One year back, the patient observed reddening of the nodule with a sudden increase in its size. The patient's records revealed that five months prior she had an incisional biopsy for the growth, which was diagnosed as chronic inflammatory enlargement.

Extraoral examination revealed no facial asymmetry. Intraoral examination revealed a diffuse erythematous swelling in between the left maxillary canine and first premolar [Figure 1] and [Figure 2]. The swelling measured 2.6 cm anteroposteriorly, 4.2 cm buccopalatally, and extended to the occlusal level superoinferiorly.
Figure 1: Preoperative swelling ― Buccal view

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Figure 2: Preoperative swelling ― Palatal view

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On palpation, the swelling was firm in consistency and tender. The overlying mucosa was immovably attached to the underlying bone.

The intraoral periapical, panoramic, and occlusal radiographs revealed a multilocular lesion with diffuse radio-opacity [Figure 3]. As the radiographs were non-diagnostic, Dentascan was done for further clarification. ACT scan revealed multilocular radiolucency along with slight cortical expansion and cortical perforation [Figure 4], [Figure 5] and [Figure 6].
Figure 3: Intraoral periapical view of the swelling

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Figure 4: Computed tomographic scan showing multilocular radiolucency in between 23 and 24

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Figure 5: Cortical expansion and perforation seen

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Figure 6: Multilocular radiolucency seen in three sections of the computed tomographic scan

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After thorough scaling and root planing of the adjoining teeth, the patient was scheduled for excisional biopsy. Complete excision of the swelling along with slight osseous resection was done extending well beyond the swelling margin [Figure 7].
Figure 7: Immediate post excision

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The histopathological examination revealed fragments of a myxomatous tissue [Figure 8] and [Figure 9]. The myxomatous tissue was even found infiltrating the osseous tissue, suggestive of a fibro-osseous lesion. Small islands of odontogenic epithelium were diagnostic of OM.
Figure 8: Highly cellular myxomatous tissue (H and E, 10×)

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Figure 9: Myxomatous tissue infiltrating osseous lamellae (H and E, 40×)

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Collaborating the clinical, radiological, and histological findings, the diagnosis was confirmed as odontogenic myxoma.

The patient has been put on a periodic recall program every month and recurrence has not been observed in the last six months [Figure 10], [Figure 11] and [Figure 12]. The patient is to be recalled every six months thereafter.
Figure 10: One week post operation

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Figure 11: Postoperative palatal view at six months

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Figure 12: Postoperative buccal view at six months

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   Discussion Top


Odontogenic myxoma is a rare tumor of the bone, and almost exclusively of the jaws, comprising of around 3 - 6% of all odontogenic tumors. [8] OM is found to occur most commonly in young patients with a predilection for females. The tumor is more common in the posterior parts of the mandibular jaw. The present case reports the tumor in the anterior part of maxilla. Although not very common, cases in the anterior maxilla are also seen.

Incisional biopsy of the patient was suggestive of a chronic inflammatory lesion. In all probability a superficial biopsy from the tumor margin may have resulted in a false conclusion.

Panoramic and occlusal radiographs were not very diagnostic. As is reported in the literature, the tumor presents a variable radiographic picture. The typical soap bubble or tennis racket appearance is not always seen. Zhang et al. [9] examined 41 cases of OM and found that on conventional radiographs, odontogenic myxomas presented varying radiographic appearances, which could be divided into six types as follows: Type I-unilocular; Type II-multilocular (including honeycomb, soap bubble, and tennis racquet patterns); Type III-involvement of local alveolar bone; Type IV-involvement of the maxillary sinus; Type V-osteolytic destruction, and Type VI-a mix of osteolytic destruction and osteogenesis. In the present case a type VI mixed picture of radiolucency and radio-opacity was visible.

Radiographically, a number of lesions resemble OM and should be differentially diagnosed from ameloblastoma, intraosseous hemangioma, aneurysmal bone cyst, central giant cell granuloma, metastatic tumor, and in some cases,of unilocular lesions, simple cysts.

For further clarification, a CT scan of the maxillary jaw was done. Mac Donald-Jankowski [10] compared the radiographic aspects of OM in conventional radiographs with Computed Tomography. The CT revealed bony cortices and its perforations with greater clarity.

Microscopically, spindle and stellate fibroblasts are associated with a basophilic ground substance and myxomatous tissue. It 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.

The lack of a capsule and infiltrative growth pattern is responsible for a high rate of recurrence when conservative enucleation and curettage are performed [11],[12]

Complete surgical excision of the lesion was performed. Periodic recall visits for the patient are planned, as the recurrence rate of the lesion is high. [7] Radical excision of the maxilla would be required if recurrence is noted.

To conclude, the odontogenic myxoma may present variable features on radiographs and superficial biopsies may be misleading. Due to the scarcity of studies using MR imaging, the characteristics of the myxoma have not been established satisfactorily. Therefore, additional studies are necessary to improve the diagnosis of this lesion, using the new imaging techniques that are now available to image this pathology.

An innocuous gingival enlargement presentation may sometimes be a disguise for a more serious pathology. Thorough exploration of the swelling and utilization of the available diagnostic tools may help screen such pathologies.

 
   References Top

1.Ward TO, Rooney GE. Asymptomatic expansion of the mandible. J Am Dent Assoc 1989;119:169-70.  Back to cited text no. 1
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2.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. 2
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3.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. 3
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4.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. 4
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5.Regizi JA, Sciubba JJ. Odontogenic tumours. In: Regizi JA, Sciubba JJ, Jordan RC, edtiors. Oral Pathology: Clinical - Pathologic correlations. 4th ed. Philadelphia: WB Saunders Company Ltd; 2003. p. 278-9.  Back to cited text no. 5
    
6.Katz JO, Underhill TE. Multilocular radiolucencies. Dent Clin North Am 1994;38:63-81.  Back to cited text no. 6
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7.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. 7
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8.Gnepp DR. Odontogenic cysts and tumours. In: Gnepp DR, editor. Diagnostic Surgical Pathology of head and neck. 1st ed. Philadelphia: WB Saunders Company Ltd; 2001. p. 643.  Back to cited text no. 8
    
9.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. 9
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10.Neville BW, Damm DD, Allen CM, Bouquot JE. Tumours of Odontogenic Ectomesenchyme. In: Neville BW, Damm DD, Allen CM, Bouquot JE, editors. Oral and Maxillofacial Pathology. 2 nd ed. Philadelphia: W.B. Saunders Company Ltd; 2002. p. 635.  Back to cited text no. 10
    
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
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12.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. 12
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12]



 

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