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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 14  |  Issue : 3  |  Page : 250-253

A rare case of odontogenic myxoma of the mandible


1 Department of Oral Pathology and Microbiology, Sharad Pawar Dental College and Hospital, DMIMS (Deemed to be University), Sawangi (M), Wardha, Maharashtra, India
2 Department of Oral Medicine and Radiology, Sharad Pawar Dental College and Hospital, DMIMS (Deemed to be University), Sawangi (M), Wardha, Maharashtra, India

Date of Submission10-Jun-2019
Date of Decision16-Jul-2019
Date of Acceptance30-Aug-2019
Date of Web Publication2-May-2020

Correspondence Address:
Dr. Preethi Sharma
Sharad Pawar Dental College and Hospital, Sawangi (M), Wardha, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdmimsu.jdmimsu_52_19

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  Abstract 


Odontogenic myxoma (OM) is a benign tumor originating from the embryonic mesenchymal component of the tooth germ. In 2003, World Health classification, OM is classified as benign tumor under odontogenic ectomesenchymal origin with or without included odontogenic epithelium. It is a rare benign locally aggressive odontogenic neoplasm attributed to the biological nature of lesion. The bones mostly affected are the jawbones. Clinically, it presents as a painless, slow-growing, expansile lesion of the jaws chiefly involving the mandible. Here, we report a rare case of OM in a 14-year-old female, where a large portion of the mandible was involved within a short span of 1 year. The lesion revealed typical clinical, radiographical, and histopathological characteristics of a myxoma.

Keywords: Aggressive, myxoma, odontogenic


How to cite this article:
Sharma P, Chaudhary M, Gawande M, Patel S. A rare case of odontogenic myxoma of the mandible. J Datta Meghe Inst Med Sci Univ 2019;14:250-3

How to cite this URL:
Sharma P, Chaudhary M, Gawande M, Patel S. A rare case of odontogenic myxoma of the mandible. J Datta Meghe Inst Med Sci Univ [serial online] 2019 [cited 2020 May 28];14:250-3. Available from: http://www.journaldmims.com/text.asp?2019/14/3/250/283593




  Introduction Top


Odontogenic myxomas (OMs) are noted as a painless, slowly increasing swelling or lack of symmetry of the affected jaw.[1],[2],[3] The growth of tumor may be rapid infiltrating the neighboring soft-tissue structures. Most OMs are located intraosseously, and few peripheral variants have also been described.[4],[5] Intraosseous myxomas other than the jaws were reported in the clavicle, in femur[6] and in ribs.[7] They are mostly detected in the 2–4th decades of life with a slight female predilection.[8] The site most commonly affected is mandibular molar region.[9] Radiographically, it presents as a honeycomb or mottled appearance of bone.[10] It may also appear as a destructive, expanding radiolucency with multilocular pattern.[11] Histologically, the tumor comprises of stellate-shaped cells in abundant myxoid stroma.[10]


  Case Report Top


A 14-year-old female patient was referred to the Department of Oral Pathology with a chief complaint of swelling in the lower left posterior region of jaw since 1 year. The patient was apparently alright 1 year back after which she noticed a swelling in the left mandibular posterior region of jaw, which was initially small in size and gradually increased in the span of 1 year. The patient complains of pain in the same region since 2 days which is dull aching and continuous in nature. There was no history of anesthesia or paresthesia in that region, no history of mobility or exfoliation of teeth from that region and no history of trauma. The past medical and dental history was not contributory. On extraoral examination, bilateral asymmetry was present due to swelling present on the mandibular posterior region extending anterioposteriorly from the left corner of mouth to the angle of mandible and superoinferiorly from the left ala-tragus line to the lower border of mandible [Figure 1]. The size of the lesion was 3 cm × 4 cm approximately, roughly oval in shape with smooth surface and irregular margins [Figure 2]. The color was that of the adjacent mucosa. On palpation, consistency was firm to hard and tenderness was present. No crackling and crepitus was present. Submandibular lymph nodes were palpable bilaterally, each of size approximately 0.5 cm × 0.5 cm, roughly round, firm, mobile, and nontender. The provisional diagnosis was made as odontogenic cyst or odontogenic tumor. Clinically, dentigerous Cyst develops around the crown of an unerupted tooth and is associated with a missing tooth with an extraoral hard swelling seen in young patients in 2nd and 3rd decade of life. In this case, the patient was 14-year-old with a possibility of developing follicle with 38 and considering the extraoral swelling and expansion we considered an underlying dentigerous cyst.
Figure 1: Extraoral examination

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Figure 2: Intraoral examination

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On inspection, expansion was present on the buccal self-area in relation to 36, 37, 38 regions. Obliteration and tenderness were present in the buccal shelf area with mild expansion on the lingual side. Investigations advised are orthopantomography (OPG), posteroanterior (PA) mandible, computed tomography (CT), and biopsy of the lesion. OPG showed well-defined multilocular radiolucency from the medial of 36 to coronoid notch. The periphery of the lesion is corticated and thinning of the inferior and posterior cortex in horizontal and vertical ramus is shown in [Figure 3]a. Scalloping of the superior cortex and lateral cortex is shown in [Figure 3]b. To appreciate the buccal and lingual expansion PA mandible was advised [Figure 4]a. Thinning and expansion of the buccal and lingual cortical plate were appreciated on the left side [Figure 4]b. A single well-defined expansile lytic lesion arising from the left ramus of mandible extending the body of the mandible is seen. Here, the internal matrix is not seen because it is a homogeneous low-density noncalcified matrix which cannot be appreciated in CT [Figure 5]. Clinicoradiographic diagnosis was made as OM. After the radiographic examination, biopsy was performed and the section was viewed under compound microscope. Histopathologically, under scanner view hematoxylin and eosin (H and E) stained lesional tissue section shows cellular, basophilic loose myxoid connective tissue stroma [Figure 6]. Under low power, H and E stained lesional tissue section shows numerous loosely arranged stellate-shaped cells and few spindle-shaped cell dispersed throughout the connective tissue stroma. The connective tissue stroma is intensely basophilic in nature, suggestive of myxoid stroma. Few endothelium lined blood capillaries, haphazardly arranged collagen fibers and chronic inflammatory cell infiltrate chiefly comprising of lymphocytes is seen in the connective tissue stroma. Another field shows areas of calcification [Figure 7]. Under high power, all the findings of low power were confirmed; the stellate-shaped cells are anastomosing with cytoplasmic process dispersed throughout the loose myxoid connective tissue stroma [Figure 8]. The histopathological diagnosis was made as OM. Considering the rarity of the occurrence of lesion, special stain alcian blue was performed and it was positive as the presence of mucoid material, i.e., hyaluronic acid is demonstrated by alcian blue staining [Figure 9]. On the basis of clinical, radiological, and histopathological features, the final diagnosis was made as OM. Surgical enucleation and curettage of the lesion were performed and the patient was kept under follow-up with no evidence of recurrence.
Figure 3: (a) Orthopantomogram showing a well-defined multilocular radiolucency appreciated from the medial of 36 to coronoid notch. (b) Orthopantomogram showing scalloping of the superior cortex and lateral cortex is seen

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Figure 4: (a) Posteroanterior mandible shows thinning and expansion of the buccal and lingual cortical plate on the left side. (b) Thinning and expansion of the buccal and lingual cortical plate is seen on the left side

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Figure 5: Axial computed tomography shows a single well-defined expansile lytic lesion arising from the left ramus of mandible extending the body of the mandible

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Figure 6: H and E stained tissue section under scanner view

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Figure 7: H and E stained tissue section under low power view

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Figure 8: H and E stained tissue section under high power view

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Figure 9: Alcian blue staining

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  Discussion Top


In 1863, the German Pathologist Rudolf Virchow was the first to describe the histopathological features of myxofibroma. In 1947, myxomas of the jaws was first described by Thoma and Goldman.[12] In 1992, WHO classification, the term “Myxoma” was used along with the OM and myxofibroma as alternative terms.[13] The pathogenesis of OMs includes the dental papilla, dental follicle, and periodontal tissue as the possible germ centers.[1] They are locally aggressive, nonencapsulated, nonmetastasizing neoplasm that infiltrate bone marrow spaces.[14] In 1992, the WHO defined OM as a locally invasive neoplasm consisting of rounded and angular cells lying in abundant mucoid stroma.[13] The histochemical findings in OMs were reviewed by Farman et al.[15] The ground substance consists of 80% hyaluronic acid and 20% chondroitin sulfate. The alkaline phosphatase activity was shown by the tumor cells. The myxoid matrix also shows positivity with alcian blue staining but periodic acid staining may be negative. Takahashi et al. studied OMs immunohistochemically using a number of antibodies such as S-100, vimentin, transferrin, ferritin, alpha-1 antichymotrypsin, alpha-1 antitrypsin, neuron-specific enolase, CK-1, and others. Staining patterns of spindle, stellate, and hyaline cells suggested a dual fibroblastic-histiocytic origin.[16] Type I and type IV collagen, tenascin, fibronectin, and proteoglycans are the different proteins exhibited by the matrix. OMs have been characterized as secretory.[17] The treatment plan varies from local excision, curettage, and enucleation to radical resection. OMs are considered as a slow-growing neoplasm; it may be infiltrative and aggressive with high recurrence rates. Recurrence rates have been reported with an average of 25%.[18] Clinical and radiographic follow-up should be maintained periodically regardless of the treatment method.


  Conclusion Top


OM is an uncommon benign odontogenic tumor with inconclusive clinical and radiographic features. Hence, histopathological features should be taken into consideration for accurate diagnosis and treatment plan.

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.
Sivakumar G, Kavitha B, Saraswathi T, Sivapathasundharam B. Odontogenic myxoma of maxilla. Indian J Dent Res 2008;19:62-5.  Back to cited text no. 1
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Neville BW, Damm DD, Allen CM, Bouguot JE. Oral and Maxillofacial Pathology. 2nd ed. Pennysylvania: WB Saunders Company Ltd.; 2004. p. 635-7.  Back to cited text no. 10
    
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Langlais RP, Langland OE, Lortje PJ. Diagnostic Imaging of the Jaws. USA: Williams and Wilkins Waverly Company Ltd.; 1995. p. 722-4.  Back to cited text no. 11
    
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Thoma KH, Goldman HM. Central myxoma of the jaw. Oral Surg Oral Med Oral Pathol 1947;33:B532-40.  Back to cited text no. 12
    
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Kramer IR, Pindborg JJ, Shear M. Histologic Typing of Odontogenic Tumors. 2nd ed. Berlin: Springer-Verlag; 1992.  Back to cited text no. 13
    
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Barros RE, Dominguez FV, Cabrini RL. Myxoma of the jaws. Oral Surg Oral Med Oral Pathol 1969;27:225-36.  Back to cited text no. 14
    
15.
Farman AG, Nortjé 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. 15
    
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Takahashi H, Fujita S, Okabe H. Immunohistochemical investigation in odontogenic myxoma. J Oral Pathol Med 1991;20:114-9  Back to cited text no. 16
    
17.
Jaeger M, Santos J, Domingues M, Ruano R, Araújo N, Caroli A, et al. A novel cell line that retains the morphological characteristics of the cells and matrix of odontogenic myxoma. J Oral Pathol Med 2000;29:129-38.  Back to cited text no. 17
    
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Barker BF. Odontogenic myxoma. Semin Diagn Pathol 1999;16:297-301.  Back to cited text no. 18
    


    Figures

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



 

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