Rare presentation of odontogenic myxoma – A case report

AC Varsha; Ajay Parihar; Prashanthi Reddy; Ajay L Sutare

doi:10.4103/jiaomr.jiaomr_252_20

CASE REPORT

Year : 2021 | Volume : 33 | Issue : 3 | Page : 336-339

Rare presentation of odontogenic myxoma – A case report

AC Varsha, Ajay Parihar, Prashanthi Reddy, Ajay L Sutare
Department of Oral Medicine and Radiology, Government College of Dentistry, Indore, Madhya Pradesh, India

Date of Submission	01-Dec-2020
Date of Decision	05-May-2021
Date of Acceptance	29-Jul-2021
Date of Web Publication	28-Sep-2021

Correspondence Address:
Dr. A C Varsha
Department of Oral Medicine and Radiology, Government College of Dentistry, Sardar Patel Marg, Indore, Madhya Pradesh - 452 001
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jiaomr.jiaomr_252_20

Abstract

Odontogenic myxoma is a benign locally aggressive odontogenic tumor that arises from the mesenchymal portion of the tooth germ. They occur most frequently in the second or third decade of life and rarely occur before the age of 10 and after the age of 50. These are slow-growing tumors, but they may be locally aggressive, especially in the maxilla. Here we report the case of odontogenic myxoma in a 25-year-old male patient in the mandibular anterior region, which is usually seen in the posterior aspect of the mandible.

Keywords: Mandible, mesenchymal, myxoma, odontogenic tumor

How to cite this article:
Varsha A C, Parihar A, Reddy P, Sutare AL. Rare presentation of odontogenic myxoma – A case report. J Indian Acad Oral Med Radiol 2021;33:336-9

How to cite this URL:
Varsha A C, Parihar A, Reddy P, Sutare AL. Rare presentation of odontogenic myxoma – A case report. J Indian Acad Oral Med Radiol [serial online] 2021 [cited 2021 Nov 29];33:336-9. Available from: https://www.jiaomr.in/text.asp?2021/33/3/336/326898

Introduction

Odontogenic myxoma is a rare benign tumor that occurs in tooth-bearing areas of the maxilla and mandible.^[1] According to the World Health Organization (WHO) classification of odontogenic tumors, odontogenic myxoma is included under benign mesenchymal odontogenic tumors.^[2]

Odontogenic myxoma is uncommon, accounting for only 3%–6% of the odontogenic tumor and having slight female predilection.^[3] They are usually asymptomatic and found incidentally on the radiograph. The surgical approach to odontogenic myxoma ranges from a conservative approach to aggressive resections. A recurrence rate of 25% was reported and is explained by the lack of encapsulation of the tumor.^[1]

Patient Information

A 25-year-old male patient attended the Department of Oral Medicine Diagnosis and Radiology, complaining of swelling in the lower front teeth region for 1 month. Initially, the patient noticed a pea-sized swelling in the mandibular anterior region which was asymptomatic. Swelling gradually increased to the present size, causing retroclination of the lower anterior. Past medical and dental history was non-contributory and there was no history of trauma.

Timeline of history

Clinical Findings

On general examination, the patient was moderately built and nourished, vital signs were normal, and well oriented to time, place, and person. On extraoral examination, mild diffuse swelling of size 3 cm × 3 cm was seen in the mandible which extended from midline to the level of the right corner of the mouth [Figure 1]a. The superoinferior extension was from the vermilion border of the lower lip to the inferior border of the mandible. No change in color or texture of the overlying skin was seen. On palpation, the swelling was non-tender, hard in consistency with no local rise of temperature. Intraorally, a well-defined expansile swelling was seen involving the mandibular anterior region extending from 32 region crossing midline to 44 region [Figure 1]b. The superoinferior extension was from the alveolar ridge to the vestibule. The size of the lesion was approximately 3 cm × 4 cm. Obliteration of the labial, as well as the lingual vestibule, were seen with 33 to 43 regions along with mesiolingual inclination of 31,32,41,42,43 [Figure 1]c. On palpation, the swelling was non-tender and hard in consistency with the prominent expansion of buccal and lingual cortical plates.

Figure 1: (a) Profile showing mild diffuse swelling in the right side of mandible. (b) A well-defined expansile swelling in the mandibular anterior region extending from 32 region crossing midline to 44 region. (c) Obliteration of labial and lingual vestibule due to expansion of cortical plates

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Based on clinical presentation, a provisional diagnosis was made as Central giant cell granuloma.

Diagnostic assessment

For further investigations, an orthopantomogram (OPG) was made which showed a unilocular elliptical radiolucency with sclerotic borders extending from 35 to 47 and superoinferiorly from the alveolar ridge to the lower border of the mandible [Figure 2]a. Displacement of 31,32,33,43 distally and 41,42 medially was observed. For understanding the extent of the lesion, CBCT was performed and showed hypodensity with well-defined cortical borders. The axial view showed expansion and thinning of buccal and lingual cortical plates [Figure 2]b. The sagittal view showed irregularity present at the root tip of 46 to 34, suggestive of external root resorption [Figure 2]c.

Figure 2: (a) OPG showing unilocular elliptical radiolucency with sclerotic borders extending from 35 to 47 region. (b) The axial view of CBCT showing expansion and thinning of buccal and lingual cortical plates. (c) Sagittal view showing irregularity at the root tip of 46 suggestive of external root resorption

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Incisional biopsy findings suggested connective tissue stroma with odontogenic islands. For definitive diagnosis, excisional biopsy was done and sections revealed spindle-shaped to angular tumor cells distributed in myxomatous stroma, which confirmed the case of odontogenic myxoma [Figure 3]a.

Figure 3: (a) Excisional biopsy showing spindle-shaped to angular tumor cells distributed in myxomatous stroma. (b) OPG after 1-year follow-up showing considerable bone formation

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Therapeutic intervention

Considering the age, asymptomatic nature of the disease, and radiographic features, conservative management with enucleation and curettage was done with the extraction of 34–45.

Follow-up and outcomes

During the 1-year follow-up, we observed no sign of recurrence clinically and radiographically and areas of bone formation are evident radiographically [Figure 3]b.

Discussion

Odontogenic myxoma is an uncommon aggressive intraosseous lesion accounting for less than 3%–6%^[3] with an incidence of roughly 0.07 fresh cases per million people per year.^[4] Two variants of myxoma are identified in the head and neck,^[5] those derived from facial bones, which are further subdivided into true osteogenic myxoma and odontogenic myxoma and those derived from facial soft tissue.^[6] They occur most often within the second or third decade of life and show slight female predilection.^[3] Little is known about the molecular mechanism involved in disease initiation and progression.^[7]

The maxillary lesions usually extend to the maxillary sinus, and the mandibular ones surround the mandibular canal.^[8] Odontogenic myxoma rarely crosses the midline^[9] and a similar rare finding was noticed in this case presented.

Radiographically, odontogenic myxoma presents as a well-defined osteolytic lesion. The “soap-bubble, honeycomb or tennis-racket trabeculation” appearance can be observed^[8] and in the present case, a unilocular radiolucency with sclerotic borders and few intersecting septa were evident at the inferior margin. Jéssica Helena Dotta et al.^[4] concluded, among 1363 diagnosed cases between 1987 and 2018, multilocular aspect was seen in (57.49%), followed by unilocular aspect (32.87%), and mixed aspect (9.64%). The Posterior mandible (59.48%) was the common site of involvement followed by the anterior region (16.23%). In the maxilla, the most involved area was the posterior region (52.28%), followed by the anterior region (19.65%) with 2.98% case. In mandible involvement of anterior and posterior region occurred in 4.1% and within the maxilla in 2.98%.

Microscopically, odontogenic myxoma is composed of stellate to spindle-shaped cells in a mucoid-rich intercellular matrix, with no encapsulation and scattered residual bony trabeculae^[4] and these findings were evident in this case reported.

The clinical and radiological differential diagnosis of the present case is shown in [Table 1] and [Table 2], respectively. Generally, the diagnosis is based on the radiologic and histopathologic features.^[10]

Table 1: Clinical differential diagnosis of Odontogenic myxoma^[10]

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Table 2: Radiological differential diagnosis of Odontogenic myxoma^[10]

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Even though rare clinical and radiological presentations made this case a diagnostic challenge, excisional biopsy findings made a definitive diagnosis. Another biggest strength of this case is regular follow-up and there is no sign of recurrence clinically and radiographically.

Surgical treatment of odontogenic myxoma ranges from enucleation and curettage to segmental resection and hemimandibulectomy.^[2] Treatment decision between conservative and radical approaches frequently depend on the dimensions of the lesion. Disease recurrence usually occurs within 2 years following incomplete removal. However, follow-up is advised for 5 years, as cases of late disease recurrence have been reported.^[7]

Patient perspective

“I had good interaction with the physician during all the diagnostic and treatment procedures and I am satisfied with the treatment outcome as my symptoms improved.”

Conclusion

Odontogenic myxoma is a benign locally aggressive tumor mostly seen in the second or third decade of life with a slight female predilection. This case is reported because of its unusual site of occurrence and unilocular appearance. Since there are many overlapping lesions, the final diagnosis was made based on clinical, radiological, and histopathological findings.

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

1.	Koga YK, Saijo H, Hoshi K, Takato T, Mori Y. Surgical management of odontogenic myxoma: A case report and review of the literature. BMC Res Notes 2014;7:214.
2.	Speight PM, Takata T. New tumour entities in the 4^th edition of the World Health Organization Classification of Head and Neck tumours: Odontogenic and maxillofacial bone tumours. Virchows Arch 2018;472:331-9.
3.	White SC, Pharoah MJ. Oral Radiology Principles and Interpretation. 6^th ed. Mosby: St. Louis; 2009. p. 175-90, 221-2.
4.	Dotta JH, Miotto LN, Spin-Neto R, Ferrisse TM. Odontogenic myxoma: Systematic review and bias analysis. Eur J Clin Invest 2020;50:e13214.
5.	Landa LE, Hedrick MH, Nepomuceno-Perez MC. Recurrent myxoma of the zygoma: A case report. J Oral Maxillofac Surg 2002;60:704-8.
6.	Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors: Analysis of 706 cases. J Oral Surg 1978;36:771-8.
7.	Kauke M, Safi A-F, Kreppel M, Grandoch A, Nickenig H-J, Zöller JE, et al. Size distribution and clinicoradiological signs of aggressiveness in odontogenic myxoma— three-dimensional analysis and systematic review. Dentomaxillofac Radiol 2018;47:20170262.
8.	Oliveira SV, Rocha AC, Ceccheti MM, Gallo CB, Alves FA. Odontogenic myxoma in a child treated with enucleation and curettage. Autops Case Rep 2018;8:e2018042.
9.	Li T, Sun L, Luo H. Odontogenic myxoma: A clinicopathologic study of 25 cases. Arch Pathol Lab Med 2006;130:1799-806.
10.	Wood NK, Goaz P, Kallal RH. Multilocular radiolucencies. In: Differential Diagnosis of Oral and Maxillofacial Lesions. 5^th ed. St. Louis, Missouri: Mosby Elsevier; 2007. p. 342-3.