CASE REPORT - ODONTOGENIC ORIGIN
|Year : 2019 | Volume
| Issue : 4 | Page : 83-86
Odontogenic myxoma in an 8-year-old girl: A case report with review of literature
Divyesh Wankhedkar, Sangeeta Patankar, S Gokul, Shubhra Sharma
Department of Oral Pathology, YMT Dental College, Mumbai, Maharashtra, India
|Date of Submission||30-Jan-2018|
|Date of Acceptance||28-Dec-2018|
|Date of Web Publication||22-Feb-2019|
Shriniketan 19th A Road, Anand Vihar Society, Khar West, Mumbai - 400 052, Maharashtra
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Aggressive enlargements of maxilla in pediatric patients are uncommon and present with diagnostic and therapeutic dilemma. The decision on therapeutic modality is based on an early and accurate diagnosis, minimizing disease-associated morbidity which is of utmost importance considering the young age and thereby resulting in better prognosis. Odontogenic myxoma is a locally aggressive lesion which is primarily seen in relation to odontogenic apparatus in mandibular posterior region in association with an impacted tooth. This presentation describes a unique case of odontogenic myxoma of anterior maxilla in an 8-year-old girl with emphasis on its diagnosis and treatment planning.
Keywords: Maxilla, odontogenic myxoma, pediatric neoplasm
|How to cite this article:|
Wankhedkar D, Patankar S, Gokul S, Sharma S. Odontogenic myxoma in an 8-year-old girl: A case report with review of literature. J Oral Maxillofac Pathol 2019;23, Suppl S1:83-6
|How to cite this URL:|
Wankhedkar D, Patankar S, Gokul S, Sharma S. Odontogenic myxoma in an 8-year-old girl: A case report with review of literature. J Oral Maxillofac Pathol [serial online] 2019 [cited 2020 Jan 23];23, Suppl S1:83-6. Available from: http://www.jomfp.in/text.asp?2019/23/4/83/252719
| Introduction|| |
The term myxoma was coined by Virchow in 1863 to describe a group of tumors that had histologic resemblance to the mucinous substance of the umbilical cord. Myxomas are benign, slow-growing and locally aggressive mesenchymal neoplasms. They can be found in various body parts such as skin, subcutaneous tissue and the heart (left atrium), but rarely in the head and neck region. Myxoma of the head and neck region can occur either in the subcutaneous tissue or the facial bones with increased predilection to occur in relation to odontogenic apparatus.
Thoma and Goldman first described odontogenic myxoma of the jaw in 1947. It is a nonencapsulated benign tumor of the jaws which can be divided into two groups: (1) tumors that arise specifically in jaw bones (most common type) and (2) those that arise in soft tissues of that area. The origin of odontogenic myxoma is believed to be from the ectomesenchyme of a developing tooth or from undifferentiated mesenchymal cells in the periodontal ligament.
Odontogenic myxoma mostly occurs in the second or third decade of life and is common in mandibular posterior region. Smaller lesions are asymptomatic while large lesions cause painless expansion of bone. Radiographically, myxoma shows unilocular or multilocular radiolucency with irregular or scalloped margins. Microscopic picture shows stellate-shaped cells dispersed in a loose myxoid stoma. We report a unique case of odontogenic myxoma in an 8-year-old girl affecting the anterior maxillary area which is a rare occurrence, both in terms of age and site.
| Case Report|| |
An 8-year-old girl reported with painless, gradually enlarging swelling in anterior maxilla for 3 months. On examination, a well-circumscribed growth was seen in the anterior maxillary region measuring 4.1 cm × 4 cm in size extending from labial vestibule to the palatal bone in the region deciduous first molar teeth. Overlying mucosa was inflamed with area of ulceration due to incisional biopsy [Figure 1].
On palpation, growth was firm in consistency, nontender without any bleeding on probing. Lymph nodes were not palpable. Radiographic picture showed radiolucency with defined borders in the anterior maxillary area, buccal and lingual cortical plates were destroyed and developing permanent central and lateral incisors were seen to be displaced. Based on the radiographic evaluation, provisional diagnosis of Juvenile ossifying fibroma and osteosarcoma was considered [Figure 2] and [Figure 3].
Incisional biopsy performed at another institute gave a histopathological diagnosis of aggressive fibrous lesion.
Considering the extent of lesion and incisional biopsy report, complete surgical excision with partial maxillectomy was performed and the excised tissue was sent for histopathological analysis.
Macroscopically, the specimen received in our laboratory measured 3 cm × 4 cm × 2 cm and composed of both soft and hard tissue. The macroscopic appearance of the lesion showed slimy surface and firm in consistency. Deciduous teeth and impacted permanent teeth were present within the lesion.
Microscopic examination showed a well-circumscribed lesion consisting of myxoid areas composed of sparsely distributed stellate-shaped fibroblasts and odontogenic islands in a loose stroma [Figure 4]. The presence of inactive odontogenic islands in the deeper connective stroma was evident [Figure 5]. The peripheral capsule was densely fibrous with reactive bone formation.
Based on the above findings, the lesion was diagnosed as odontogenic myxoma. On 6-month follow-up, uneventful healing was observed.
| Discussion|| |
Odontogenic myxoma is a rare aggressive intraosseous lesion. Myxomas are most commonly associated with unerupted teeth and derived from mesenchymal tissue of the tooth germ consisting of a myxomatous ground substance with scattered undifferentiated spindled mesenchymal cells. Although it is a benign neoplasm, it may be infiltrative, aggressive and may recur.
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. In our case, it was seen in an 8-year-old girl which is an unusual finding. The mandible appears to be more frequently affected than the maxilla, especially the posterior region. However, cases have been reported of odontogenic myxoma appearing in posterior maxilla. Anterior maxillary involvement in our case is a rare occurrence.
The aggressive nature of odontogenic myxoma is well documented in the literature. Odontogenic myxoma of the maxilla is less frequent, but behaves more aggressively than that of the mandible, as it has tendency to spread to the maxillary sinus.
Odontogenic myxoma grows without symptoms as a painless swelling and gets noticed only after reaching a considerable size. Radiographic picture can show either unilocular or multilocular appearance with defined or diffuse borders. A unilocular radiolucency is generally seen in children and in lesions present in the anterior region of the jaw. It is a benign neoplasm without encapsulation. A spectrum of fibrous connective tissue stroma is present ranging from myxoid to densely hyalinized and from relatively acellular to cellular appearance. Calcification may or may not be present. It is distinguished by the presence of sparse cords and islands of inactive odontogenic epithelium. Sivakumar and Kavitha suggested that odontogenic myxoma is a tumor of a dual fibroblastic-histiocytic origin and the cells comprising odontogenic myxoma are of myofibroblastic origin.
It is not radiosensitive, and surgery is the treatment of choice. The infiltrative growth pattern is responsible for high rate of recurrence when conservative enucleation and curettage are performed. However, Bravo-Burguillos et al. stated that conservative surgical excision done in infants did not show any clinical or radiographic evidence of recurrence for 3-year posttreatment follow-up. Recurrence is minimized with extensive partial or total resection of the lesion. While doing resection particularly in the maxilla, proximity of vital structures should be considered.
A summary of odontogenic myxoma documented in the literature during the period from 2010 onward is presented in [Table 1].
|Table 1: Summary of odontogenic myxoma affecting the maxilla documented in the literature|
Click here to view
| Conclusion|| |
A unique case of odontogenic myxoma is reported in an 8-year-old girl involving the anterior maxillary region, and according to the literature, occurrence of this lesion in patients <10 years of age is rare. Odontogenic myxoma shares common clinical and radiographic features with various other odontogenic and fibrous tumors. To establish accurate diagnosis and treatment plan, histopathological diagnosis is of utmost importance.
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
Conflicts of interest
There are no conflicts of interest.
| References|| |
Munjal N, Bharadwaj V, Garg B, Sood N. Odontogenic myxoma of the maxilla: A clinical case report and review of literature. Otolaryngol Online J 2013;3:1-10.
Shah A, Lone P, Latoo S, Ahmed I, Malik A, Hassan S, et al.
Odontogenic myxoma of the maxilla: A report of a rare case and review on histogenetic and diagnostic concepts. Natl J Maxillofac Surg 2011;2:189-95.
] [Full text]
Thoma KH, Goldman HM. Central myxoma of the jaws. Am J Orthop 1947;33:532-40.
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.
Rajendran R, Sivapathasundharam B. Shafer's Textbook of Oral Pathology. 7th
ed. India: Elsevier; 2012;299-300.
Sarode TP, Malik NA. Odontogenic myxoma in a child: Diagnostic and treatment dilemmas. J Indian Sot Pedod Prev Dent 2002;20:68-72.
Kim J, Ellis GL. Dental follicular tissue: Misinterpretation as odontogenic tumors. J Oral Maxillofac Surg 1993;51:762-7.
Sivakumar G, Kavitha B, Saraswathi TR, Sivapathasundharam B. Odontogenic myxoma of maxilla. Indian J Dent Res 2008;19:62-5.
] [Full text]
Gnepp DR. Diagnostic Surgical Pathology of Head and Neck. London: W.B Saunders Company Ltd.; 2001. p. 643.
Reichart PA, Philipsen HP. Odontogenic Tumors and Allied Lesions. London: Quintessence Publishing Co Ltd.; 2004.
Reddy SP, Naag A, Kashyap B. Odontogenic myxoma: Report of two cases. Natl J Maxillofac Surg 2010;1:183-6.
] [Full text]
Gosh BC, Huvos AG, Gerold FP, Miller TD. Myxoma of the jaw bones. Cancer 1973;31:237-40.
Oygür 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.
Kiresur MA, Hemavathy S. An aggressive odontogenic myxoma of the maxilla. Indian J Dent 2014;5:214-7. [Full text]
Deron PB, Nikolovski N, den Hollander JC, Spoelstra HA, Knegt PP. Myxoma of the maxilla: A case with extremely aggressive biologic behavior. Head Neck 1996;18:459-64.
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.
Bravo-Burguillos ER, Pozo Kreilinger JJ, Muñoz Caro JM, Parra AR. Infant odontogenic myxoma: Case report and literature review of a specific entity recently described. Arch Otolaryngol Rhinol 2016;52:6-8.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]