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Case Report

A large and rapidly expanding odontogenic


myxoma of the mandible
Sajad Ahmad Buch, Subhas G. Babu, Kumuda Rao, Shruthi Rao, Renita Lorina Castelino
Department of Oral Medicine and Radiology, A B Shetty Memorial Institute of Dental Sciences, Deralakatte, NITTE University, Mangalore,
Karnataka, India

A B S T R A C T

Odontogenic myxoma (OM) is a locally invasive benign tumour of the jaw originating from primordial mesenchymal tooth forming
tissues. The average age for patients with OM is 25–30 years. OM has a predilection for posterior mandible. When it occurs in the
maxilla, it is more invasive than that in the mandible. The recurrence rate of OM ranges between 10–33% with an average of 25%,
hence, proper treatment design and close postoperative follow‑up for the initial 2 years has been recommended. This is a case of a
30‑year‑old female showing a large expansile, rapidly growing lesion on the left side of the face involving left mandibular body and
ramus. OM cases of this size and such rapid growth are very few in literature, and hence, makes this case one of the rare cases.
Radiological investigations helped us to arrive at a diagnosis which was verified later by pre and postoperative histopathological
examination. The patient was treated with left hemi‑mandibulectomy and reconstruction was done using free fibula graft with titanium
implant. The patient is presently under follow‑up.

Key words: Cone beam computed tomography, mandible, odontogenic myxoma, septa

Introduction and third decades of life. Females have predilection over


males and more often it affects the mandible than maxilla.[6]
The mandibular sites most often affected are molar and
Odontogenic myxoma (OM) of the jaws was first described
ramus region, whereas in case of maxilla, the most affected
by Goldman and Thoma in 1947.[1] Myxomas of the
sites are premolar and first molar area.[7] OM is considered
soft tissue are frequent but intraosseous myxomas are
as a slow growing tumor with the capability of reaching to a
rare and almost always found in jaws.[2] The exclusive
considerable size with no significant signs and symptoms.[8]
occurrence of OM in the teeth bearing regions of the
jaws, occasionally being associated with missing and We managed the case of a 30‑year‑old female patient of
unerupted teeth and presence of odontogenic epithelium OM of the mandible with hemi‑mandibulectomy of the
indicate its odontogenic origin.[3] The World Health left side that proved to be justified owing to lack of any
Organization (WHO) in 2003 classified OM as a benign recurrences. Our treatment plan was based on the literature
neoplasm arising from odontogenic ectomesenchyme reporting high recurrence rates when OM of such sizes
with or without odontogenic epithelium.[4] OM is an have been treated by conservative approach.
unusual benign tumor comprising 3–6% of all tumors of
odontogenic origin.[5] OM usually occurs during the second This is an open access article distributed under the terms of the
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DOI: Cite this article as: Buch SA, Babu SG, Rao K, Rao S, Castelino RL. A large
10.4103/jomr.jomr_49_16 and rapidly expanding odontogenic myxoma of the mandible. J Oral Maxillofac
Radiol 2017;5:22-6.

Address for correspondence: Dr. Sajad Ahmad Buch, Department of Oral Medicine and Radiology, A B Shetty Memorial Institute of Dental
Sciences, Deralakatte, NITTE University, Mangalore ‑ 575 018, Karnataka, India. E‑mail: buchh.sajad@gmail.com

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Buch, et al.: Odontogenic myxoma of the mandible

Case Report the left mandibular region was given as provisional diagnosis
with a differential diagnosis of odontogenic myxoma. For
further investigations, orthopantomograph  (OPG) was
A 30‑year‑old female patient reported to the department of
made which showed features of diffused mutilocular
Oral Medicine and Radiology with a painless swelling of the
radiolucency with honey comb and tennis racket
left side of the face since the last 5 months. The swelling was
appearance involving the left body and the entire ramus
small initially and increased rapidly to its current size. There
of the mandible [Figure 2]. There were apparent signs
was no history of any pain or paresthesia and no difficulty
of root resorption with respect to 36,37 and 38. The
in breathing, eating, or swallowing. The patient’s medical
radiolucency which measured approximately 6 cm × 4 cm
and dental history was non-contributory. The patient was
extended anteroposteriorly from the mesial aspect of
moderately built and nourished. The patient’s vital signs
tooth number 35 to the posterior border of ramus, and
were normal at the time of reporting with a blood pressure
superioinferiorly it extended from the mandibular notch
of 120/80 mmHg, pulse rate of 76 beats/minute, and a
to the lower border of the mandible. The multilocular
respiration rate of 18/minute. On clinical examination, an
radiolucency also involved the alveolar process thereby
extraoral swelling was noticed corresponding to the left
causing displacement of 37 and 38 in the occlusal direction.
body and ramus of the mandible [Figure 1a]. The swelling
Angular multilocular compartments separated by straight
extended laterally from the left corner of the mouth up to
a point just in front of the left tragus of the ear. Superiorly, septa forming triangular, square, and rectangular spaces
the swelling extended from a point corresponding to a line were seen on OPG. Septa going beyond the periphery of
connecting the left corner of the mouth and left tragus of the lesion can be seen clearly on OPG (arrows), indicating
the ear, whereas it extended inferiorly up to the left inferior the invasiveness of the lesion. For understanding the extent
border of the mandible. The swelling was evident on left of the lesion, the patient was scanned using Cone Beam
side of the face [Figure 1b] when compared to the right Computed Tomography (CBCT) (Planmeca Promax 3D
side [Figure  1c]. The swelling was firm and nontender. MID (Planmeca Finland) at 90 kvp, 8 mA, with an exposure
A solitary left submandibular lymph node approximately time of 13.55 s and 20.2 cm height × 17.5 cm diameter scan
1.0 × 1.5 cm in size was palpable and slightly tender on volume. CBCT revealed a mixed radiolucent‑radiopaque
palpation. On intraoral examination, an ill‑defined swelling internal pattern of the tumor with buccolingual cortical
was noticed in the left buccal vestibule extending from expansion (arrows) on axial section [Figure 3a] with
tooth number 35 to left retromolar region [Figure 1d]. The buccal and lingual cortical plate perforations, straight
oral hygiene of the patient was poor. There was lingual sharp septa (arrow) on coronal CBCT section [Figure 3b],
displacement of 37 and 38. The teeth number 35 and 36 and sharp straight septa going beyond the periphery of
showed normal vitality response. There were root stumps lesion (arrow) on sagittal section of CBCT [Figure 3c].
with respect to 37, whereas 38 had grade  III mobility. Other findings were also in accordance with the OPG
Benign tumor of odontogenic origin (ameloblastoma) of findings. Hence, we arrived at a radiographic diagnosis of
odontogenic myxoma. Incisional biopsy was undertaken,
and sent for histopathological examination, which showed
randomly arranged spindle‑shaped, stellate, and round
cells in a loose myxoid stroma suggestive of myxoma.

a b

c d
Figure 1: (a) Extraoral photograph shows swelling in the left mandible. Figure 2: Panoramic radiograph shows a large ill‑defined multilocular
(b) Left lateral view of the face showing a diffused swelling. (c) Normal right radiolucency in the left mandibular body and ramus with honeycomb, tennis
lateral view of the face. (d) Intraoral view showing the left vestibule filled racket appearance with straight septae along the periphery of the lesion
with a large swelling (arrows)

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Buch, et al.: Odontogenic myxoma of the mandible

Keeping in mind the destructive nature of the lesion, Discussion


wide local excision with hemi‑mandibulectomy was carried
out [Figure 4a]. The reconstruction procedure was carried
Odontogenic myxoma (OM) is a benign tumor derived from
out by free fibula flap with pedicle and titanium plate
embryonic mesenchymal elements of a developing tooth
fixation  [Figure 4b]. The surgical site was closed using
including the dental follicle, dental papilla, or periodontal
3-0 ethilon suture material, by vertical mattress suture
ligament.[9] It begins with a slow growing tumor but can
pattern [Figure 4c]. The resected segment [Figure 4d] was
lead to massive cortical bone expansion and destruction
sent for histopathological investigation which confirmed
of bone at its later stages, implying its infiltrative behavior.
myxoma [Figure 5]. The surgical procedure was uneventful [10]
The relative incidence of OM in Asia, Europe, and
and was followed by alternate suture removal, removal of America has been found to be between 0.5% and 17.7%.
all sutures and removal of cast of left leg on 11th, 14th, [11]
Various studies done on OM were unable to come to a
and 21st postoperative days, respectively. The patient was definite common age group for its occurrence, however,
discharged on postoperative day 24, and was evaluated most of the studies have shown increased probability in the
after 3 months of the surgical procedure [Figure 6]. The age group of 22.7 to 36.9 years.[12] It is rare in children and
patient showed satisfactory healing with no complications.

a a b
b

c d
c
Figure 4: (a) Intraoperative image showing gelatinous, loose structure of
Figure 3: (a) CBCT images show (axial section) expansion of buccal and tumor mass. (b) Intraoperative image showing placement of reconstruction
lingual cortical plates (arrows). (b) CBCT images show (coronal section) plate. (c) Immediate postoperative image showing closure of surgical site
cortical expansion with sharp septa (arrow). (c) CBCT images show with sutures in place. (d) Image of resected specimen, left mandibular body,
(sagittal section) angular compartments with septae along the periphery of and ramus with condyle
the lesion (arrows)

a b

c
Figure 5: Histopathologic image: H and E stained section (10×) showing Figure 6: (a) Postoperative image (3 months follow up). (b) Postoperative
spindle‑shaped cells in loose myxoid stroma with delicate fibrils and dense image (3 months follow up) showing uneventful healing. (c) Postoperative
collagen fibers panoramic radiograph (3 months) showing titanium plate in place

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Buch, et al.: Odontogenic myxoma of the mandible

adults who are over 50 years of age.[13] The patient’s age in The studies have shown no cure with radiotherapy alone,
our case was 30 years and the affected site was mandible, and hence, OM is considered to be radioresistant.[22] As
which was in accordance with the available literature. OM OM is not encapsulated and its infiltration does not lead to
has a female predilection, with a male‑to‑female ratio of any destruction of the immediate hard tissues, the risk of
1:1.5.[14] It rarely presents with pain unless the lesion invades recurrence increases when conservative approach is applied
into the surrounding structures and results in neurological during its treatment.[19] These factors help to explain high
signs.[15] Halfpenny et al. stated that OM in most of the recurrence rate of OM that ranges between 10–33% with
cases presents as an asymptomatic swelling and can grow 25% considered to be the average recurrence rate.[23] Hence,
to considerable dimensions before diagnosis, as was seen our patient was managed with total excision of the lesion
in our case.[16] The patient reported here was asymptomatic and is under follow‑up at present.
despite its large size and locally aggressive behavior. The
lesion presented in our case was aggressive despite its Conclusion
mandibular location, which is contrary to the literature,
showing more aggressive behavior of such lesions in the
The future use of advanced imaging modalities such
maxilla compared to that in the mandible.[12] OM affects
as Computed Tomography and Cone Beam Computed
mandible more than the maxilla, with mandibular molar
Tomography may infer new information regarding
regions being the most common affected sites.[17] OM may
odontogenic myxoma lesions with its effect on the
become symptomatic at later stages because of expansion
surrounding structures. With proper treatment, the
of lesion and encroaching upon neighbouring structures,
prognosis of odontogenic myxoma is good and long
and symptoms can be of pain, particularly in maxilla due
term follow‑up of cases is necessary, particularly those
to its association with surrounding soft tissues.[16]
treated with a more conservative approach. Proper initial
Radiological appearance of OM is usually a unilocular or clinicoradiological diagnosis followed by corresponding
multilocular radiolucency having well‑defined or diffused histopathological report helps in arriving at an appropriate
margin. The size of the lesion and locularity are interrelated. diagnosis and aids in devising a treatment modality with
OM lesions which are larger than 4 cm follow a multiloculated minimal recurrences.
presentation, whereas smaller lesions seem to be unilocular
in appearance and the presentation is often defined as Declaration of patient consent
soap bubble, honeycomb, ground‑glass, or tennis racket The authors certify that they have obtained all appropriate
pattern.[18] This feature is in uniformity with our case which patient consent forms. In the form the patient(s) has/have
presented with multilocular radiolucency and tennis racket given his/her/their consent for his/her/their images and
appearance, typical of larger lesions. The interpretation other clinical information to be reported in the journal.
of radiographic features is vital in arriving at a diagnosis The patients understand that their names and initials will
in OM. The internal structure of OM usually consists of not be published and due efforts will be made to conceal
curved and coarse septa, however when straight and thin their identity, but anonymity cannot be guaranteed.
septa are encountered, it is considered as the characteristic
Financial support and sponsorship
feature of odontogenic myxoma.[19] The detection of
Nil.
straight sharp septa on either the conventional radiographs
or CBCT aids in arising at a diagnosis of OM. Our case
Conflicts of interest
presented with similar thin, sharp and straight septae
There are no conflicts of interest.
in both the panoramic and CBCT sections, considered
as characteristic of OM. Ameloblastoma, odontogenic
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