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International Journal of Surgery Case Reports 112 (2023) 108945

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International Journal of Surgery Case Reports


journal homepage: www.elsevier.com/locate/ijscr

Case series

Surgical management of odontogenic myxomas: A case series


Hannah Trode a, *, Celso Pouget b, Marouane Talbi c, Etienne Simon d, e, Muriel Brix e, f
a
Faculty of Medicine, Lorraine University, 9 avenue de la Forêt de Haye, 54505 Vandoeuvre-lès-Nancy, France
b
Department of Anatomical Pathology, Nancy University Hospital Center, Hôpitaux de Brabois, Rue du Morvan, Vandœuvre-lès-Nancy, France
c
Department Maxillo-facial and Plastic Surgery, Regional Metz Hospital, 1 allée du Château, 57 085 Ars-Laquenexy, France
d
Department Maxillo-facial and Plastic Surgery, Nancy University Hospital Center, Hôpital Central, 29 avenue de Lattre de Tassigny, Nancy, France
e
Lorraine University, Faculty of Medicine, Vandoeuvre-lès-Nancy, France
f
Department Maxillo-facial and Plastic Surgery, Nancy University Hospital Center, Hôpital Central, 29 avenue de Lattre de Tassigny, Nancy, France

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction: Odontogenic myxoma is a relatively rare bone tumor involving exclusively the jaws. Despite its
Cases series of odontogenic myxomas benign nature, odontogenic myxoma can exhibit aggressive, locally invasiveness and has a high potential of
Surgical management of odontogenic myxoma recurrence. Surgical treatment can be conservative of radical. The choice of surgical procedure is controversial,
Odontogenic tumors
and there are no established guidelines.
Management algorithm of odontogenic
myxoma
Case series presentation: We present a case series of three cases of odontogenic myxomas. This case series illus­
trates the variability of the radioclinical presentation of odontogenic myxoma and the resulting surgical man­
agement, ranging from simple enucleation to mandibular interruptive resection surgery with free flap
reconstruction.
Discussion and conclusion: Through this case series, we highlighted and described decision criteria contributing to
treatment choice and summarized this in an algorithm. Radiological tumor characteristics and also patient
specific factors such as age need to be considered to make a personalized decision to each patient.

1. Introduction objective is to highlight the decision criteria that influence the choice of
surgical procedures.
According to the latest 2017 WHO classification, odontogenic myx­
oma is categorized as a benign odontogenic mesenchymal tumor. 2. Methods
Odontogenic tumors comprise a heterogeneous group of benign or ma­
lignant lesions arising from epithelial and/or mesenchymal components This case series include three cases of odontogenic myxoma reported
involved in odontogenesis [1]. These tumors are exclusively found at the Maxillofacial Surgery Department at Nancy University Hospital
within the maxillo-mandibular region. Odontogenic myxoma ranks as and at the Metz Regional Hospital.
the third most prevalent odontogenic tumor after odontoma and ame­ This case series has been reported in line with PROCESS criteria [3].
loblastoma, but it remains relatively rare [2].
Despite its benign nature, odontogenic myxoma can display aggres­ 3. Results
sive behavior, local invasiveness, and a substantial potential for
recurrence. 3.1. Case 1 presentation
The treatment of odontogenic myxoma necessitates surgical inter­
vention, which can range from conservative to more radical approaches. A 26-year-old female patient was referred to the Maxillofacial Sur­
Due to its low prevalence, no established guidelines currently exist for its gery Department at Nancy University Hospital by her dentist due to
management. radiological findings revealing a recurrence of an asymptomatic
We present then an analysis of the clinical, radiological, and pri­ mandibular osteolytic lesion. She had no other specific medical history.
marily therapeutic aspects of three cases of odontogenic myxomas re­ Five years prior, the 21-year-old patient had undergone the tooth 36
ported at Nancy University Hospital and Metz Regional Hospital. Our extraction along with with curettage of the mandibular lesion by her

* Corresponding author.
E-mail address: hannah.trode@hotmail.fr (H. Trode).

https://doi.org/10.1016/j.ijscr.2023.108945
Received 23 September 2023; Received in revised form 6 October 2023; Accepted 7 October 2023
Available online 17 October 2023
2210-2612/© 2023 Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
H. Trode et al. International Journal of Surgery Case Reports 112 (2023) 108945

dentist.
A cone beam computed tomography (CBCT) showed a poorly
delineated 17-mm long multilocular hypodensity surrounding the roots
of tooth 36, with a resorbed distal root. There was no cortical effraction.
Some intra-lesional septa were observed (Fig. 1A). Pathological analysis
identified an odontogenic myxoma.
Only one radiography was performed one month postoperatively.
Thereafter, no further follow-up was not conducted. CBCT was per­
formed only 5 years later, primarily to assess the alveolar bone level for
the purpose of rehabilitating tooth 36 (Fig. 1B).
Patient was asymptomatic and clinical examination was normal.
Facial CT scan showed a voluminous multilocular osteolytic lesion
extending approximately 55 mm between teeth 34 and 38, with multiple
septas. Both vestibular and lingual cortical bone were thinned, with a 20
mm rupture observed in the lingual cortical bone adjacent to tooth 38
(Figs. 1B and 2).
Conservative treatment was performed under general anesthesia by
an experienced maxilla-facial surgeon, consisting of enucleation of the
lesion with meticulous curettage of the resection cavity and extractions
of teeth 37 and 38. Post-operative prescriptions included analgesics,
Amoxicillin-clavulanic acid antibiotic therapy, and antiseptic
mouthwash.
Pathological analysis revealed proliferation of low cell density,
composed of cells arranged in an abundant, loose, fibrous myxoid
background. No identifiable epithelial structures were present. Immu­
nohistochemical staining yielded negative results for AE1/AE3, MUC4,
P-S100, CD34, and EMA markers. Ki67 proliferation index was low
(Fig. 3).
The patient was currently undergoing follow-up. Reossification of
Fig. 2. Facial CT scan revealing 5-year recurrence of a larger odontogenic
the surgical site defect was in progress after 15 months. myxoma, with lingual cortical effraction.

3.2. Case 2 presentation and the basilar margin, resulting in a pseudo-nodule with a diameter of
approximately 50 mm (Fig. 4).
A 34-year-old patient with no previous medical history presented to The lesion showed slight enhancement after contrast injection.
the Maxillofacial Surgery Department of the Regional Hospital of Metz A biopsy was performed, which revealed the diagnosis of odonto­
for a painless right submandibular swelling evolving for one year. genic myxoma.
On clinical examination, a firm right submandibular swelling was Subsequently, the patient underwent a segmental mandibular
observed, along with intraoral swelling on the lingual surface of the resection from tooth 32 to the mandibular condyle, obtaining margins of
mandible in the retromolar region. Moreover, the oral mucosa appeared 1 to 1.5 cm by an experienced maxilla-facial surgeon. Mandibular
healthy. There was no hypoesthesia in the inferior alveolar nerve reconstruction was performed simultaneously using a micro-
territory. anastomosed free fibula flap, osteosynthesized with custom-made tita­
Contrast-enhanced cervicofacial CT-scan revealed a large multi­ nium plates. Some cervical lymph nodes were fortuitously indentified
locular mandibular osteolytic lesion measuring up to 65 mm in its long during surgery and were removed for analyse. The patient remained
axis. This lesion extended from the right parasymphysis to 1 cm below hospitalized for 15 days following the procedure. Post-operative pre­
the right condylar head. The lesion featured angular bony septa within scriptions included analgesics, Amoxicillin-clavulanic acid antibiotic
it, and there were multiple cortical effractions, particularly along the therapy, antiseptic mouthwash, scar care and the recommendation of an
lingual border. appropriate diet.
At the mandibular angle, there was an expansion of the lingual cortex Macroscopically, upon cross-section, the lesion appeared grayish,

Fig. 1. Constructed pantomographies.


A: Localized odontogenic myxoma around tooth 36 with root resorption.
B: 5-year recurrence of a larger odontogenic myxoma, displacing tooth 35.

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H. Trode et al. International Journal of Surgery Case Reports 112 (2023) 108945

Fig. 3. Rounded to spindle-shaped cells dispersed in a partly fibrous myxoid matrix Hematoxylin eosin staining ×20.

Fig. 4. Preoperative CT scan showing extensive osteolysis of the cortical bone, mainly lingual, basilar edge (Fig. 3A) and involvement of the right condylar
head (Fig. 3B).

translucent, and mucinous. surgeon. Four months later, four implants were placed. Dental rehabil­
Pathological analysis revealed an odontogenic fibromyxoma char­ itation was achieved with an implant-supported bridge one year after
acterized by stellate, spindle-shaped or round cells with cytoplasmic resection surgery (Fig. 6).
expansions, without mitotic activity, dispersed within a myxoid sub­ After forty months post-surgery, there was no radiological evidence
stance containing collagen fibers (Fig. 5). No tumor capsule was of recurrence.
observed, but peripheral fibrous densification was present. The medul­
lary space was infiltrated by proliferation and no odontogenic epithe­
lium was identified. Margins of resection were free of tumoral lesion. 3.3. Case 3 presentation
Lymph node analysis showed a non-specific reactive lymphadenitis.
To achieve implant-supported rehabilitation, the patient received a A 15-year-old patient with no medical history was referred by his
gingival graft from palatal mucosal on the fibula flap, by an oral dentist to the Oral Surgery Department of the Metz Regional Hospital
due to the incidental radiological discovery of a radiolucent lesion in the

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H. Trode et al. International Journal of Surgery Case Reports 112 (2023) 108945

Fig. 5. Histological sections.


A: Proliferation of spindle cells within a fibromyxoid matrix, infiltrating the adjacent bone trabeculae (arrow), without tumor capsule - Hematoxylin and eosin stain
×2,5.
B and C: Cellularity within the matrix from low (Fig. 7B) to high (Fig. 7C) collagen density – Hematoxylin and eosin stain ×10.

Fig. 6. Dental panoramic radiograph 40 months post-operatively.

mandibular symphysis. There was no evidence of recurrence detected 40 months


The lesion was entirely asymptomatic. Upon clinical examination, no postoperatively.
abnormalities were noted. There was no vestibular swelling, no pain and
the teeth adjacent to the lesion were vital. 4. Discussion
CBCT showed a well-defined multilocular radiolucency adjacent to
the apex of the teeth 31 to 34, measuring 19 × 15 mm. Cortical bone Odontogenic myxoma is a relatively rare benign tumor first
appeared locally thinned, but no signs of resorption or tooth displace­ described in 1947 by Thoma and Goldman [4]. According to the latest
ment were observed (Fig. 7). 2017 WHO classification of head and neck tumors, it is classified as a
A surgical procedure under general anesthesia was performed, con­ benign odontogenic mesenchymal tumor [1].
sisting of enucleation of the lesion and curettage of the excision cavity. Although its odontogenic origin is a subject of debate, several factors
This surgical intervention was conducted by an experienced oral sur­ suggest its association with tooth development. These factors include its
geon. Post-operative care included the prescription of analgesics, frequent occurrence in the maxillo-mandibular site, particularly in the
Amoxicillin antibiotic therapy, antiseptic mouthwash and the recom­ dentate region, its resemblance to different tissues of a developing tooth,
mendation of an appropriate diet. the inconsistent presence of an odontogenic epithelium and the possible
Pathological findings showed a poorly defined, non-encapsulated association with an impacted or delayed eruption tooth [5–7].
lesion characterized by proliferation of small spindle-shaped or stel­ The intraosseous (or central) location of odontogenic myxoma is the
late cells with condensed nuclei devoid of atypical character and most common. All the presented cases in the series are central forms.
without significant mitotic activity. These cells were arranged in an Similar to other odontogenic tumors, it can also be localized in the
abundant myxoid background. Immunohistochemical study using anti- peri-maxillary soft tissues such as the gingiva and alveolar mucosa
CK AE1/AE3 antibodies confirmed the absence of epithelial structure (peripheral form) [8].
(Fig. 8). The frequency of odontogenic myxoma among odontogenic tumors

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H. Trode et al. International Journal of Surgery Case Reports 112 (2023) 108945

Fig. 7. Constructed pantomography showing a multilocular osteolytic lesion.

Fig. 8. Histological sections - Hematoxylin and eosin stain ×10.


A: Spindle cells proliferation within a myxoid matrix.
B: Residual intralesional bone trabeculae (*) within the myxoid proliferation.

varies widely across studies, ranging from 0.5 to 20 % [9,10]. Most to another, highlighting the importance of considering a wide range of
authors have described a slight female predominance [11,12]. This benign and potentially malignant differential diagnoses, particularly
condition can occur at any age, but it tends to have a predilection for when periosteal reaction is present [15].
individuals in their third decade of life [10,13]. The predominant location is the mandibular region, more commonly
Most of the odontogenic myxomas are incidentally discovered in the posterior than the anterior part [11,13,16]. In our series, all cases
through radiography and are asymptomatic, as was observed in two of were located in the mandible, as are two thirds of odontogenic myxomas
our patients. Otherwise, odontogenic myxoma appears as a painless, [17].
slow-growing swelling that may lead to facial deformity [14]. Odontogenic myxoma is a non-encapsulated, locally invasive tumor
Radiologically, the majority of cases show unilocular or multilocular that infiltrates adjacent bone. Microscopically, it is characterized by a
radiolucency capable of eroding and expanding cortical bone. The proliferation of spindle or star-shaped cells, dispersed in a myxoid to
multilocular form, as observed in our cases, is the most common pre­ fibromyxoid matrix. The presence of a significant amount of collagen
sentation. A “soap bubble”, “honeycomb”, or “tennis racket” appearance may catgorize the tumor as an odontogenic fibromyxoma. Only one of
is characteristic, but not specific to odontogenic myxoma. Dental our cases is an odontogenic fibromyxoma, with strong collagenous
displacement is more frequent than dental resorption [11]. component.
The radiological presentation can vary significantly from one lesion Some bone trabeculae may be visible within the lesion, contributing

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H. Trode et al. International Journal of Surgery Case Reports 112 (2023) 108945

to the “soap bubble” radiological appearance or suggesting a mixed treatment should be pursued [10,13,31]. Kauke et al. state that the
lesion. The presence of an odontogenic epithelium (lamina remnant) is maximum diameter alone is insufficient for assessing the aggressiveness
possible in 13 % of cases but is not essential for the diagnosis [8,13,18]. of the lesion and determining the appropriate treatment approach [32].
Although odontogenic myxoma is classified as a benign tumor, it Various scanographic characteristics also necessitate careful
possesses the potential for locally invasiveness into surrounding tissues, consideration: type of locularity, appearance of the cortices (integrity,
sometimes leading to extensive tumors [19]. effraction), appearance of the tumor margins, tumor location (complex
In cases of extensive, aggressive-appearance osteolytic lesions, a bi­ surgical access), involvement of the basilar margin, height of residual
opsy is often performed initially to establish a diagnosis, as was the case bone [10,22,24,26,27,30,31,33].
in case 2. A prior diagnosis is crucial for planning an appropriate surgical Conservative treatment is more delicate in cases of cortical discon­
strategy [20]. tinuity and multilocular tumors. A history of recurrence is also a sig­
Treatment of odontogenic myxoma is surgical. Due to its low inci­ nificant factor in the decision-making process.
dence, there is no standardized recommendation for its management. In addition to tumor characteristics, patient-specific factors need to
The choice of treatment is a topic of debate in the literature. Treatment be considered, in particular age, co-morbidities and compliance with
options can vary from conservative approaches, such as simple enucle­ follow-up [17,21,28].
ation, to more radical procedures involving resection with margins of up As a summary, we propose a decision-making algorithm based on
to 1.5 cm [21]. both radiological criteria and patient-specific characteristics, to help
Recurrence rate is estimated to range from 10 and 43 % [22]. Ac­ determine the most suitable treatment approach (Fig. 9).
cording to Chrcanovic et al., this rate is significantly influenced by the
type of treatment performed (conservative or radical). 5. Conclusion
Absence of capsule, local tumor infiltration, gelatinous and friable
aspect of the tumor make a complete removal difficult. These factors are Odontogenic myxoma is a benign but its potential for aggressiveness
sources of recurrence after treatment [23–25]. and tissue destruction cannot be underestimated.
Radical treatment allows an improved control of the risk of recur­ The potential for recurrence and its difficulty of removal must be
rence by conducting an extended excision in healthy areas, but at the taken into account when planning the management. The balance be­
expense of aesthetic and functional sequelae. tween benefits and risks must be carefully assessed and customized to
In case 1, the patient presented a recurrence of an odontogenic each patient. Regular follow-up remains crucial, regardless of the chosen
myxoma measuring 55 mm long, with invasion of the lingual cortex. In treatment approach.
this case, the height of residual bone after enucleation and extensive
curettage, as well as the absence of basilar edge involvement are Consent
decision-making criteria in favor of conservative treatment [26,27].
After reviewing the literature, Kansy et al. suggest that radical sur­ Written informed consent was obtained from the patients for publi­
gery does not always seem to be the most appropriate to reduce the cation of these case reports and accompanying images. Written informed
recurrence rate [28]. Conservative surgery, whenever feasible, results in consent was obtained from the patient's parents/legal guardian for
fewer morphological and functional sequelae than radical surgery [29]. publication and any accompanying images.
Case 1 illustrates the importance of regular post-treatment follow-up, A copy of the written consent is available for review by the Editor-in-
especially when conservative treatment is employed. Five years after the Chief of this journal on request.
initial surgery, the patient experienced a recurrence that reached a size
of 55 mm.
Ethical approval
According to Chrcanovic et al., recurrence occurs within 2 years
post-surgery in 73 % of cases [13]. A rigorous follow-up during the
Exception from ethical approval because the study was a case series
initial post-operative years is essential and should be maintained over
of 3 case reports. This was research not involving the human person.
the long term. This vigilance facilitates the early detection of potential
Patients provided written consent to undergo the procedures described
recurrences, regardless of the type of treatment performed.
and for their data and anonymous images to be published.
Long term recurrence can still occurs, even following radical surgery.
MRI, due to its ability to explore soft tissues, could be valuable in the
Funding
follow-up of odontogenic myxoma, particularly in cases where doubts
exists regarding recurrence.
No funding was received.
In case 2, the patient presented an extensive lesion. CT scan indicated
multiple cortical effractions, and extensive cortical expansion notably
affecting the basilar margin and mandibular angle. The bone thickness Author contribution
was very low or absent in several areas, which reduces the consolidation
potential. Conservative treatment was therefore difficult to achieve Conceptualization, Data Curation: Trode Hannah, Celso Pouget,
[29]. Marouane Talbi, Etienne Simon, Muriel Brix.
Extension of the tumor to the condyle in this patient was also an Supervision: Muriel Brix.
argument in favor of radical treatment. Indeed, in situations involving Writing -original draft: Trode Hannah.
complex surgical access location, a wide resection is recommended in Writing -review and editing: Trode Hannah, Muriel Brix.
order to minimize the risk of recurrence that could potentially extend Validation: Trode Hannah, Celso Pouget, Marouane Talbi, Etienne
further, possibly up to the skull base, as exemplified in this case [28,30]. Simon, Muriel Brix.
In case 3, conservative treatment is performed immediately for both
diagnostic and therapeutic purposes, due to the well-defined appearance Guarantor
and small size of the lesion.
Different decision-making criteria have been delineated in the Ms Trode Hannah.
literature and illustrated through these cases. They mainly concern
tumor characteristics. Research registration number
The size of odontogenic myxoma is often employed by several au­
thors as a criterion for determine whether conservative or radical Not applicable.

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H. Trode et al. International Journal of Surgery Case Reports 112 (2023) 108945

Fig. 9. Decision support algorithm for the management of odontogenic myxoma [6,9,12,25,26,29–31,33–35].

Conflict of interest statement [12] A.C.U. Vasconcelos, F.M. Silveira, A.P.N. Gomes, S.B.C. Tarquinio, A.P.V. Sobral, J.
A.A. de Arruda, et al., Odontogenic myxoma: a 63-year retrospective multicenter
study of 85 cases in a Brazil population and a review of 999 cases from literature,
All authors declare that there are no financial and personal re­ J. Oral Pathol. Med. Off. Publ. Int. Assoc. Oral Pathol. Am. Acad. Oral Pathol. 47
lationships with other people or organizations that could inappropri­ (1) (2018) 71–77.
ately influence their work. [13] B.R. Chrcanovic, R.S. Gomez, Odontogenic myxoma: an updated analysis of 1,692
cases reported in the literature, Oral Dis. 25 (3) (2019) 676–683.
[14] Y. Kawase-Koga, H. Saijo, K. Hoshi, T. Takato, Y. Mori, Surgical management of
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