Unusual giant complex odontoma: A case report

Unusual giant complex odontoma: A case report

Journal Pre-proof Unusual Giant Complex Odontoma: A Case Report ´ Pieretti Bueno Mariana Lobo Bergamini Fernando Melhem Natalia Elias Paulo Henrique B...

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Journal Pre-proof Unusual Giant Complex Odontoma: A Case Report ´ Pieretti Bueno Mariana Lobo Bergamini Fernando Melhem Natalia Elias Paulo Henrique Braz-Silva Emanuela Prado Ferraz

PII:

S2468-7855(19)30295-2

DOI:

https://doi.org/doi:10.1016/j.jormas.2019.12.009

Reference:

JORMAS 779

To appear in:

Journal of Stomatology oral and Maxillofacial Surgery

Received Date:

11 December 2019

Accepted Date:

16 December 2019

Please cite this article as: Bueno NP, Bergamini ML, Elias FM, Braz-Silva PH, Ferraz EP, Unusual Giant Complex Odontoma: A Case Report, Journal of Stomatology oral and Maxillofacial Surgery (2020), doi: https://doi.org/10.1016/j.jormas.2019.12.009

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Unusual Giant Complex Odontoma: A Case Report Natália Pieretti Buenoa, Mariana Lobo Bergaminib, Fernando Melhem Eliasa, Paulo Henrique Braz-Silvab, Emanuela Prado Ferraza a

Department of Oral and Maxillofacial Surgery, Prosthesis and Traumatology, School of

Dentistry, University of Sao Paulo, Sao Paulo, SP, Brazil. b

Department of Stomatology, School of Dentistry, University of Sao Paulo, Sao Paulo, SP,

Brazil. * Corresponding Author Emanuela Prado Ferraz Department of Oral and Maxillofacial Surgery, Prosthesis and Traumatology, School of

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Dentistry, University of São Paulo Lineu Prestes Avenue, 2227 05088-000, São Paulo, SP, Brazil

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Tel: +55 11 2648 8038

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Email: [email protected]

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Abstract Odontomas are benign, non-aggressive, and the most common odontogenic tumor of the jaws. Composed of dental tissues, it can be classified as compound or complex odontomas depending on their radiological and histological features. Among them, complex odontomas are less common and usually is presented as a small and asymptomatic radiopaque mass surrounded by a radiolucent halo, found on routine radiographic examination. Although benign tumors, odontomas can reach large sizes leading to facial asymmetry and decreasing bone strength, which predisposes fractures and infection. Our aim was to present a case report of an unusual giant mandibular odontoma on the left mandibular angle and ramus successfully treated by surgical excision and highlight the importance of the earlier diagnostic to minimize damages.

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Key-words: Odontogenic tumors; Complex Odontoma; Giant; Surgical removal

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Introduction Odontomas are the most common odontogenic benign tumor of the jaws. According to the World Health Organization, odontomas are considered mixed tumors due to their origin from epithelial and ectomesenchymal cells and are classified as Compound or Complex (1). Compound odontomas consist of many tooth-like structures while Complex odontomas present dental tissues occurring in more or less disorderly pattern (2) The etiology of the Odontomas is unknown, although it has been implicated in some pathological conditions as trauma, infection, hereditary syndromes (i.e., Gardner’s syndrome), and gene alterations that might result in an abnormal pattern of morphodifferentiation of cells that give rise to ameloblasts and odontoblasts (2,3). There is no consensus on the epidemiological and

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clinical features of these lesions (3). Complex odontomas are usually asymptomatic and located at posterior mandible and might be associated with missing or malpositioning teeth (3,4). Due to its slow growth and nonaggressive behavior, complex odontomas can reach large sizes leading to

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cortical bone expansion with facial asymmetry, and even they can erupt to the oral cavity and cause infections (5,6).

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Odontomas are commonly found and diagnosed during routine radiographic examination. Radiographic features depend on their development stage, from a radiolucent on the initial phase to a radiopaque mass at later stages (2,4). Complex odontomas present irregular and disorganized

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radiopaque mass, usually surrounded by a well-defined thin radiolucent halo (2,4). The diagnosis is based on clinical and radiographic/tomographic images and must be confirmed with the histopathological findings. The differential diagnosis from other calcified bone lesions such as

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osteoma, fibrous dysplasia, florid osseous dysplasia, or ossifying fibroma is important to dictate the appropriate treatment (5).

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Despite benign features and limited grown potential, the treatment option is surgical

removal. The lack of treatment might lead to tissue degeneration and cyst formation, teeth eruption disturbances, mandibular bone fracture, exposure to the oral cavity, and even infection (2,5-7). This paper aims to report a giant complex odontoma and to emphasize the importance of the radiographic findings and treatment planning.

Case Report

A 42-year male patient was referred to Oral Surgery Clinic of Dental School, complaining of limitation of mouth opening and asymptomatic swelling at the left mandibular angle for the past year, after a bone biopsy at the retromolar ipsilateral region. The medical history was unremarkable. Extraoral examination revealed a slight facial asymmetry with a smooth swelling at the left mandibular angle, and the mouth opening was limited to 23 mm. Intra-oral examination revealed the absence of the left mandibular third molar, mucosal fenestration, and exposition of overlying hard tissue, with signs of local infection (Figure 1A).

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The panoramic X-ray and cone-beam computed tomography images revealed a large amorphous radiopaque mass circumscribed by a thin irregular radiolucent halo measuring approximately 8 centimeters (Figure 1 B-H). The radiopaque mass extended from the distal to the mandibular second molar and overlying the third molar to the coronoid process. The mandibular canal was displaced inferiorly, but no neurosensory symptoms were noticed. An incisional bone biopsy was carried out under local anesthesia, and the histological examination revealed an irregular arrangement of dental tissues such as enamel matrix, tubular dentin, cementum, and pulp-like connective tissue (Figure 2), confirming the diagnosis of complex odontoma. A second surgical procedure for total lesion excision was performed under general anesthesia and

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through a submandibular surgical approach to expose the mandibular ramus and body. Briefly, a 4cm skin and subcutaneous incision were made below the inferior border of the mandible to expose the underlying platysma muscle that was bluntly dissected to protect a marginal branch of the facial nerve, directing to the inferior mandibular border. The pterigomasseteric sling and

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lining periosteum were divided to expose the mandibular body and angle. It was evident the expansion of the external cortical plate, which was removed using piezoelectric burs under saline

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irrigation (Figure 3A). The lesion was reduced and removed by blocks. After the removal of the entire lesion, the cavity was cleaned, and the wound was closed by layers (Figure 3 B-G). The

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post-operative period was uneventful and the patient six months after removal, with no radiographical signs of recurrence (Figure 4 A-B). A new histological examination was performed

the third molar area.

Discussion

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and confirmed the diagnosis of complex odontoma, followed by connective tissue surrounding

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Complex odontomas are characterized as a conglomerate and radiopaque amorphous

mass, usually located in the posterior mandible surrounding by a well-defined radiolucent rim on the radiographic exams, which leads to considering differential diagnoses from other bone lesions as osteoma, fibrous dysplasia, ossifying fibroma or ameloblastic fibro-odontoma (5,7). A strong relationship between complex odontoma and ameloblastic fibro-odontoma (AFO) has been reported (8). Based on the stage of development of both lesions, they can be histological and radiographic indistinguishable (8). Consequently, it was decided to group AFO under odontomas as developing odontomas (1). Accurate evaluation of clinical and complementary characteristics is essential to establish the correct diagnosis and the appropriate treatment. In rare cases, odontomas can reach large volumes and are labeled as Giant Odontomas (5-7,9,10). Giant odontomas can produce considerable cortical bone expansion leading to swelling and facial asymmetry, limited mouth opening, pain, teeth displacement (5-7,9,10). In the present case, the patient had a dental history of bone biopsy a few months ago, followed by

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persistent infection. The intra-oral examination revealed mucosal fenestration and the exposition of overlying hard tissue with signs of local infection. Probably, during the osteotomy for bone biopsy, the strip mucosa distal to the second molar was traumatized, leading to a delay on mucosa repair followed by local and persistent infection (11). Commonly, the treatment of complex odontomas consists of surgical excision, with no or low morbidity. In contrast, the treatment of giant odontomas presents higher rates of morbidity due to its size and proximity to important anatomical structures as an inferior alveolar artery and nerve. The surgical approach and the need for bone reconstruction should be considered according to clinical and radiographic features. It is essential to consider that temporary or permanent damage of the marginal branch of the facial nerve might be a complication of the submandibular

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approach; however, intra-oral access would not allow full visualization of the lesion and its adequate excision (10, 12).

The lesion was removed by a piecemeal resection using a piezoelectric tip, which does not cut soft tissue, thus preserving important adjacent structures and reducing postoperative

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discomfort. As the lingual cortex and the basal bone were preserved, the risk of a postoperative mandibular fracture was considered low, and no reconstruction was performed, as well as

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maxillomandibular immobilization. The patient was oriented to avoid physical contact and kept a soft diet for 30 days. Mandibular fractures, followed by the lesion or associated tooth removal

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are described, and alternative procedures like two-stages surgery or mandibular immobilization may be indicated (10, 12). Preoperative planning with accurate imaging exams, proper surgical technique, and the postoperative patient collaboration are essential for treatment success.

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CT scans evidenced an enlargement of the mandibular coronoid process ipsilateral to the giant odontoma. This finding, another rare condition, may be associated with increased temporalis

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muscle activity and mandibular hypomobility, explaining the persistent trismus (13). The patient is on physiotherapy, showing slowly but a progressive improvement on mouth opening. We will further consider a coronoidectomy, if necessary. Conclusion

Giant odontomas can cause cortical bone expansion with jaw asymmetry and limited

mouth opening. The treatment option is surgical removal and the recurrence is uncommon. Onestage surgery reduces morbidity, avoiding a second procedure for graft donor site or permanent damage to the inferior alveolar nerve. Ethical approval Not required. Financial support and sponsorship Nil. Authorship disclosure

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Conception: NPB, FME, PHBS, EPF.; Literature search: NPB, EPF; Manuscript preparation, editing and review: NPB, MLB, FME, PHBS, EPF. Conflict of interest

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No benefit of any kind will be received either directly or indirectly by the authors.

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References 1. Wright JM, Vered M. Update from the 4th Edition of the World Health Organization Classification of Head and Neck Tumours: Odontogenic and Maxillofacial Bone Tumors. Head Neck Pathol 2017;11:68-77. doi: 10.1007/s12105-017-0794-1. 2. Satish V, Prabhadevi MC, Sharma R. Odontome: A Brief Overview. Int J Clin Pediatr Dent 2011;4:177-85. doi: 10.5005/jp-journals-10005-1106 3. Hidalgo-Sánchez O, Leco-Berrocal MI, Martínez-González JM. Meta-analysis of the epidemiology and clinical manifestations of odontomas. Med Oral Patol Oral Cir Bucal. 2008;13:E730–E734. 4. Shafer WG, Hine MK, Levy BM. Textbook of Oral Pathology. 4th Ed. Philadelphia: WB

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Saunders. 1983. 5. Park JC, Yang JH, Jo SY, Kim BC, Lee J, Lee W. Giant complex odontoma in the posterior mandible: A case report and literature review. Imaging Sci Dent 2018;48:289-93. doi: 10.5624/isd.2018.48.4.289.

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6. Perumal CJ, Mohamed A, Singh A, Noffke CE. Sequestrating giant complex odontoma: a case

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7. Vengal M, Arora H, Ghosh S, Pai KM. Large erupting complex odontoma: a case report. J Can

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Dent Assoc 2007;73:169-73.

8. Singh AK, Kar IB, Mishra N, Sharma P. Ameloblastic fibroodontoma or complex odontoma:

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Two faces of the same coin. Natl J Maxillofac Surg 2016;7:92-5. doi: 10.4103/0975-

9. Spini PH, Spini TH, Servato JP, Faria PR, Cardoso SV, Loyola AM. Giant complex odontoma

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of the anterior mandible: report of case with long follow up. Braz Dent J 2012;23:597600.

10. Chrcanovic BR, Jaeger F, Freire-Maia B. Two-stage surgical removal of large complex odontoma. Oral Maxillofac Surg 2010;14:247-52. doi: 10.1007/s10006-010-0206-0.

11. Sculean A, Gruber R, Bosshardt DD. Soft tissue wound healing around teeth and dental implants. J Clin Periodontol 2014; 41: S6–S22 doi: 10.1111/jcpe.12206.

12. Blinder D, Peleg M, Taicher S. Surgical considerations in cases of large mandibular odontomas located in the mandibular angle. Int J Oral Maxillofac Surg. 1993 Jun;22(3):163-5. 13. Mulder CH, Kalaykova SI, Gortzak RA. Coronoid process hyperplasia: a systematic review of the literature from 1995. Int J Oral Maxillofac Surg 2012;41:1483-9. doi: 10.1016/j.ijom.2012.03.029. Figure Captions

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Figure 1. Clinical, radiographic, coronal, and axial computed-tomography images. The intra-oral image showed the clinical condition with spontaneous drainage of purulent secretion (A). The panoramic and tomographic views revealed a radiopaque lesion surrounded by a thin radiolucent ring at mandibular angle and ramus, associated to the ipsilateral impacted third-molar (B-E). It was worth to notice the expansion of buccal cortical bone and enlargement of the coronoid process (F-H). Figure 2. Photomicrography of decalcified section stained with hematoxylin and eosin of the complex odontoma. The image revealed a disorganized mass of randomly-arranged dentin mixed with enamel, matrix, and pulp-like connective tissue. Magnification: 200x. Figure 3. Intraoperative exposure of the mandibular body, angle, and ramus using a

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submandibular approach (A). Removal of buccal cortical and lesion exposure (B-C). Sectioning of the odontoma was performed with a piezoelectric bur under continuous saline irrigation and chisel (D-E). Clinical aspect of the bone defect after complete removal of the entire lesion (F).

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Macroscopic aspects of the fragments and the associated mandibular third and second molars (G). Figure 4. Post-operative panoramic X-ray, 30 (A), and 150 (B) days after the surgical procedure

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for total excision, with no signs of recurrence.

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