Le Fort I osteotomy for removal of an odontoma from the maxillary sinus: a case report and literature review

doi:10.21203/rs.3.rs-1551935/v1

PDF

Case Report

Le Fort I osteotomy for removal of an odontoma from the maxillary sinus: a case report and literature review

https://doi.org/10.21203/rs.3.rs-1551935/v1

This work is licensed under a CC BY 4.0 License

Version 1

posted 15 Apr, 2022

You are reading this latest preprint version

Odontoma is a common odontogenic tumour in the maxilla-mandibular region, but whose occurrence is rare within the maxillary sinus. The objective of the present study was to report a case of surgical removal of an extensive complex odontoma from within the maxillary sinus by using the Le Fort I osteotomy. A 22-year-old male patient presented an asymptomatic radiopaque lesion with well-defined margins in the right sinus cavity. The treatment consisted of surgical removal by using the Le Fort I osteotomy to facilitate access, full excision of the lesion and maintenance of bone integrity. The treatment was considered safe and effective as it allowed adequate access and low post-operative morbidity.

Odontoma

Maxillary sinus

Case report

Osteotomy

Cone Beam Computed Tomography

The term odontoma was first introduced in 1867 by Paul Broca to describe any tumour lesion of odontogenic origin [1]. The Word Health Organisation (2017) classifies it as a mixed benign odontogenic tumour [2]. Nowadays, odontoma is not considered a true tumour and has been defined as a hamartomatous malformation consisting of dental tissues, mainly enamel and dentine with different proportions of pulp and cement [3, 4].

Odontoma, the most common, accounts for approximately 48.4% of the odontogenic tumours, being diagnosed during the first two decades of life [5]. It can be further classified as compound (with multiple tooth-like structures) and complex (an amorphous mass of disorderly arranged dental tissues), in which the latter usually occurs in the posterior region of the mandible or anterior region of the maxilla, but being rare in the maxillary sinus [2, 6].

The treatment for odontomas consists of periodical follow-up or surgical excision. Small-medium sized odontomas can be usually removed without difficulties depending on its localisation and proximity to adjacent structures. Nevertheless, access to extensive odontomas may be complicated [3, 6]. In 1988, Armstrong and Bhardwaj were the first authors reporting the use of Le Fort I osteotomy for removal of an odontogenic tumour [7]. Despite not being a new technique for treatment of lesions in the middle third of the face, there are only two cases in the literature reporting the use of the technique for removal of an intra-sinus complex odontoma [7, 8].

The objective of the present study was to report a case of surgical removal of an extensive odontoma from within the maxillary sinus by using the Le Fort I osteotomy, as well as to present a complete literature review of similar cases.

A 22-year-old male patient with no systemic disease initially sought dental care for orthodontic treatment. Panoramic radiograph showed presence of radiopaque image in the maxillary right sinus and the patient was then referred to the Oral-Maxillofacial Surgery Service of the Federal University of Juiz de Fora Hospital (HU-UFJF/EBSERH, Juiz de Fora, Minas Gerais, Brazil). Clinically, the patient presented neither facial swelling nor symptoms suggesting presence of pathology in the maxillary right sinus.

The patient was submitted to image acquisition by means of cone beam computed tomography (CBCT) operating at FOV of 6 x 8 cm, acquisition time of 17s, 120 kVp, 5mA, isotropic voxel size of 0.4 mm and 14-bit grey levels. The tomographic images were segmented and reconstructed three-dimensionally, indicating the presence of lesion in the sinus cavity in association with a retained tooth (i.e. second molar) and hyperdense pattern suggestive of mineralised tissue. From multiplanar reconstructions of the tomographic images, it was observed that the lesion showed well-defined margins extending anteroposteriorly and lateromedially for approximately 40 mm and 25 mm, respectively, and height of 46 mm (Fig. 1). The patient was submitted to incisional biopsy for histopathological exam and the diagnosis was of complex odontoma.

The surgical procedure for complete removal of the lesion was performed under general anaesthesia. Firstly, the surgical access to the maxilla was obtained buccally by detachment of the subperiosteal non-keratinised mucosa extending from the floor of the nasal fossa to the pterygo-maxillary region. Le Fort I osteotomy was performed by using a reciprocating saw (Stryker-CORE System) and finished with chisels. After osteotomy in the pterygo-maxillary region and maxillary mobilisation (Fig. 2A), the access to the sinus cavity was made possible and the lesion divided into several fragments for removing them completely from the cavity, thus facilitating the maintenance of the integrity of the bone walls of the maxillary sinus, Next, the walls of the maxilla were levelled by using rotary burs. An inter-occlusal surgical guide was positioned in order to stabilise the maxilla and the mandible by means of maxillo-mandibular block. The maxilla was repositioned by using the fixation system 2.0 with four L-shaped plates (Engimplan, Materialise Company, Rio Claro, São Paulo, Brazil) in the adjacent regions and piriform and zygomaticomaxillary openings bilaterally. The anatomopathological pieces removed from the lesion (Fig. 2B) had irregular shape, rough surface, hard consistency and brownish colour, then being sent to histological analysis (Fig. 2C-E).

The post-operatory tomographic images suggested proper positioning in addition to evidencing complete removal of the tumour (Fig. 3). The patient has been followed up for eight months and showing favourable evolution with occlusal stability, good healing aspect and no post-operatory complication.

The patient signed an informed consent form before undergoing the surgery and to allow publication of the images. All the procedures were carried out according to the Helsinki Declaration of the World Medical Association.

In order to facilitate the comparison with other similar case reports published elsewhere, we have made a search on PubMed database by using the keywords [complex odontoma] and [maxillary sinus]. Eighteen articles on complex odontomas in maxillary sinus were selected, but one did not fully describe the access and thus was excluded [9]. However, the search revealed 18 cases in the period between 1956 and 2022, with one of the articles reporting on two patients. These data are listed in Table I.

The mean age of the patients was 22.21 ± 8.86 years old, ranging from 11 to 48 years. The male:female ratio was 2.2:1. Data demonstrated that intra-sinus complex odontoma is more prevalent on the left side (63.15%), with a mean size of 38 x 34.3 mm. However, there is divergence in the literature as studies report that the majority of these lesions are detected during the second decade of life (mean age of 14 years), with little or no difference between genders [4].

Although intra-sinus odontoma is not common, the maxillary sinus is a site susceptible to pathologies of odontogenic origin due to its close anatomical relationship with teeth and periodontal tissues [25]. As well as in the present case, odontoma is usually found as an incidental radiographic finding, but the involvement of maxillary sinus should be carefully evaluated and the lesion removed because its growth can lead to local and systemic impairment. Among the possible complications associated to odontoma, one can site deviation or absence of tooth eruption, alveolar bone expansion [7, 8, 11, 14, 16, 18], regional lymphadenopathy [13, 21], nasal obstruction [10, 13, 19, 23], epistaxis, diplopia and sinusitis [10, 24]. Moreover, cystic formation may concomitantly occur, resulting in bone destruction [15, 21].

Different surgical approaches for removal of tumours in the middle third of the face have been described. The Caldwell-Luc technique was first described by George Caldwell in 1893 and Henri Luc in 1897 as a surgical approach for lesions in the maxillary sinus [26]. Such an approach is the most used conventional intraoral technique, in which the lesion is accessed through the anterior and lateral walls of the maxillary sinus. However, conventional techniques are not recommended for large or posterior lesions due to the limited visualisation and consequent impairment of the access to the lesion, which can damage the adjacent structures [27]. Moreover, these approaches also tend to cause bone defects on the sinusal walls, which may not be repaired and thus require reconstruction with donor site for bone grafting. If these osteotomy gaps are extensive, then the overlying soft tissues can collapse into the space normally occupied by those walls [8].

As an alternative to the conventional surgical approaches, the Le Fort I osteotomy provides better access to lesions located in the posterior region of the maxilla as the lesion’s margins and adjacent bone structures can be visualised. Therefore, this surgical approach allows accurately delineating the osteotomies to be performed, which ensures complete resection of the lesion and prevents relapse consequently [8, 27–29]. It is considered a versatile technique because one can change the horizontal osteotomy level depending on the location of the tumour and its proximity to other structures, such as orbit floor and skull base [30]. Scolozzi & Lombardi [29] emphasise that this technique is not aimed only at posteriorly located lesions, but also at those involving one or more maxillary walls, thus reducing loss bone.

In addition to the present study, only Armstrong & Bhardwj [7] and Korpi et al. [8] reported the use of Le Fort I osteotomy for removal of an intra-sinus complex odontoma. This technique allows better surgical predictability, causes no occlusal alteration and has low rate of post-operative morbidity. The possible complications are the same ones which may occur during the surgical correction of a dento-facial deformity. Hass Junior et al. [31] performed a systematic review and found that only 8.5% of the patients submitted to Le Fort I osteotomy had a post-operative complication. The most frequent adverse effects include haemorrhage, pulp necrosis, bucco-sinusal fistulas (oro-antral) lesions of nasolacrimal duct and secondary infections [28–31]. In the present case report, there were neither immediate nor long-term complications.

The surgical approach using the Le Fort I osteotomy was considered safe and effective for removal of this lesion as it permits better access, surgical predictability and optimal maintenance of the integrity of the maxillary bone.

Funding: This study did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Competing Interests: The authors declare that they have no known competing financial interests or personal relationships which could have appeared to influence the work reported in this article.

Author Contributions: All authors contributed to the study conception and design. Examination and surgery were performed by Livia Marques dos Santos, Zarife Tirapani Adum Resende, Bruno Augusto Benevenuto de Andrade and Daniel Amaral Alves Marlière. The first draft of the manuscript was written by Yuri de Lima Medeiros and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

Ethics approval: The manuscript did not require ethical approval. All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Consent to participate: Informed consent was obtained from the patient.

Consent to publish: The authors affirm that human research participants provided informed consent for publication.

Data availability statement: The datasets generated during and/or analysed during the current study are available from the corresponding author on reasonable request.

Satish V, Prabhadevi MC, Sharma R (2011) Odontome: a brief overview. Int J Clin Pediatr Dent 4:177–85.
EI-Naggar AK. Chan JKC, Grandis JR, Takata T, Slootweg PJ (2017) WHO Classification of Head and Neck Tumours: Odontogenic and Maxillofacial Bone Tumours. IARC: Lyon 224-6.
Hidalgo-Sánchez O, Leco-Berrocal MI, Martínez-González JM (2008) Metaanalysis of the epidemiology and clinical manifestations of odontomas. Med Oral Patol Oral Cir Bucal 13:E730-4.
Maltagliati A, Ugolini A, Crippa R, Farronato M, Paglia M, Blasi S, Angiero F (2020) Complex odontoma at the upper right maxilla: Surgical management and histomorphological profile. Eur J Paediatr Dent 21:199–202.
Silveira FM, Macedo CCS, Borges CMV, Mauramo M, Vasconcelos ACU, Soares AB, Martinez EF, de Araujo VC, Vered M, Salo T, Passador-Santos F (2021) Odontogenic tumors: An 11-year international multicenter study. Oral Dis 27:320–324.
Ćabov T, Fuchs PN, Zulijani A, Ćabov Ercegović L, Marelić S (2021) Odontomas: Pediatric Case Report and Review of the Literature. Acta Clin Croat 60:146–152.
Armstrong JE, Bhardwaj AK (1988) Le Fort I down-fracture of the maxilla: application to benign antral neoplasm. J Otolaryngol 17:288–92.
Korpi JT, Kainulainen VT, Sándor GK, Oikarinen KS (2009) Removal of large complex odontoma using Le Fort I osteotomy. J Oral Maxillofac Surg 67:2018–21.
Ferrer Ramírez MJ, Silvestre Donat FJ, Estelles Ferriol E, Grau García Moreno D, López Martínez R (2001) Recurrent infection of a complex odontoma following eruption in the mouth. Med Oral 6:269–75.
Gupta M, Das D (2015) Extensive complex odontoma in the maxillary sinus pushing 3rd molar near the orbital floor causing transiente diplopia and chronic sinusitis: a rare presentation and surgical management. J Maxillofac Oral Surg 14:338–40.
Visioli ARC, de Oliveira E Silva C, Marson FC, Takeshita WM (2015) Giant complex odontoma in maxillary sinus. Ann Maxillofac Surg 5:123–6.
Murphy C, O'Connell JE, Cotter E, Kearns G (2014) Management of large erupting complex odontoma in maxilla. Case Rep Pediatr 2014:963962.
Arunkumar KV, Vijaykumar, Garg N (2012) Surgical management of an erupted complex odontoma occupying maxillary sinus. Ann Maxillofac Surg 2:86–9.
Isler SC, Demircan S, Soluk M, Cebi Z (2009) Radiologic evaluation of an unusually sized complex odontoma involving the maxillary sinus by cone beam computed tomography. Quintessence Int 40:533–5.
Sales MA, Cavalcanti MG (2009) Complex odontoma associated with dentigerous cyst in maxillary sinus: case report and computed tomography features. Dentomaxillofac Radiol 38:48–52.
Singer SR, Mupparapu M, Milles M, Rinaggio J, Pisano D, Quaranta P (2007) Unusually large complex odontoma in maxillary sinus associated with unerupted tooth. Report of case and review of literature. N Y State Dent J 73:51–3.
Cabov T, Krmpotić M, Grgurević J, Perić B, Jokić D, Manojlović S (2005) Large complex odontoma of the left maxillary sinus. Wien Klin Wochenschr 117:780–3.
Mupparapu M, Singer SR, Rinaggio J (2004) Complex odontoma of unusual size involving the maxillary sinus: report of a case and review of CT and histopathologic features. Quintessence Int 35:641–5.
Clayman GL, Marentette LJ (1989) Complex odontoma of the maxillary sinus with a complete dentition. Otolaryngol Head Neck Surg 101:581–3.
Shatz A, Calderon S (1987) Complex odontoma in the maxillary sinus. An unusual presentation. Ann Dent 46:38–40.
Visscher JG, Güven O, Elias AG (1982) Complex odontoma in the maxillary sinus. Report of 2 cases. Int J Oral Surg 11:276–80.
Curreri RC, Messer EJ, Abramson AL (1975) Complex odontoma of the maxillary sinus: report of case. J Oral Surg 33:45–8.
Caton RB, Marble HB Jr, Topazian RG (1973) Complex odontoma in the maxillary sinus. Oral Surg Oral Med Oral Pathol 36:658–62.
Christensen RW (1956) Complex composite odontoma involving the maxilla and maxillary sinus; report of a case. Oral Surg Oral Med Oral Pathol 9:1156–64.
Press SG (2008) Odontogenic tumors of the maxillary sinus. Curr Opin Otolaryngol Head Neck Surg 16:47–54.
Huang YC, Chen W (2012) Caldwell-Luc operation without inferior meatal antrostomy: a retrospective study of 50 cases. J Oral Maxillofac Surg 70:2080–4.
Alexander R, Weber WD, Theodos LV, Friedman JS (1992) The treatment of large benign maxillary tumors via Le Fort I downfracture: report of two cases and review of the literature. J Oral Maxillofac Surg 50:515–7.
Quirino LC, Carvalho PHA, Santos JCD, Trento GDS, Gabrielli MAC, Pereira-Filho VA (2020) Le Fort I Down-Fracture for Management of Midface Tumors. J Craniofac Surg 31:e608-e12.
Scolozzi P, Lombardi T (2010) Removal of large complex odontoma using Le Fort I osteotomy. J Oral Maxillofac Surg 68:950–1.
Soares ECS, de Medeiros JR, Bezerra TP, Nogueira CB, Costa FW, Nogueira AS (2013) The use of Le Fort I Approach in the resection of an extensive ossifying fibroma. Oral Maxillofac Surg 17:209–12.
Haas Junior OL, Guijarro-Martínez R, de Sousa Gil AP, da Silva Meirelles L, de Oliveira RB, Hernández-Alfaro F (2017) Stability and surgical complications in segmental Le Fort I osteotomy: a systematic review. Int J Oral Maxillofac Surg 46:1071–87.

No competing interests reported.

PDF

Version 1

posted 15 Apr, 2022

You are reading this latest preprint version

Le Fort I osteotomy for removal of an odontoma from the maxillary sinus: a case report and literature review

Status:

Version 1

Abstract

Figures

Introduction

Case Report

Discussion

Conclusion

Statements And Declarations

References

Competing Interests

Status:

Version 1