References

Thoma KH, Goldman HM. Central myxoma of the jaw. Oral Surg Oral Med Oral Pathol. 1947; 33:532-540
Kansy K, Juergens P, Krol Z, Paulussen M, Baumhoer D, Bruder E Odontogenic myxoma: diagnostic and therapeutic challenges in paediatric and adult patients – a case series and review of the literature. J Craniomaxillofac Surg. 2012; 40:271-276
Simon EN, Merkx MA, Vuhahula E, Ngassapa D, Stoelinga PJ. Odontogenic myxoma: a clinicopathological study of 33 cases. Int J Oral Maxillofac Surg. 2004; 33:333-337
Lo Muzio L, Nocini P, Favia G, Procaccini M, Mignogna MD. Odontogenic myxoma of the jaws: a clinical, radiologic, immunohistochemical, and ultrastructural study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1996; 82:426-433
Martinez-Mata G, Mosqueda-Taylor A, Carlos-Bregni R, de Almeida OP, Contreras-Vidaurre E, Vargas PA Odontogenic myxoma: clinico-pathological, immunohistochemical and ultrastructural findings of a multicentric series. Oral Oncol. 2008; 44:601-607
Noffke CE, Raubenheimer EJ, Chabikuli NJ, Bouckaert MM. Odontogenic myxoma: review of the literature and report of 30 cases from South Africa. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007; 104:101-109
Leiser Y, Abu-El-Naaj I, Peled M. Odontogenic myxoma – a case series and review of the surgical management. J Craniomaxillofac Surg. 2009; 37:206-209
Subramaniam SS, Heggie AA, Kumar R, Shand JM. Odontogenic myxoma in the paediatric patient: a review of eight cases. Int J Oral Maxillofac Surg. 2016; 45:1614-1617
Department of Health and Social Care. Ionising Radiation (Medical Exposure) Regulations 2017: guidance. https://www.gov.uk/government/publications/ionising-radiation-medical-exposure-regulations-2017-guidance (Accessed 29 October 2018)
Kaczmarzyk T, Mojsa I, Stypulkowska J. A systematic review of the recurrence rate for keratocystic odontogenic tumour in relation to treatment modalities. Int J Oral Maxillofac Surg. 2012; 41:756-767
Ribeiro Junior O, Borba AM, Alves CA, de Gouveia MM, Coracin FL, Guimaraes Junior J. Keratocystic odontogenic tumors and Carnoy's solution: results and complications assessment. Oral Dis. 2012; 18:548-557
Fabrizi L, Taylor GW, Canas B, Boobis AR, Edwards RJ. Adduction of the chloroform metabolite phosgene to lysine residues of human histone H2B. Chem Res Toxicol. 2003; 16:266-275
Barros RE, Dominguez FV, Cabrini RL. Myxoma of the jaws. Oral Surg Oral Med Oral Pathol. 1969; 27:225-236
National Institute for Health and Care Excellence (NICE). Suspected cancer: recognition and referral (NG12). https://www.nice.org.uk/guidance/ng12 (Accessed 29 October 2018)
Department of Health. Referral to treatment consultant-led waiting times rules suite. https://www.gov.uk/government/publications/right-to-start-consultant-led-treatment-within-18-weeks (Accessed 29 October 2018)
Santosa RE. Provisional restoration options in implant dentistry. Aust. 2007; 52:234-242

Odontogenic Myxoma of the Maxilla: Diagnostic Considerations, Surgical Resection and Prosthetic Rehabilitation

From Volume 45, Issue 10, November 2018 | Pages 985-990

Authors

Yehya EK Gamie

BDS, MFDS RCS(Ed)

Oral and Maxillofacial Surgery Trainee/Dental Surgeon, York Hospital/Aintree University Hospital, Liverpool, UK

Articles by Yehya EK Gamie

Zakareya Gamie

BSc(Hons), MBChB, MRCS, PGCert (Genomic Medicine)

PhD Student and Honorary Clinical Research Associate in Trauma and Orthopaedic Surgery, Newcastle University, UK

Articles by Zakareya Gamie

David Seymour

BChD, MFDS RCS(Ed), MSc ClinDen(Rest Dent) FDS RCS(Ed)

Specialty Trainee in Restorative Dentistry, Department of Restorative Dentistry, Leeds Dental Institute, Clarendon Way, Leeds LS2 9LU, UK

Articles by David Seymour

Paul H Whitfield

MBBS, BDS, FDS, FRCS

Consultant Oral and Maxillofacial Surgeon, York Hospital, Wigginton Road, York, North Yorkshire, YO31 8HE, UK

Articles by Paul H Whitfield

Abstract

A 15-year-old female with a history of orthodontic treatment was referred by her general dental practitioner (GDP) to the Oral and Maxillofacial Surgery (OMFS) department after noticing a right buccal maxillary swelling during a routine check-up. Examination and radiographic investigations revealed a lesion extending from UR3 to UR6 causing bony expansion with no evidence of root resorption. Following biopsy and histopathological analysis, a diagnosis of Odontogenic Myxoma (OM) was made. Treatment involved a segmental resection and fitting an immediate partial denture. Options for long-term rehabilitation include removable and implant-supported prostheses.

CPD/Clinical Relevance: This case highlights the importance of thorough clinical and radiographic examination, and joint treatment planning between the Restorative and OMFS departments.

Article

Odontogenic Myxoma (OM) was first described in 19471 and is a benign, locally invasive, non-metastasizing neoplasm of mesenchymal origin.2,3,4 It is rare and accounts for between 1% and 17.7% of odontogenic tumours.5 Occurring at any age, it is most common in the second and third decades of life, with a higher incidence in females and in the mandible.2,3,4,5,6 Many cases are detected during routine dental examinations.5,7 Features include slow and asymptomatic growth, tooth displacement and root resorption, delayed eruption, pain, facial asymmetry, cortical bone perforation and soft tissue invasion.2,3,4,5,6,7,8 Radiographically, OM may appear as unilocular or multilocular radiolucencies and are described as having a soap bubble, honeycomb or tennis racket appearance.3,4,,6,7,8

Initial presentation

A fit and well 15-year-old female was referred to the OMFS department at York Hospital in April 2016 after attending her GDP for a routine examination. The GDP noticed an asymptomatic firm expansion of bone in the upper right quadrant along with a slightly displaced UR4. There was no evidence of erythema, ulceration or pus discharge (Figure 1).

Figure 1. Pre-operative intra-oral view showing bony expansion adjacent to the UR3 and a displaced UR4 and UR5.

Investigations

A panoramic radiograph taken in April 2016 (Figure 2) revealed a mixed radiolucent/radio-opaque area in the right maxilla extending from the distal aspect of the UR3 to the mesial aspect of the UR6, causing displacement of teeth. A cone beam computed tomography (CBCT) scan taken in July 2016 was more useful in exposing the dimensions of this lesion, along with a sparse and course trabecular pattern and patchy calcifications (Figure 3). Expansion of the buccal plate and right maxillary sinus encroachment could also be seen but no root resorption was evident.

Figure 2. Panoramic radiograph showing a radiolucent area in the right maxilla which extends from the distal aspect of the UR3 to the mesial aspect of the UR6. Root displacement can also be seen.
Figure 3. Pre-operative CBCT scan of the right midface showing the lesion. A sparse and course trabecular pattern and patchy calcifications are visible

Further examination under general anaesthesia (GA) in August 2016 revealed a soft tissue mass extending around the upper right premolars and a thick white jelly-like material invading into bone. A biopsy and histopathological analysis showed spindle-shaped cells with an eosinophilic cytoplasm embedded in loose myxoid matrix (Figure 4). A diagnosis of OM was reported in October 2016.

Figure 4. Histopathological analysis revealing the typical spindle cells of myxoma immersed in abundant myxomatous intercellular matrix (Hematoxylin-eosin stain, magnification 10X).

Surgical treatment and restorative rehabilitation

Surgical resection was performed under GA in January 2017. A buccal and palatal full mucoperiosteal flap was raised and UR3 and UR6 were extracted. A block resection with a safe margin was carried out using a piezoelectric saw along with an osteotome. This extended to a level below the infra-orbital foramen (Figure 5 a, b). The right buccal fat pad was used for immediate reconstruction and was sutured over the defect. The resection involved the antrum but oral mucosa closure was achieved and hence an obturator was not required.

Figure 5. Surgical resection of the tumour. (a) Intra-operative view showing the patient post resection. (b) The gross pathological specimen.

An upper partial immediate denture was designed in collaboration between the restorative dentist and surgeon (Figure 6). This was constructed via impressions taken prior to the planned surgery.

Figure 6. Immediate prosthetic rehabilitation using an Essix retainer (left) or immediate partial denture (right).

An immediate Essix retainer was also constructed as a reserve option if necessary (Figure 6). This was constructed by bonding acrylic teeth to a clear vacuuform material. The immediate denture was of reasonable fit and required minimum adjustment (Figure 7).

Figure 7. Immediate partial denture in situ intraoperative.

Discussion

Features of OM vary and it should be considered in the differential diagnosis of both radiolucent and mixed radiolucent/radio-opaque lesions in both jaws for all age groups. Table 1 shows differential diagnoses in adults and children and it includes a wide variety of benign but also malignant tumours.2 Clinical, radiographic and microscopic features are important in the diagnosis of head and neck lesions. Histology with immunohistochemical analysis is most reliable in distinguishing myxoma from malignant tumours.2


Children Adults
Benign Eosinophilic granuloma Ameloblastoma
Histiocytosis Odontoma
Intraosseous haemangioma Odontogenic keratocyst
Cherubism Aneurysmal bone cyst
Cranial fasciitis Central giant cell granuloma
Malignant Rhabdomyosarcoma Rhabdomyosarcoma
Eosinphilic granuloma Carcinoma
Metastasis
Ameloblastoma

Early diagnosis is vital and it is imperative that, during routine follow-up, extra- and intra-oral examinations are thorough and systematic. Prior to the GDP referral, the patient received orthodontic treatment in secondary care. A panoramic radiograph was taken in July 2015 (Figure 8) and this lesion was missed. This highlights the importance of carefully examining and reporting radiographs along with continued education and training in line with Ionizing Radiation (Medical Exposure) Regulations 2000 (IRMER).9

Figure 8. Panoramic radiograph following orthodontic treatment approximately nine months prior to the GDP referral.

Surgical management varies depending on the location, size of tumour, age of patient and individual experience.2 This ranges from enucleation and the application of Carnoy's solution to surgical resection with a 0.5–2 cm margin and immediate microvascular free flap reconstruction.2,3,4,7,8

Carnoy's solution penetrates cancellous spaces in bone and thus devitalizes and fixes remaining tumour cells. It is preferably not to be used in the vicinity of neural structures or the maxillary sinus to prevent neuropathy or necrosis of the sinus wall.10 Other complications include infection, dehiscence and bone sequestrum formation.11 Carnoy's solution has also been reformulated without chloroform because of its association with cancer and reproductive toxicity.12

Unlike most neoplasms, the myxoma is not encapsulated and myxomatous tissue infiltrates adjacent bone without immediate destruction. This could explain why conservative approaches, such as curettage or simple local excision, have a reported recurrence of approximately 10% to 33%.4,7 Despite aggressive local growth, OMs are not believed to undergo malignant transformation or metastasize.13

This patient was initially seen in the department within two weeks after the GDP referral, which is in line with National Institute for Health and Care Excellence (NICE) suspected cancer guidelines.14 The benign tumour was resected approximately nine months afterwards. This needs to be reviewed as the Department of Health states that patients have a right to start consultant-led treatment within a maximum of 18 weeks.15

Patients facing loss of their teeth and associated structures may experience apprehension towards losing their social image or daily function.16 The restorative dentist's role is to provide functional and aesthetic restorations for a smooth transition. A cobalt chrome partial denture can be provided once healing is complete, for increased comfort and retention whilst monitoring for any recurrence. In the case of a paediatric patient, several appliances could be provided until growth is complete prior to bone augmentation and implant rehabilitation.

A minimum of 5 year follow-up is advised prior to moving onto the final reconstructive phase,7 with the first 2 years being the period when the neoplasm is most likely to recur.4 Annual panoramic radiographs for all lesions are advocated (Figure 9). CBCT imaging is more reliable for maxillary lesions.8

Figure 9. Post-operative follow-up panoramic radiograph after the surgical resection of the OM.