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Scully C. Oral and Maxillofacial Medicine.Edinburgh: Churchill Livingstone; 2013
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Soluk-Tekkeşin M, Wright JM. The World Health Organization classification of odontogenic lesions: a summary of the changes of the 2017 (4th) edition. Turk Patoloji Derg. 2018; 34 https://doi.org/10.5146/tjpath.2017.01410
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Glosser JW, Campbell JH. Pathologic change in soft tissues associated with radiographically ‘normal’ third molar impactions. Br J Oral Maxillofac Surg. 1999; 37:259-260 https://doi.org/10.1054/bjom.1999.0061
Kim J, Ellis GL. Dental follicular tissue: misinterpretation as odontogenic tumors. J Oral Maxillofac Surg. 1993; 51:762-767 https://doi.org/10.1016/s0278-2391(10)80417-3
Edamatsu M, Kumamoto H, Ooya K, Echigo S. Apoptosis-related factors in the epithelial components of dental follicles and dentigerous cysts associated with impacted third molars of the mandible. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2005; 99:17-23 https://doi.org/10.1016/j.tripleo.2004.04.016
Eliasson S, Heimdahl A, Nordenram A. Pathological changes related to long-term impaction of third molars. A radiographic study. Int J Oral Maxillofac Surg. 1989; 18:210-212 https://doi.org/10.1016/s0901-5027(89)80055-4
Yildirim G, Ataoğlu H, Mihmanli A Pathologic changes in soft tissues associated with asymptomatic impacted third molars. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008; 106:14-18 https://doi.org/10.1016/j.tripleo.2007.11.021
Kotrashetti VS, Kale AD, Bhalaerao SS, Hallikeremath SR. Histopathologic changes in soft tissue associated with radiographically normal impacted third molars. Indian J Dent Res. 2010; 21:385-390 https://doi.org/10.4103/0970-9290.70809
Rakprasitkul S. Pathologic changes in the pericoronal tissues of unerupted third molars. Quintessence Int. 2001; 32:633-638
Mesgarzadeh AH, Esmailzadeh H, Abdolrahimi M, Shahamfar M. Pathosis associated with radiographically normal follicular tissues in third molar impactions: a clinicopathological study. Indian J Dent Res. 2008; 19:208-212 https://doi.org/10.4103/0970-9290.42952
Bhushan NS, Rao NM, Navatha M, Kumar BK. Ameloblastoma arising from a dentigerous cyst – a case report. J Clin Diagn Res. 2014; 8:ZD23-25 https://doi.org/10.7860/JCDR/2014/5944.4387
Deshmane S, Khot K. Unicystic ameloblastoma arising from a dentigerous cyst: a case report. Int J Oral Health Med Res. 2016; 3:38-40
Leitner C, Hoffmann J, Kröber S, Reinert S. Low-grade malignant fibrosarcoma of the dental follicle of an unerupted third molar without clinical evidence of any follicular lesion. J Craniomaxillofac Surg. 2007; 35:48-51 https://doi.org/10.1016/j.jcms.2006.11.005
Chae MP, Smoll NR, Hunter-Smith DJ, Rozen WM. Establishing the natural history and growth rate of ameloblastoma with implications for management: systematic review and meta-analysis. PLoS One. 2015; 10 https://doi.org/10.1371/journal.pone.0117241
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Monitoring enlarged dental follicles: case report and literature review of an unusual presentation of a unicystic ameloblastoma

From Volume 48, Issue 7, July 2021 | Pages 564-569

Authors

Lily Long

BDS (Hons), MFDS

Dental Core Trainee, Department of Oral and Maxillofacial Surgery, Pinderfields Hospital, Wakefield

Articles by Lily Long

Email Lily Long

Jasveen Matharu

BDS MFDS

Oral Surgery Specialty Registrar, Department of Oral and Maxillofacial Surgery, Pinderfields Hospital, Wakefield

Articles by Jasveen Matharu

Sunil Sah

MBBS (London), BDS, MFDS, MRCS RCSEd, MSc(Oncology), FRCS(OMFS)

Oral and Maxillofacial Consultant, Department of Oral and Maxillofacial Surgery, Pinderfields Hospital, Wakefield

Articles by Sunil Sah

Abstract

An ameloblastoma is a benign, yet locally aggressive odontogenic tumour. The vast majority (80%) of ameloblastomas arise in the mandible, and unicystic ameloblastomas are commonly found in relation to an unerupted lower third molar. We present the case of a 39-year-old patient with an incidental finding of an enlarged dental follicle around an unerupted lower third molar that progressed to an extensive unicystic ameloblastoma. This ameloblastoma was decompressed and marsupialized before enucleation to reduce the risk of pathological fracture due to the extensive size of the tumour.

CPD/Clinical Relevance: The case is relevant to general dental practitioners when considering monitoring dental follicles of unerupted teeth because the enlarged dental follicle described progressed to an extensive odontogenic tumour.

Article

An ameloblastoma is a benign, yet locally aggressive odontogenic tumour accounting for 1% of tumours and cysts affecting the jaws. The vast majority (80%) of ameloblastomas arise in the mandible.1,2 Unicystic ameloblastomas account for only 10% of all ameloblastomas and are most commonly found in relation to an unerupted lower third molar.3 The WHO described four types of ameloblastoma: conventional; unicystic; peripheral/extra-osseous; and metastasizing/malignant.4

There are three proposed mechanisms for the formation of a unicystic ameloblastoma: reduced enamel epithelium: cystic degeneration of a solid ameloblastoma; and those arising from a dentigerous cyst.1,5

Significant controversy surrounds the choice of treatment modality for ameloblastomas owing to their propensity to recur. Therapy ranges from radical en-bloc resection with 1–2 cm margins to conservative decompression, marsupialization and enucleation. We present a case of a large unicystic ameloblastoma of the mandible that arose from an enlarged dental follicle surrounding an unerupted lower third molar to highlight the importance of detecting and monitoring follicular changes in general dental practice.

Case report

A 39-year-old male presented to the Pinderfields General Hospital oral and maxillofacial outpatient department in 2012 following referral from his general dental practitioner for the surgical removal of several retained roots. A DPT taken by his general dental practitioner to assess these retained roots in multiple quadrants identified an unerupted LR8 with widening of the follicle measuring 5–6 mm (Figure 1). The follicular enlargement was discussed with the patient with regard to a possible pathology. The patient was given the option of surgical management, but declined in favour of regular follow-up. The patient did not re-attend for the extractions until 2013, a new DPT demonstrated very mild distal expansion of the follicle, although it still measured 5–6 mm (Figure 2). Upon further discussion of the options of monitoring or surgical intervention, the patient opted for monitoring with radiographic review by his GDP.

Figure 1. DPT taken to assess multiple decayed teeth in 2012 shows an enlarged follicle surrounding the unerupted lower right third molar measuring approximately 6mm from crown to external margin
Figure 2. DPT taken to assess multiple decayed teeth in 2013 shows an enlarged follicle surrounding the unerupted lower right third molar.

Five years later, an urgent referral was received from the same patient's general medical practitioner regarding a right-sided facial swelling, initially diagnosed as parotitis and treated with antibiotics with limited improvement. The patient reported a 6-week history of a right-sided facial swelling, crackling sensation when yawning and an increase in swelling during the course of the day.

Examination revealed a large diffuse right-sided facial swelling extending from the lower border of the mandible to the right condyle, with crepitus on palpation. There was no limitation of mouth opening and no cranial nerve abnormalities. Intra-orally, there was an expansion of the right posterior buccal sulcus.

A further DPT revealed a large unilocular radiolucency displacing the LR8 inferiorly and extending throughout the ramus to the coronoid notch (Figure 3). A CT scan of the mandible revealed: ‘a unilocular expansile cystic lesion replacing the bone at the angle of mandible and coronoid, extending up into the condylar neck. The LR8 is displaced inferiorly with cortical thinning with marked expansion in the transverse diameter of up to 4 cm, and a craniocaudal length of over 6 cm. This is an apparently solitary lesion.’

Figure 3. DPT shows a large unilocular radiolucency displaying the LR8 inferiorly and extended throughout the ramus to the coronoid.

Owing to the size of the tumour, the patient underwent decompression and biopsy of the site under general anaesthetic via an intra-oral crevicular incision. An incisional biopsy of the cyst lining was obtained and a size 28 nasopharyngeal tube was inserted and sutured in place (Figures 4 and 5).

Figure 4. Size 28 nasopharyngeal tube used to decompress the ameloblastoma.
Figure 5. Nasopharyngeal tube sutured in place.

Post-operative antibiotics (co-amoxiclav 625 mg for 7 days), analgesia (paracetamol, ibuprofen, codeine) and chlorhexidine 0.2% mouthwash were prescribed, along with instructions to irrigate the site with warm salt water three times daily via the nasopharyngeal tube. The patient was advised to avoid all contact sports and keep to a soft diet.

Histopathology revealed a stratified squamous epithelium with a palisaded basal layer and a superficial layer of parakeratin. There was artificial clefting and detachment from the underlying stroma, as well as several areas with an appearance of stellate reticulum. A diagnosis of unicystic ameloblastoma was made.

The patient was reviewed clinically and radiographically at 3-monthly intervals. After 14 months, DPT and a CT scan demonstrated a decrease in tumour size, good bony infill and favourable movement of the inferiorly displaced LR8. Clinically, the facial swelling had decreased over this period.

The tumour was enucleated once there was a good level of bony infill within the area 15 months after decompression following the incision previously made in the LR6,7,8 region. The cyst was enucleated and a peripheral ostectomy performed using a Christmas tree bur within the bony cavity until healthy bleeding bone was reached. A decision to extract the LR8 and LR7 was made because both were closely involved with the cyst. The patient was given post-operative antibiotics for 7 days (co-amoxiclav 625 mg) and reviewed at 1 and 6 months.

Histopathology confirmed the diagnosis of a unicystic ameloblastoma.

Twelve months post-operatively the lesion had healed well. DPT (Figure 6) showed good bony infill. The patient was asymptomatic and had no sensory or motor deficits. Annual clinical and radiographic review was planned to identify any recurrence.

Figure 6. Post-operative DPT taken in 2019 shows good bony infill and no obvious pathology.

Discussion

Early recognition and monitoring

The dental follicle is part of the tooth germ, which originates from the odontogenic ectomesenchyme. Once a tooth has fully developed, the coronal part of the follicle, which is termed the pericoronal sac or follicle, can occasionally sit adjacent to the crown of unerupted teeth.6 It is made up of fibrous connective tissue and frequently contains epithelial residues of odontogenesis, which could be the start of pathology.7 Radiographically, unerupted/impacted wisdom teeth may present with a surrounding thin pericoronal radiolucency, the thickness of which considered ‘normal’ being reported as less than 2.5–3 mm.8,9,10 However, there is no generally accepted consensus on the clinical criteria to differentiate between normal and pathological conditions based on radiographic features.

Even with this ‘normal’ value in mind, various studies have reported pathological changes between 23% and 58.5% with pericoronal follicles that are 2.5 mm or less, particularly those associated with the lower third molar.11,12

Dentigerous cysts are the most common pathological change identified in dental follicular tissue, with impacted third molar pathological follicular tissues reported as being 70–86%.13,14 Ameloblastomas are the second most frequently seen pathological change, with up to 20% of all unicystic ameloblastomas forming within the wall of a dentigerous cyst.15 Several aetiological factors have been proposed for the explanation of this change, and include trauma, inflammation, nutritional deficiency and viral infections.16

There have also been reports of other pathological entities found in the pericoronal follicle of an unerupted or impacted tooth, such as calcifying odontogenic cysts, odontogenic keratocysts, myxomas, odontogenic fibromas, muco-epidermoid carcinoma and fibrosarcomas.11,12,13,14,15,16,17

The growth rate of such pathologies would be dependent on its histological nature, with a meta-analysis reporting the ameloblastoma specific growth rate as 87.84%/year.18 Dentigerous cysts can also exhibit rapid growth, having been reported as up to 5 cm over 3–4 years.19

In 2012 and 2013, the size of the follicle in our patient was 5–6 mm, and at re-attendance in 2017, this had increased to 40 mm (transverse) x 60 mm (craniocaudal), an approximate growth of 54 mm over 4 years (13.5 mm growth/year or 78%/year). It is unknown whether the tumour grew at a uniform rate during this time or whether there was a rapid period of growth preceding the patient's symptoms because the patient did not attend for regular reviews of the cyst with his GDP as advised. A systematic review and meta-analysis found that many patients with large ameloblastomas reported slow growth over a period of years, before a short episode of rapid growth which prompted them to seek medical attention. It is thought that the growth pattern is not linear, but begins slowly before accelerating.18 However, it is difficult to study the growth of ameloblastomas as treatment is initiated immediately following detection. Few case studies where a patient has refused surgical intervention, thereby allowing the growth to be monitored, have been reported, leading to low quality of evidence.

On presentation, the patient described a ‘crackling sound’ when yawning, crepitus was also found on palpation of the swelling. This phenomenon is referred to as ‘egg shell cracking’ and is an important diagnostic feature of an ameloblastoma. The slow bucco-lingual expansion of an ameloblastoma allows the periosteum to form a thin layer of bone around the expanding lesion, this bone cracks on palpation leading to crepitus.20

It can be difficult to monitor these lesions radiographically due to their tendency to expand bucco-lingually. We appreciate our case was above the recommended normal follicular size of 2.5–3 mm; however, studies have shown radiographically normal follicular radiolucencies associated with pathological entities on histological assessment. GDPs should habitually assess the size of dental follicles when reporting on radiographs, such as DPTs. Where there are concerns regarding enlargement of the follicle, regular radiographic monitoring is ideal, and we suggest these radiographic reviews be at annual or bi-annual intervals initially, with longer intervals if there are no changes. Previous imaging, CBCT and clinical assessment should also be considered.

Therefore, the role of the GDP is to ensure clinical assessment for signs of expansion in areas of impacted or unerupted teeth, carrying out peri-apical or DPT radiographic review if required, with comparison to previous imaging, and referral to secondary care oral and maxillofacial departments where there is any concern. If a DPT image has been taken in the secondary care setting, it would be valuable to have this available at the general dental practice for reference.

Decompression

Although ameloblastomas are histologically benign, management remains controversial owing to their tendency to recur. The risk of recurrence depends on the type of ameloblastoma: solid and multicystic ameloblastomas have a much higher recurrence rate (60–80%) compared to their unicystic counterparts (3–30%).21,22 For this reason, en-bloc or marginal resection and reconstructive plating is widely regarded as the treatment of choice for solid and multicystic ameloblastomas due to the unacceptably high recurrence rate following more conservative approaches. Many argue that this rule should extend to the management of unicystic ameloblastomas, especially mural types where the epithelium invades the cyst wall. A systematic review of the management of ameloblastomas found that unicystic ameloblastomas treated with simple enucleation were associated with recurrence rates ranging from 30% to 60%, with many arguing that this is unacceptably high.18 The time between initial treatment and recurrence of tumours treated in this way was up to 10 years, which highlights the importance of long-term follow up for these patients.23

Additional methods can be used during enucleation to minimise the risk of recurrence. Peripheral ostectomy can be performed immediately after enucleation, using a rotary bur within the bony cavity to remove a 2–3-mm margin, ideally until bleeding bone is achieved. This may be followed by intralesional adjunctive therapy via the application of the chemical tissue fixative, Carnoy's solution. This is usually composed of ethanol, ferric chloride and glacial acetic acid and can be formulated with or without chloroform. (As chloroform is carcinogenic, many surgeons do not use this formulation.24) One study analysed 29 cases of unicystic ameloblastoma that had a treatment regimen of enucleation followed by application of Carnoy's solution and found the recurrence rate to be 10%.25 Another study investigated eight cases of unicystic ameloblastoma treated with enucleation, peripheral ostectomy and application of Carnoy's solution, and concluded that there were no cases of recurrence at 6 years.26

It is argued that enucleation, marsupialization or decompression are justified in preference to radical surgery, with the latter having risk of loss of teeth, masticatory dysfunction, psychological effects and aesthetic changes.27 Studies have shown that the unicystic variety shows less invasive characteristics, and therefore responds more favourably to conservative approaches.28

Owing to the extensive size of the tumour described in this case, marsupialization and decompression were used to decrease the size of the tumour, prior to enucleation. Thus, reducing the risk of pathological fracture. Decompression of the tumour also allowed for bony infill around the roots of adjacent teeth and the inferior alveolar nerve, therefore reducing the risk of damage to such structures if surgery had been undertaken immediately.

A systematic review found that marsupialization without further treatment led to lower recurrence rates than enucleation alone.29 Marker et al recommended that decompression should be performed for over a year, as described here (15 months), as the longer the decompression, the better the prognosis.30

Conclusion

This case highlights the importance of dental follicular assessment, especially in relation to unerupted third molars, because of the risk of pathological change and ameloblastoma formation within enlarged dental follicles. Annual or bi-annual radiographic assessment should be carried out for any follicles found to be greater than the accepted normal range (>3 mm). These intervals may be extended over time if no changes are detected. DPT imaging may be difficult for determining expansion, and further imaging may be of value. Where there is concern, referral to the local oral and maxillofacial department for further assessment is recommended.

This case also highlights the successful treatment of a unicystic ameloblastoma with decompression and marsupialization, with subsequent enucleation, avoiding radical surgery and any associated post-operative complications. We would like GDPs to be aware of this treatment modality.