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British Journal of Oral and Maxillofacial Surgery xxx (2016) xxx–xxx

Argument for the conservative management of mandibular


ameloblastomas
Jahrad Haq a , Sarah Siddiqui b , Mark McGurk c,∗
a SpR in Oral & Maxillofacial Surgery, King’s College Hospital, Denmark Hill, London, SE5 9RS
b Dental Student, Guy’s Hospital, Great Maze Pond, London, SE1 9RT
c Prof of Oral and Maxillofacial Surgery, University College London, Gower St, London, WC1E 6BT

Accepted 20 July 2016

Abstract

Ameloblastoma is a rare, benign, odontogenic tumour that affects the mandible more commonly than the maxilla. Solid or multicytic variants
are often resected and the defects reconstructed with a free flap. To establish the outcome after enucleation and application of Carnoy’s solution,
irrespective of histological subtype, we used the hospital’s histology database to identify all the patients treated between 2001 and 2014 by
one surgeon. Variables included patients’ characteristics, histological subtype, radiological appearance, follow-up period, and incidence of
recurrence. A total of 27 patients (13 male) were included, mean age 41 years (range 12-79). Fifteen (56%) had solid multicystic lesions, and
there was an overall predominance of the follicular or plexiform variant, or both. Of the 23 preoperative radiographs that were available, 17
lesions were unicystic, 5 multilobular and scalloped with no septa, and one had aggressive features of multilocularity and a poorly defined
peripheral margin. The mean duration of follow up was 38 months (range 3-156). Three patients had recurrence at 20, 27, and 35 months
postoperatively, and each had repeat enucleation and application of Carnoy’s solution. Reconstruction was not necessary, and to date none
has recurred. This study shows the potential benefits of conservative surgery and sterilisation of the cystic cavity with Carnoy’s solution.
Recurrence is low, and with vigilant surveillance, similar repeat procedures have been effective when necessary. A longer follow-up period
and larger numbers of patients are now needed to corroborate these findings.
© 2016 Published by Elsevier Ltd on behalf of The British Association of Oral and Maxillofacial Surgeons.

Keywords: Ameloblastoma; Carnoy’s; Enucleation

Introduction a high rate of recurrence (up to 60%-80%) if not ade-


quately removed.2 They have virtually no tendency to
In 2005 the World Health Organization (WHO) described metastasise.
ameloblastomas as benign, locally invasive, polymorphic Ameloblastomas affect young people. They are often diag-
neoplasms that consist of proliferating odontogenic epithe- nosed around the age of 36 years and they affect both sexes
lium (usually with a follicular or plexiform pattern) lying equally.3 Tumours present as painless bony swellings or inci-
in a fibrous stroma.1 They are slow-growing tumours dental findings on radiological examination, and as they are
with an affinity for bone, not soft tissue, and they have difficult to recognise in the early stages, they are often rel-
atively large at diagnosis and can fill the ramus or body of
the mandible. Radiological appearances can be unilocular,
∗ Corresponding author at: Prof of Oral and Maxillofacial Surgery, Uni-
multilobular, or multilocular.4
versity College London, Gower St, London, WC1E 6BT.
E-mail addresses: J.Haq@nhs.net (J. Haq), sarah.siddiqui@kcl.ac.uk
Histologically, there are four patterns: solid multicystic,
(S. Siddiqui), mark.mcgurk@btinternet.com (M. McGurk). extraosseous peripheral, desmoplastic, and unicystic.1 The

http://dx.doi.org/10.1016/j.bjoms.2016.07.017
0266-4356/© 2016 Published by Elsevier Ltd on behalf of The British Association of Oral and Maxillofacial Surgeons.

Please cite this article in press as: Haq J, et al. Argument for the conservative management of mandibular ameloblastomas. Br J Oral
Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.bjoms.2016.07.017
YBJOM-4957; No. of Pages 5
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2 J. Haq et al. / British Journal of Oral and Maxillofacial Surgery xxx (2016) xxx–xxx

unicystic variant can be further divided into three subtypes


depending on mural involvement. Subtype 1 cysts are lined
by variable epithelium with no infiltration into the cystic wall.
Those in subtype 2 show intraluminal, plexiform, epithelial
proliferation without infiltration, and those in subtype 3 show
either a follicular or plexiform pattern of invasion by epithe-
lium into the cystic wall. 5 We question the clinical relevance
of these patterns.
Traditionally, treatment has depended on the histologi-
cal features. The solid multicystic, and subtype 3 unicystic
ameloblastomas are considered aggressive with a high poten-
tial to recur, so the approach has been to resect the lesion and
repair the defect with a microvascular graft.6–8 The unicys-
tic forms are thought less likely to recur, but although a more
conservative approach is recommended, in reality it is seldom
adopted.9–11
Evidence now suggests that the natural history of
mandibular ameloblastomas should be reviewed.12,13 Several
small case studies have reported long periods of remission and
potential cure after enucleation and application of Carnoy’s
solution.3 This technique has evolved from the management
of keratocysts, and its use in the treatment of ameloblastomas
is promising with recurrence of 10%.14
We report 27 consecutive patients with ameloblastoma of
the mandible who were treated by enucleation and applica-
tion of Carnoy’s solution. Although the study has limitations,
most notably the number of cases and duration of follow up,
we hope that it will show that there is a safe alternative to
resection of the jaw. We also hope that it will stimulate debate
and act as a catalyst for further research into the management
of these tumours.

Methods

Since 2001 at the head and neck unit at Guy’s Hospital,


we have treated patients with ameloblastoma conservatively.
We retrospectively analysed the case notes from the time Fig. 1. Radiological subclassification. (A) Unilocular: no septa, well-defined
of operation of all patients treated for ameloblastoma of margin; (B) mulitlobular: some septa, well-defined margin; (C) multilocular:
the mandible by the same surgeon. Those with maxillary multiple septa and satellite lesions, poorly-defined margin.
ameloblastomas were excluded. Each patient had clinical
and radiological evaluation by plain film or cone-beam com-
puted tomography (CT). In each case, the in-house pathology of the enucleation, we remove the teeth associated with the
team reviewed the histological examination of the biopsy lesion. On exposing the mandible we carefully remove the
specimen. Information collected included patients’ charac- overlying buccal plate to expose the full extent of the tumour
teristics, histological classification and radiological features and avoid leaving a bony overhang. We then carefully enu-
of the lesions before and after operation, operative details, cleate the tumour by creating a plane between it and the bone.
and status of the disease at the last review. The preopera- To do this we pack ribbon gauze into the space around the
tive radiological features were categorised into three groups periphery of the tumour. The remnants of the lingual plate can
(Fig. 1). be freed with a rotary instrument or chisel so that the tumour
and embedded teeth can be delivered as a unit. This approach
Technique leaves a clear bony cavity with patches of exposed perios-
teum. The cavity is carefully inspected and any lacunae are
We excise mucosal fistulas or mucosa attached to the tumour, drilled back to bleeding bone. The bony cavity is then filled
but preserve mucosa that is not attached by gently peel- twice with Carnoy’s solution (1 g of ferric chloride dissolved
ing it off the mucoperiosteal flap. In most cases, as part in 6 ml of absolute alcohol, 3 ml of chloroform, and 1 ml of

Please cite this article in press as: Haq J, et al. Argument for the conservative management of mandibular ameloblastomas. Br J Oral
Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.bjoms.2016.07.017
YBJOM-4957; No. of Pages 5
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J. Haq et al. / British Journal of Oral and Maxillofacial Surgery xxx (2016) xxx–xxx 3

glacial acetic acid), which is left in place for three minutes


each time.
We protect the adjacent soft tissues with gauze and
Vaseline® petroleum jelly (Unilever, Leatherhead, UK) then
wash out the cavity with normal saline before closing the
mucoperiosteal flap directly.

Results

Between 2001 and 2014, 31 patients were treated for


mandibular ameloblastomas. Four of them had traditional
resection of the mandible and 27 (13 male) were managed
conservatively by enucleation and application of Carnoy’s
solution. Initial and definitive histological diagnoses, radio-
logical appearance, follow-up period, and recurrence, are
shown in Table 1. In two cases, the initial diagnosis of uni-
cystic ameloblastoma was changed when the primary sample
indicated the solid multicystic variant. A total of 8 (30%)
involved the left body/ramus, 10 (37%) the right body/ramus,
and 9 (33%) involved the parasymphyseal region or crossed
the midline. The mean dimension of the lesions was 5 dental
Fig. 2. Images showing good bony infill after enucleation and sterilisation
units (range 3-13), which meant that most filled the body of
with Carnoy’s solution of a large multilobular ameloblastoma. The alveolus
the mandible or ramus. is sufficient for placement of dental implants and rehabilitation without the
Assessment of the 23 preoperative radiographs suggested need for a bone graft.
that 17 showed a unilocular pattern, 5 a multilobular scal-
loped pattern without septa, and one had aggressive features
of multilocularity and a poorly defined peripheral margin. Discussion
Radiographs were not available for four patients. The mean
period of follow up was 38 months (range 3-156). Two The recurrence rate of unicystic and solid multicystic
patients were lost to follow up, one of whom had moved to ameloblastomas ranges widely and depends on the treatment
Australia. Attempts to contact them both were unsuccessful. and histological subtype.2,11 In response to the risk of recur-
Fig. 2 shows a typical response to the current conservative rence, management has evolved over the last 5 decades from
approach. repeated enucleation to the current standard of mandibular
resection and repair by microvascular graft. It is accepted
that small unicystic lesions are less likely to recur and can be
Recurrence managed by enucleation, but in reality, as most are silent and
tend to be large when they are discovered, surgeons choose
Three patients (11%) developed recurrence, in all cases the to resect the jaw. This approach has become more common
radiological size was less than 1 cm. The first patient had as microvascular grafts can reliably repair the defect.
had enucleation of a unicystic ameloblastoma and applica- Whether mandibular ameloblastomas merit such an
tion of Carnoy’s solution in 2009 with no dental extractions, aggressive approach is questionable. This tumour affects a
and the initial radiological appearances showed a multilo- relatively young population, and resection with its atten-
bular lesion. When a 0.5 cm recurrence was noted in 2012 dant facial deformity can have a considerable impact at a
the procedure was repeated and teeth removed. The second time when patients are seeking partners and employment. We
patient had an extensive recurrent solid multicystic, multi- know of no evidence that recurrent ameloblastomas change
locular ameloblastoma that crossed the midline and filled in nature and are more difficult to treat if they recur. The
the body of the mandible bilaterally. At 35 months there tumour is of odontogenic origin and experience shows that it
was a small focus of recurrent tumour in the area around has a strong affinity for bone and rarely migrates into soft tis-
the incisors that was easily removed. The third patient had sues as the periosteum forms a barrier and stops it. If it does,
a multilobular lesion that recurred within 27 months in two then the lesion has changed its nature. Recurrent disease is
small foci. After repeat enucleation of the sold multicystic therefore contained in the mandible, normally in a discrete
ameloblastoma and sterilisation with Carnoy’s solution there focus, and is easy to manage, particularly if caught early. The
has been no subsequent re-recurrence to date. No radiological conservative approach is safe, and nothing is lost if a small
unilocular lesions have recurred irrespective of histological recurrent lesion appears in the bone. However, this is not the
subtype. case with ameloblastomas of the maxilla, as recurrent tumour

Please cite this article in press as: Haq J, et al. Argument for the conservative management of mandibular ameloblastomas. Br J Oral
Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.bjoms.2016.07.017
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Table 1
Initial and definitive histological diagnoses, radiological appearance, follow-up period, and recurrence.
Initial histological diagnosis Definitive histological diagnosis Radiological appearance Follow up (months) Recurrence
Ameloblastoma NOS Unicystic Not available 156 No (moved to Australia)
Plexiform Plexiform/follicular Not available 44 No
Plexiform/follicular Plexiform/follicular Unilocular 98 No
Ameloblastoma NOS Plexiform Not available 66 No
Ameloblastic fibrodentinoma Ameloblastic fibrodentinoma Unilocular 87 No
Ameloblastoma NOS Plexiform Unilocular 16 No
Ameloblastoma NOS Follicular Unilocular 71 No
Plexiform Plexiform Unilocular 13 (Deceased - other cause) No
- Unicystic Multilobular 54 Yes
Unicystic Solid/multicystic Unilocular 49 No
Solid/multicystic Solid/multicystic Not available Lost to follow up Unknown
Features of ameloblastoma No evidence of ameloblastoma Unilocular 38 No
Ameloblastoma NOS Solid/multicystic Multilocular 41 Yes
- Solid/multicystic Unilocular 42 No
- Solid/multicystic Multilobular 32 No
- Solid/multicystic Unilocular 27 No
Solid/multicystic Unicystic Unilocular 15 No
Ameloblastoma NOS Solid/multicystic Unilocular 15 No
Solid/multicystic Solid/multicystic Unilocular 26 No
Solid/multicystic Solid/multicystic Multilobular 21 Yes
Solid/multicystic Unicystic Unilocular 17 No
Solid/multicystic Solid/multicystic Unilocular 17 No
- Solid/multicystic Multilobular 11 No
Granular variant Solid/multicystic Multilobular 3 No
Solid/multicystic Solid/multicystic Unilocular 8 No
Solid/multicystic Solid/multicystic Unilocular 9 No
Solid/multicystic Solid/multicystic Unilocular 10 No

NOS: Not Otherwise Specified.

can escape into the labyrinth of bony passages in the ethmoid The conservative approach challenges a number of
and reappear in the base of the skull. Our study is therefore accepted precepts. As the preoperative histological classifi-
restricted to mandibular lesions. cation is not reliable, management must be influenced by the
Between 1940 and 1970, ameloblastomas were treated radiological appearance. Size alone is not a defining factor,
by enucleation because the ability to reconstruct a resected instead it is tumour that is pushing forwards rather than one
mandible was limited.15–22 The papers we studied did not that is progressing rapidly. Large, scalloped lesions are ideal
suggest that the tumours threatened life but they acknowl- for conservative management, but multiple, small locules can
edged that recurrence was treated by further enucleation. The be difficult to enucleate. We did not consider the histologi-
current use of wide excision with resection margins of about cal description of the tumour when planning treatment and it
1 cm has arisen because surgeons have sought to achieve seems to have had little impact on outcome.
zero recurrence, and not because the tumour is a threat to Stoelinga reported a reduction in the recurrence of ker-
life. atocysts from 60% to less than 10% after the use of
The histological subtype of the tumour guides contem- Carnoy’s solution,23 and the solution was also beneficial
porary management, and the treatment of solid multicystic when used to treat ameloblastomas.24 Our findings support
lesions is more aggressive. In practice, however, to obtain this view. Conservative management does not eliminate the
a clear margin of 1 cm there is a low threshold for seg- risk of recurrence, whereas in most cases, resection does, but
mental mandibular resection of large ameloblastomas, and resection can have a considerable impact on young people,
consequently, the histological subtype has little impact on and to our knowledge this has not been evaluated. We have
management. There is also another diagnostic dimension to found that little is lost if a small number of patients develop
consider. It can sometimes be difficult to obtain a represen- recurrence. If kept under review the lesions are single, dis-
tative biopsy specimen so a preoperative classification of crete, and easy to eliminate by a second enucleation, and even
ameloblastoma is not reliable. In our series, 4 (15%) initial if resection is ultimately required, it can be postponed until
preoperative classifications were altered when the main spec- later in life when the patient is settled.
imen was evaluated, and one diagnosis of ameloblastoma was This paper has some important shortcomings. Despite the
completely revised to a dental cyst. If these patients had had number of cases being large for a single surgeon, the overall
their jaws resected, the situation would have been far more number is small. The duration of follow up is inadequate,
serious. and ideally should be at least 10 years, almost a surgical

Please cite this article in press as: Haq J, et al. Argument for the conservative management of mandibular ameloblastomas. Br J Oral
Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.bjoms.2016.07.017
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J. Haq et al. / British Journal of Oral and Maxillofacial Surgery xxx (2016) xxx–xxx 5

lifetime. A prospective randomised controlled trial is needed, 7. Carlson ER, Marx RE. The ameloblastoma: primary, curative surgical
but the cost of such a long-term study is prohibitive, and management. J Oral Maxillofac Surg 2006;64:484–94.
8. Bianchi B, Ferri A, Ferrari S, et al. Mandibular resection and reconstruc-
funding bodies will not consider paying for research into a
tion in the management of extensive ameloblastoma. J Oral Maxillofac
condition that has an incidence of one in a million. The only Surg 2013;71:528–37.
practical way forward is to develop national guidelines so 9. Gardner DG. Some current concepts on the pathology of ameloblas-
that treatment is uniform, and to establish a central database tomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1996;82:
on which to record patients and their treatment. The evidence 660–9.
10. Ghandhi D, Ayoub AF, Pogrel MA, MacDonald G, Brocklebank LM,
will then show which treatment is most appropriate.
Moos KF. Ameloblastoma: a surgeon’s dilemma. J Oral Maxillofac Surg
We hope that this article will stimulate debate and pro- 2006;64:1010–4.
mote further interest in the management of mandibular 11. Nakamura N, Higuchi Y, Mitsuyasu T, Sandra F, Ohishi M. Compari-
ameloblastoma. We also hope that the gradual accumulation son of long-term results between different approaches to ameloblastoma.
of experience will result in treatment that is more conserva- Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;93:13–20.
12. Lau SL, Samman N. Recurrence related to treatment modalities of uni-
tive.
cystic ameloblastoma: a systematic review. Int J Oral Maxillofac Surg
2006;35:681–90.
13. Rajeshkumar BP, Rai KK, Geetha NT, Shivakumar HR, Upasi A.
Conflict of Interest Carnoy’s in aggressive lesions: our experience. J Maxillofac Oral Surg
2013;12:42–7.
14. Lee PK, Samman N, Ng IO. Unicystic ameloblastoma—use of Carnoy’s
We have no conflicts of interest
solution after enucleation. Int J Oral Maxillofac Surg 2004;33:263–7.
15. Small IA, Waldron CA. Ameloblastomas of the jaws. Oral Surg Oral
Med Oral Pathol 1955;8:281–97.
Ethics Statement/confirmation of patient permission 16. Gardner AF, Apter MB, Axelrod JH. A study of twenty-one instances of
ameloblastoma, a tumor of odontogenic origin. Oral Surg Anesth Hosp
Dent Serv 1963;21:230–7.
There is no identifying information in this submission
17. Masson JK, McDonald JR, Figi FA. Adamantinoma of the jaws: a clin-
icopathologic study of 100 histologically proved cases. Plast Reconstr
Surg Transplant Bull 1959;23:510–25.
References 18. Monks FT. Treatment of adamantinoma by conservative surgery: a
review. J Oral Surg Anesth Hosp Dent Serv 1964;22:171–7.
1. Gardner DG, Heikinheimo K, Shear M, Philipsen HP, Coleman H. 19. Georgiade N, Masters F, Horton C, Pickrell K. The ameloblastoma
Ameloblastomas. In: Barnes L, Eveson JW, Reichart P, Sidransky D, edit- (adamantinoma) and its surgical treatment. Plast Reconstr Surg (1946)
ors. World Health Organization classification of tumors. Pathology and 1955;15:6–14.
genetics: head and neck tumours. Lyon: IARC Press; 2005. p. 296–300. 20. Goldwyn R, Constable J, Murray JE. Ameloblastoma of the jaw. A clin-
2. Pogrel MA, Montes DM. Is there a role for enucleation in the management ical study. N Engl J Med 1963;269:126–9.
of ameloblastoma? Int J Oral Maxillofac Surg 2009;38:807–12. 21. Gorlin RJ. The pathology of ameloblastomas and its relationship to treat-
3. Reichart PA, Philipsen HP, Sonner S, Ameloblastoma:. biological profile ment. Trans Int Conf Oral Surg 1970:230–53.
of 3677 cases. Eur J Cancer B Oral Oncol 1995;31B:86–99. 22. Stout RA, Lynch JB, Lewis SR. The conservative approach to ameloblas-
4. Chapelle KA, Stoelinga PJ, de Wilde PC, Brouns JJ, Voorsmit RA. tomas of the mandible. Plast Reconstr Surg 1963;31:554–62.
Rational approach to diagnosis and treatment of ameloblastomas and 23. Stoelinga PJ. Long-term follow-up on keratocysts treated according to a
odontogenic keratocysts. Br J Oral Maxillofac Surg 2004;42:381–90. defined protocol. Int J Oral Maxillofac Surg 2001;30:14–25.
5. Ackermann GL, Altini M, Shear M. The unicystic ameloblastoma: a 24. Stoelinga PJ, Bronkhorst FB. The incidence, multiple presentation and
clinicopathological study of 57 cases. J Oral Pathol 1988;17:541–6. recurrence of aggressive cysts of the jaws. J Craniomaxillofac Surg
6. Shatkin S, Hoffmeister FS, Ameloblastoma:. a rational approach to ther- 1988;16:184–95.
apy. Oral Surg Oral Med Oral Pathol 1965;20:421–35.

Please cite this article in press as: Haq J, et al. Argument for the conservative management of mandibular ameloblastomas. Br J Oral
Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.bjoms.2016.07.017

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