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European Annals of Otorhinolaryngology, Head and Neck diseases xxx (2016) xxx–xxx

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Original article

Ameloblastoma of the jaws: Management and recurrence rate

A. Laborde a,b,∗ , R. Nicot a,b , T. Wojcik c,d,e , J. Ferri a,b,d,e , G. Raoul a,b,d,e
a
CHU de Lille, Hôpital Roger-Salengro, Oral and Maxillofacial Surgery Department, 59000 Lille, France
b
Université Lille Nord de France, UDSL, 59000 Lille, France
c
Centre Oscar-Labret, Head and Neck Department, 59000 Lille, France
d
Inserm U 1008, Controlled Drug Delivery Systems and Biomaterials, 59000 Lille, France
e
International Association of Oral and Maxillofacial Medecine (IAOMM), 7, bis rue de la créativité, 59650 Villeneuve d’Asq, France

a r t i c l e i n f o a b s t r a c t

Keywords: Introduction: Ameloblastoma is a rare, benign odontogenic tumour associated with a high recurrence rate.
Ameloblastoma It accounts for 1% of all tumours of the jaws. The purpose of this study was to compare the ameloblastoma
Odontogenic tumours recurrence rate according to the type of treatment: radical or conservative.
Recurrence
Patients and methods: All patients with a diagnosis of ameloblastoma between 1991 and 2013 were
retrospectively identified in order to extract topographic, radiological, and histological data and the type
of treatment: conservative (marsupialization, enucleation, curettage) or radical (segmental resection)
and to compare the recurrence rate according to the type of treatment.
Results: Twenty-seven patients were included, managed by conservative treatment (CT) in 22 cases and
radical treatment (RT) in 14 cases. The recurrence rate was 90.9% in the CT group and 9.1% in the RT group
(P = 0.025) with a mean follow-up of 56.2 months.
Discussion: The recurrence rate after conservative treatment was higher than that after radical treatment.
These results are similar to those reported in the literature. The choice of treatment must be adapted to
the macroscopic and histological characteristics of each tumour and to the patient.
© 2016 Published by Elsevier Masson SAS.

1. Introduction
Ameloblastoma is a rare, benign, slowly-growing odontogenic
tumour. It accounts for 1% of all tumours of the jaws and 11% of all
odontogenic tumours [1], tumours arising from epithelial and/or
ectomesenchymal tissue participating in the formation of the teeth
[2]. According to the 2005 World Health Organisation (WHO) histo-
logical classification of head and neck tumours, ameloblastoma can
be classified into 4 subtypes: solid/multicystic (follicular or plexi-
form), extraosseous/peripheral, desmoplastic and unicystic [2].
Ameloblastomas are characterised by an aggressive potential for
local invasion and a high recurrence rate, requiring a precise histo-
logical diagnosis and surgical treatment, the modalities of which
have not yet been clearly defined. Although conservative treat-
ment (marsupialization, enucleation, curettage) preserves integrity
of the bones and allows continued growth of the mandible [3],
it appears to be associated with a high recurrence rate, ranging
between 55% and 90% [4], while radical treatment can leave major
cosmetic and functional sequelae and may require free flap recon-
struction [5,6]. The choice between these two treatment modalities
therefore appears to be an essential issue in the management of
these tumours.
We therefore conducted a retrospective study of all ameloblas-
tomas of the jaws managed in our department between 1991 and
2013. The clinical, radiological, and histological characteristics of
these tumours and the type of treatment were recorded. The pri-
mary objective of this study was to describe the recurrence rate of
these ameloblastomas as a function of the type of treatment.
2. Patients and methods
All patients admitted to the Lille Hospital Oral and Maxillofacial
Surgery Department for ameloblastoma of the jaws between 1991
and 2013 were retrospectively included. Patients with missing data
were excluded from the study.
The following data were extracted from review of each patient’s
medical charts:

• clinical setting: age at the time of the first visit, gender;

∗ Corresponding author at: CHU de Lille, Hôpital Roger-Salengro, Oral and Max-
• circumstances of the diagnosis: local pain, swelling, sensory loss
illofacial Surgery Department, 59000 Lille, France.
E-mail address: amelyla@hotmail.com (A. Laborde). of the lips and chin, loose teeth, mucosal fistula;

http://dx.doi.org/10.1016/j.anorl.2016.09.004
1879-7296/© 2016 Published by Elsevier Masson SAS.

Please cite this article in press as: Laborde A, et al. Ameloblastoma of the jaws: Management and recurrence rate. European Annals of
Otorhinolaryngology, Head and Neck diseases (2016), http://dx.doi.org/10.1016/j.anorl.2016.09.004
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Fig. 1. Flow chart.

• tumour site: mandible or maxilla. The mandibular sector was
divided into 5 regions:
◦ incisor-canine sector,
◦ premolar-molar sector,
◦ posterior sector (from the third molar the ramus of the
mandible),
◦ large anterior sector (overlapping incisor-canine and premolar-
molar sectors),
◦ and large posterior sector (overlapping premolar-molar and
posterior sectors);
• diagnostic chronology: preoperative or postoperative;
• radiographic presentation: unilobed defect, multilobed defect,
cortical bone invasion, root resorption, impacted tooth, soft tissue
infiltration, fracture;
• histological type: follicular, plexiform, mixed (follicular and
plexiform), desmoplastic, unicystic, peripheral, acanthomatous,
granular;
• first-line, second-line and third-line treatments: conservative
(marsupialization, enucleation and curettage of the macroscopic
lesion, simple bone resection) or radical (maxillectomy, segmen-
tal mandibulectomy);
• number of recurrences;
• duration of follow-up.
These data were analysed by xlstat software. A descriptive anal-
ysis of the population was initially performed. Two groups were
then distinguished according to the type of treatment in order to
compare recurrence rates.
Fisher’s exact test was used to compare recurrence rates
between the radical treatment group and the conservative treat-
ment group with a limit of significance of P < 0.05.
3. Results
A total of 31 patients were included in the study and 27 were
included in statistical analysis. Four patients were excluded due to
missing medical data (Fig. 1).
3.1. Description of the population
Descriptive characteristics of the population are summarized in
Table 1.
The population presented a male predominance: 17 males
(59.3%). The mean age of the patients was 46.3 years with a

Please cite this article in press as: Laborde A, et al. Ameloblastoma of the jaws: Management and recurrence rate. European Annals of
Otorhinolaryngology, Head and Neck diseases (2016), http://dx.doi.org/10.1016/j.anorl.2016.09.004
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Table 1
Description of the population.

Sample size %

Gender
Male 16 59.3
Female 11 40.7
Total 27

Initial clinical characteristics

Pain 12 44.4
Swelling 18 66.6
Sensory loss of the lips and mandible 5 18.5
Loose teeth 6 22.2
Fistula 6 22.2

Tumour site
Maxilla 7 25.9
Mandible
Incisor-canine (1) 1 3.7 Fig. 2. Panoramic radiograph showing a multicystic ameloblastoma.
Premolar-molar (2) 5 18.5
Retromolar (3) 7 25.9
Overlapping (1) + (2) 2 7.4 recurrence rate was significantly higher (90.9%) in the conserva-
Overlapping (2) + (3) 5 18.5 tive treatment group than in the radical treatment group (9.1%)
(P = 0.025) (Table 2). Only one case of recurrence was observed
Radiographic characteristics
Presentation in the radical treatment (RT) group, in a patient treated by right
Unicystic 15 55.5 posterior marginal mandibulectomy. The two groups of patients
Multicystic 10 37 presented similar demographic characteristics.
Other
Root resorption 9 33.3
Bone invasion 22 81.5 4. Discussion
Soft tissue thickening 6 22.2

Diagnosis of ameloblastoma In this study, the recurrence rate after conservative treatment
Preoperative 14 51.8 was higher than that after radical treatment. The therapeutic man-
Based on histology 13 48 agement of ameloblastomas is a complex issue, as it must be as
Histological type minimally destructive as possible due to the benign nature of this
Follicular 8 29.6 lesion, but must be sufficiently extensive to prevent subsequent
Cystic 3 11.1 recurrence.
Folliculocystic 2 7.4
Two alternative approaches are therefore proposed at the
Fibroblastic 0
Acanthomatous 2 7.4
present time. The conservative approach can consist of enucleation
Basaloid 1 3.7 or curettage, sometimes preceded by marsupialization. Enucleation
Granular 1 3.7 has been precisely defined as dissection of an intraosseous cavity,
Plexiform 6 22.2 while preserving its integrity [7]. When this definition cannot be
Follicular and plexiform 0
completely observed, curettage must be associated, consisting of
Folliculocystic and plexiform 1 3.7
Desmoplastic 0 revision of the residual cavity using a drill (sphere or resin) or bone
Unicystic 0 curette. The radical approach consists of segmental bone resection.
Undetermined 3 11.1

4.1. Recurrence rate

standard deviation of 17.4 years. The main presenting complaint
was swelling, in 18 patients (66.6%). The predominant tumour site
was the mandible: 20 patients (74.1%) (Fig. 2). The most common
histological types were follicular (29.6%) and plexiform (22.2%)
(Table 1). The mean duration of follow-up was 56.2 months with a
standard deviation of 44.2 months.
3.2. Comparison of the two groups according to the type of
treatment
A total of 36 events were observed in 27 patients: 22 events in
the conservative treatment (CT) group and 14 events in the radical
treatment (RT) group, as 8 patients required a second treatment for
recurrence and 2 patients required a third treatment (Fig. 1). The
The problem of recurrence starts at the time of diagnosis: either
a biopsy has been performed prior to surgery to confirm the diagno-
sis and determine the histological type in order to propose radical
or conservative treatment, or conservative biopsy resection is per-
formed, with no possibility of reliable frozen section examination,
and the indication for radical treatment is then decided on the basis
of the histological results or after a surveillance period.
Several studies have reported a higher recurrence rate after con-
servative treatment compared to radical treatment [8–14]. Data of
the literature are summarized in Table 3. The results of our study are
Table 3
Recurrence rates after conservative treatment (CT) and radical treatment (RT)
reported in the literature.

Table 2 References CT RT n Year

Recurrence rate of ameloblastomas as a function of conservative or radical treat- Sehdev et al. [8] 93% 13% 81 1974
ment, based on 36 events. Müller and Slootweg [9] 58.00% 15.00% 56 1985
Conservative treatment Radical treatment P Ueno et al. [10] 45.60% 8.70% 91 1989
Olaitan et al. [11] 40.00% 8.30% 93 1993
Recurrence 10 (90.9%) 1 (9.1%) Nakamura et al. [12] 33.30% 7.10% 78 2002
No recurrence 12 (48%) 13 (52%) Hong et al. [13] 29.30% 11.6 and 4.5% 23 2007
Total 22 (61.2%) 14 (38.8%) 0.025a 9
a Hertog and van der Waal [14] 53.00% 0.00% 20 2009
Fisher’s exact test.

Please cite this article in press as: Laborde A, et al. Ameloblastoma of the jaws: Management and recurrence rate. European Annals of
Otorhinolaryngology, Head and Neck diseases (2016), http://dx.doi.org/10.1016/j.anorl.2016.09.004
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therefore consistent with previously published results, as a review
of the literature reveals a higher recurrence rate after conservative
treatment (range: 29.3–93%) than after radical treatment (range:
0–21%) (Table 3). Apart from the study by Hong et al. in 2007 [13],
which demonstrated a significant difference (P = 0.004) based on
direct comparison of the 2 groups, a review of the literature failed
to demonstrate a statistically significant difference between con-
servative treatment and radical treatment. Our study confirms this
tendency by reporting a higher recurrence rate in the conservative
treatment group than in the radical treatment group.
However, this study is limited by its small sample size and the
results must therefore be interpreted cautiously. This type of prob-
lem is frequently encountered in the case of rare diseases such as
ameloblastoma.
4.2. Radical or conservative treatment?
However, several studies nevertheless recommend conser-
vative treatment depending on the macroscopic appearance of
the ameloblastoma. Gardner et al. [4] recommended conserva-
tive treatment for unicystic forms, and radical treatment for
solid/multicystic forms. Similarly, Reichart et al. [15] distinguished
unicystic forms from the other forms of ameloblastoma and rec-
ommended conservative treatment for this group of tumours.
The histological type also appears to be an important factor
determining the potential for recurrence of ameloblastomas. Hong
et al., in a series of 239 patients published in 2007 [13], reported a
higher recurrence rate for follicular tumours (25%) with no cases of
recurrence for extraosseous/peripheral and desmoplastic tumours.
Ruhin-Poncet et al. [16] also compared recurrence rates accord-
ing to histological type in a series of 109 patients: 5 patients
with a follicular form presented one recurrence and 16 patients
presented two recurrences. Nakamura et al. [12], in a series of
78 cases, reported a higher recurrence rate for follicular (26.3%),
plexiform (21.7%) and mixed (follicular and plexiform) (33.3%)
tumours, regardless of the type of treatment.
These various studies led to the new WHO classification in 2005
[2], which distinguishes various macroscopic types of ameloblas-
tomas: solid/multicystic, extraosseous/peripheral, desmoplastic,
unicystic, allowing the most appropriate treatment to be proposed
for the most common histological types.
According to the WHO histological classification,
extraosseous/peripheral and unicystic tumours have a better
prognosis than solid/multicystic and desmoplastic tumours and
could be suitable for conservative management consisting of
simple enucleation. The WHO emphasizes the need for negative
resection margins for solid/multicystic and desmoplastic tumours.
Due to its retrospective design, this study comprise a case-mix
of all macroscopic and histological forms of ameloblastoma. The
results of this study must therefore be interpreted cautiously.
4.3. Particular case of patients under the age of 18 years
The most appropriate treatment for young, growing patients
remains controversial. Some studies have tried to address this
issue. Takahashi et al. [3] studied a population of 27 patients with
a mean age of 12.3 years, in whom they recommended conser-
vative treatment because of an often less aggressive histological
type (plexiform), in order to allow continued mandibular growth
and consequently limit the major cosmetic and functional sequelae
observed at this age. Secondary resection in the event of recurrence
proved to be effective and allowed less mutilating treatment [17].
Please cite this article in press as: Laborde A, et al. Ameloblastoma of the jaws: Management and recurrence rate. European Annals of
Otorhinolaryngology, Head and Neck diseases (2016), http://dx.doi.org/10.1016/j.anorl.2016.09.004
4.4. Systemic dissemination and malignancy
Surgical resection is considered to be the only effective treat-
ment for ameloblastoma. In their review of the literature, Carlson
et al. [18] reported the radioresistance and chemoresistance of
ameloblastomas. Radiotherapy can also be responsible for second
tumours, such as sarcomas [19]. However, some studies, such as
that by Sauk et al. [20], have tried to develop targeted chemother-
apy for sonic hedgehog (SHH)-dependent tumours in order to
block signals responsible for systemic dissemination of certain
ameloblastomas.
According to Dissanayake et al. [21], the recommended treat-
ment for metastatic ameloblastoma associated with cervical lymph
nodes comprises cervical lymph node dissection, while the addition
of chemotherapy and radiotherapy protocols did not provide any
conclusive responses in this study.
Kurppa et al. [22] reported the presence of a BRAF V600E muta-
tion in 63% of cases of ameloblastoma in their study. This mutation
is responsible for resistance of ameloblastomas to anti-epidermal
growth factor receptor (EGFR) targeted therapies. These studies
could open up new therapeutic perspectives based on the use of
targeted therapies in selected patients.
The predominant site of metastasis of ameloblastoma is the lung
[23], for which curative treatment consists of surgery. Slootweg
et al. [24] also highlighted the possibility of lung metastases in
patients with multiple recurrences. We have even observed a case
of bilateral lung metastases in a patient with malignant transforma-
tion into ameloblastic carcinoma. This adult patient presented with
follicular ameloblastoma of the maxilla treated by partial maxillec-
tomy, with recurrence 4 years after initial management.
Finally, some ameloblastomas can be fatal due to the anatomi-
cal site. Nastri et al., in a series of 13 ameloblastomas of the jaws
published in 1995 [25], reported 3 cases of local cerebral invasion
resulting in the death of 2 patients. Posterior mandibular or maxil-
lary tumour sites require larger resection margins than tumours of
the mandibular symphysis due to the higher risk of local invasion
and more difficult redo surgery.
4.5. Resection margins and reconstruction
While radical treatment has been clearly established as first-
line treatment for follicular and non-unicystic forms, the issue of
resection margins has not been clearly resolved. Hong et al. [13]
did not observe any statistically significant difference between
resection with wide margins and segmental resection. In contrast,
based on a review of the literature, Carlson et al. [18] recommended
1 to 1.5 cm resection margins.
When segmental resection is envisaged for a large tumour,
immediate free flap reconstruction [5] should be considered. Vay-
vada et al. [5] reported no recurrence in a series of 11 patients
undergoing free flap reconstruction with a mean follow-up of
29.3 months. Chaine et al. [6] reported similar results in a series
of 44 patients with a mean follow-up of 53 months. We have per-
sonally treated 7 patients by first-line or second-line segmental
mandibulectomy in this series and have not observed any case of
recurrence.
5. Conclusion
The results of this study appear to show a higher recurrence
rate in the group treated conservatively, confirming the results of
most of the published studies on this subject. However, conserva-
tive treatment still plays a major role in the management of certain
forms of ameloblastoma. The macroscopic type, histological type,
and the patient’s age and medical history are major determinants
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in the choice of treatment. Radical surgery is now facilitated by
reconstruction techniques that improve functional and cosmetic
rehabilitation of the oral cavity, justifying radical treatment accord-
ing to clearly defined criteria.
Disclosure of interest
The authors declare that they have no competing interest.
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