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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
CHU de Lille, Hôpital Roger-Salengro, Oral and Maxillofacial Surgery Department, 59000 Lille, France
Université Lille Nord de France, UDSL, 59000 Lille, France
Centre Oscar-Labret, Head and Neck Department, 59000 Lille, France
Inserm U 1008, Controlled Drug Delivery Systems and Biomaterials, 59000 Lille, France
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.
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 cosmetic and functional sequelae and may require free flap recon-
struction [5,6]. The choice between these two treatment modalities
Ameloblastoma is a rare, benign, slowly-growing odontogenic therefore appears to be an essential issue in the management of
tumour. It accounts for 1% of all tumours of the jaws and 11% of all these tumours.
odontogenic tumours [1], tumours arising from epithelial and/or We therefore conducted a retrospective study of all ameloblas-
ectomesenchymal tissue participating in the formation of the teeth tomas of the jaws managed in our department between 1991 and
[2]. According to the 2005 World Health Organisation (WHO) histo- 2013. The clinical, radiological, and histological characteristics of
logical classification of head and neck tumours, ameloblastoma can these tumours and the type of treatment were recorded. The pri-
be classified into 4 subtypes: solid/multicystic (follicular or plexi- mary objective of this study was to describe the recurrence rate of
form), extraosseous/peripheral, desmoplastic and unicystic [2]. these ameloblastomas as a function of the type of treatment.
Ameloblastomas are characterised by an aggressive potential for
local invasion and a high recurrence rate, requiring a precise histo- 2. Patients and methods
logical diagnosis and surgical treatment, the modalities of which
have not yet been clearly defined. Although conservative treat- All patients admitted to the Lille Hospital Oral and Maxillofacial
ment (marsupialization, enucleation, curettage) preserves integrity Surgery Department for ameloblastoma of the jaws between 1991
of the bones and allows continued growth of the mandible [3], and 2013 were retrospectively included. Patients with missing data
it appears to be associated with a high recurrence rate, ranging were excluded from the study.
between 55% and 90% [4], while radical treatment can leave major 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: (A. Laborde). of the lips and chin, loose teeth, mucosal fistula;
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),
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2 A. Laborde et al. / European Annals of Otorhinolaryngology, Head and Neck diseases xxx (2016) xxx–xxx

Fig. 1. Flow chart.

• tumour site: mandible or maxilla. The mandibular sector was • duration of follow-up.
divided into 5 regions:
◦ incisor-canine sector, These data were analysed by xlstat software. A descriptive anal-
◦ premolar-molar sector, ysis of the population was initially performed. Two groups were
◦ posterior sector (from the third molar the ramus of the then distinguished according to the type of treatment in order to
mandible), compare recurrence rates.
◦ large anterior sector (overlapping incisor-canine and premolar- Fisher’s exact test was used to compare recurrence rates
molar sectors), between the radical treatment group and the conservative treat-
◦ and large posterior sector (overlapping premolar-molar and ment group with a limit of significance of P < 0.05.
posterior sectors);
• diagnostic chronology: preoperative or postoperative; 3. Results
• radiographic presentation: unilobed defect, multilobed defect,
cortical bone invasion, root resorption, impacted tooth, soft tissue A total of 31 patients were included in the study and 27 were
infiltration, fracture; included in statistical analysis. Four patients were excluded due to
• histological type: follicular, plexiform, mixed (follicular and missing medical data (Fig. 1).
plexiform), desmoplastic, unicystic, peripheral, acanthomatous,
granular; 3.1. Description of the population
• first-line, second-line and third-line treatments: conservative
(marsupialization, enucleation and curettage of the macroscopic Descriptive characteristics of the population are summarized in
lesion, simple bone resection) or radical (maxillectomy, segmen- Table 1.
tal mandibulectomy); The population presented a male predominance: 17 males
• number of recurrences;
(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),
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ANORL-598; No. of Pages 5 ARTICLE IN PRESS
A. Laborde et al. / European Annals of Otorhinolaryngology, Head and Neck diseases xxx (2016) xxx–xxx 3

Table 1
Description of the population.

Sample size %

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
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.
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 The problem of recurrence starts at the time of diagnosis: either
was the mandible: 20 patients (74.1%) (Fig. 2). The most common a biopsy has been performed prior to surgery to confirm the diagno-
histological types were follicular (29.6%) and plexiform (22.2%) sis and determine the histological type in order to propose radical
(Table 1). The mean duration of follow-up was 56.2 months with a or conservative treatment, or conservative biopsy resection is per-
standard deviation of 44.2 months. formed, with no possibility of reliable frozen section examination,
and the indication for radical treatment is then decided on the basis
3.2. Comparison of the two groups according to the type of of the histological results or after a surveillance period.
treatment Several studies have reported a higher recurrence rate after con-
servative treatment compared to radical treatment [8–14]. Data of
A total of 36 events were observed in 27 patients: 22 events in the literature are summarized in Table 3. The results of our study are
the conservative treatment (CT) group and 14 events in the radical
treatment (RT) group, as 8 patients required a second treatment for Table 3
recurrence and 2 patients required a third treatment (Fig. 1). The 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),
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4 A. Laborde et al. / European Annals of Otorhinolaryngology, Head and Neck diseases xxx (2016) xxx–xxx

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

Please cite this article in press as: Laborde A, et al. Ameloblastoma of the jaws: Management and recurrence rate. European Annals of
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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),