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British Journal of Oral and Maxillofacial Surgery 48 (2010) 549554

Ameloblastoma in children and adolescents

Jing Zhang a,1 , Zexu Gu a,1 , Lin Jiang a , Jinlong Zhao b , Meiyu Tian a , Jun Zhou c, ,
Yinzhong Duan a,
a Department of Orthodontics, School of Stomatology, The Fourth Military Medical University, 145 West Changle Road, Xian, PR China
b Department of Oral and Maxillofacial Surgery, School of Stomatology, The Fourth Military Medical University,
145 West Changle Road, Xian, PR China
c Department of Oral Pathology, School of Stomatology, The Fourth Military

Medical University, 145 West Changle Road, Xian, PR China

Accepted 26 August 2009

Available online 24 September 2009

Abstract

This study first reviewed the data of 37 patients aged 18 years and younger with ameloblastoma over a 16-year period and then reviewed
the literature on this subject from 1970 to 2009. Of 37 patients with ameloblastoma, 23 were male and 14 were female, a ratio of 1.6:1.
The mean age was 14.8 years. All lesions were in the mandible. Clinical typing included 28 solid type and 9 unicystic type. Ten cases were
recurrent (27.0%). A series of literature review disclosed 233 well-documented cases of ameloblastoma in children and adolescents. The ages
ranged from 4 to 20 years with a mean age of 14.5 years. The distribution among males and females were almost identical: 53.6% (125/233)
males and 46.4% (108/233) females (1.16:1). The mandible was affected in 225 (96.6%), the maxilla in 8 (3.4%). Histologically, solid type
(63.1%) predominated over unicystic type (36.9%). Of 226, 123 (54.4%) patients were treated with radical resection, 103 (45.6%) underwent
conservative method. Owing to a high recurrent rate of ameloblastoma, solid type of tumors should be approached with radical surgical
treatment, while conservative measure can be applied selectively to unicystic type. Long-term follow-up is important because recurrence may
appear years after tumor removal.
2009 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: Ameloblastoma; Children and adolescent; Clinical features; Management

Introduction ous large cases series of ameloblastoma have been reported,

Ameloblastoma is the most common benign odontogenic
tumor accounting for approximately 1% of tumors and cysts
of the jaw and 10% of odontogenic tumors.1,2 According to
data in the literature, the disease occurs in all age groups
but the peak incidence is in the third and fourth decades.36
The tumor in young people is considered a rarity and it
account for approximately 1015% of all reported cases of
ameloblastoma.7,8 A review of literature revealed that numer-
while only a few articles discussed this tumor in children and
adolescents and few had a sizeable number of patients in their
series. The purpose of this study is to review clinical features
and management of ameloblastoma in 37 patients no more
than 18 years of age and summarize a series of previously rel-
evant reports. We hope this study may contribute to further
meta-analysis, increase cognition of the lesion, and develop
a rational treatment in children and adolescents.

Materials and methods

Corresponding author. Tel.: +86 29 84776151.

The material was collected from the medical records of all

Corresponding author. Tel.: +86 29 84776131; fax: +86 029 84776131.
E-mail addresses: jingz77@163.com (J. Zhang),
yonghe@fmmu.edu.cn (J. Zhou), duanyz@fmmu.edu.cn (Y. Duan).
1 Both the authors contributed equally to this work.
children and adolescents 18 years of age with histopatho-
logical diagnosis of ameloblastoma at the Department of
Oral Pathology, School of Stomatology, The Fourth Military

0266-4356/$ see front matter 2009 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2009.08.020
550 J. Zhang et al. / British Journal of Oral and Maxillofacial Surgery 48 (2010) 549554

Medical University, China, between January 1993 to Decem-

ber 2008. Of 267 patients with ameloblastoma, 37 patients
were included in this study. We reviewed patient age, sex,
location, radiographic appearance, clinical features, treat-
ment, recurrence and follow-up.
A literature search for all patients 20 years old or younger
with a sizeable number of ameloblastoma reported from 1970
to 2009 was undertaken. Some relevant reports which were
isolated one or two cases and reports earlier than 1970 contain
data that would now considered as histologically inaccurate
were excluded. In reported literature, some authors divided
the classic intraosseous ameloblastoma into two type (solid Fig. 2. Two years later, there was no evidence of recurrence, the lesional area
and multicystic), we described as one type in this article. was filled with normal bone and the apical of canine tooth and the second
premolar were complete.

Results

This series consisted of 23 males and 14 females, a ratio of

1.6:1. The age ranged from 5 to 18 years (mean 14.8 3.09
years) at the time of diagnosis. Twenty-eight patients had pri-
mary ameloblastomas, while 9 patients, whose primary tumor
had being treated elsewhere, developed recurrent disease.
All the lesions were located in the mandible. Lesions occu-
pied the body-angle-ramus in 19 cases (51.4%), followed by
mandibular body (8, 21.6%), symphysis (5, 13.5%), the body-
angle (4, 10.8%) and the angle-ramus in 1 case. Fourteen
(37.8%) cases were located on the left side, 18 (48.6%) on the
right side, and 5 (13.5%) cases crossed the midline. A painless
swelling was the most common symptom, experienced by 20
(54.1%) of 37 patients. Teeth mobility in the area of tumor
with a painful swelling was observed in three patients and in
seven patients the teeth mobility with a painless swelling was
noted.
All patients were managed surgically. Twenty-nine
(78.4%, 7 unicystic, 22 solid type) patients were
employed conservative surgical management (curettage, cur-
rettage + cautery or decompression, Figs. 1 and 2). Eight
patients (21.6%, 6 solid and 2 unicystic type) underwent rad-
ical resection (segmental or hemi-mandibulectomy and iliac
bone graft) and there were no local recurrences (Figs. 3 and 4).
Fig. 3. Radiograph of an 18-year-old female with multilocular radiolucent
lesion in the left mandible. The lesion was located in the mandibular canine
and angle, extending into the ramus. Tooth displacement and root resorption
were evident. Fine septa and rough border with bone ridge (arrow) were
seen.
On the basis of established histologic criteria, we observed
that 9 (24.3%) of 37 exhibited unicystic type and 28 (75.7%)
showed solid type. Of the 28 solid tumors, a follicular pattern
was identified in 18 cases (48.7%), a plexiform in 4 cases
(10.8%) and a mixed in 6 cases (16.2%). The subdivision of
unicystic ameloblastoma did not describe. The radiographic
findings were consistent with a multilocular radiolucency in
16 cases (16/37, 43.2%), whereas 21 (56.8%) were unilocular
in appearance. There were 4 unicystic type (4/9, 44%) lesions
exhibited in multilocular appearance, while 16 (16/28, 57%)
solid type lesions presented in unilocular image.
By correlating radiographic findings with histologic type,
we found that 16 (57.1%) cases of solid tumor manifested

Fig. 1. The panoramic radiograph of an 11-year-old boy demonstrated a

unilocular lesion with well-defined borders in the right mandibular body. Fig. 4. Immediate postoperative panorex following hemi-mandibllectomy
Root resorption and tooth displacement were seen. and iliac bone graft with plate fixation of the defect.
 J. Zhang et al. / British Journal of Oral and Maxillofacial Surgery 48 (2010) 549554
with unilocular radiolucent lesions, whereas minor of uni-
cystic tumors presented in multilocular radiolucent lesions
(4/16, 25%).
Postoperative follow-up period ranged from 3 months to
6 years. Of the total of 10 recurrences, 9 patients had one
recurrence, 1 with two recurrences. Histologically, nine were
solid type, one was unicystic type. Twenty-seven patients
(73%) were lost to follow-up after 2 years.
Review of the literature
Ameloblastoma was rare in the pediatric population and only
10 relevant publications were included in this study (Table 1).
A total of 233 patients (233/1470, 15.9%), 125 were male and
108 were female, a ratio of 1.16:1. The mean age was 14.5
years (range from 4 to 20 years). The mandible was the most
common site of occurrence in all races; 225 (96.6%) tumors
were encountered in the mandible and 8 (3.4%) in the maxilla,
with an overall mandible:maxilla ratio of 28.1:1.
Majority of the tumor (89.2%, 181 of 203) were found in
patients between 11 and 20 years old, only 10.8% (22 of 203)
under 11 years old. Mandibular posterior region was the most
commonly involved, followed by the symphyseal region.
Radiologically, 56.7% (120 of 212) of ameloblastomas were
multilocular and 43.4% (92 of 212) were unilocular (19 cases
were not recorded and 2 were ameloblastic carcinoma). His-
tologically, solid type occupied 63.1% (111 of 176), unicystic
type was 36.9% (65 of 176).
Seven patients had no treatment, the remaining were man-
aged surgically. Of 226, 123 (54.4%) patients were treated
with radical resection, while 103 (45.6%) patients under-
went conservative surgical management. The recurrence rate
after radical therapy was lower than conservative therapy.
The follow-up period varied from 1 month to 19 years. Most
patients were lost to follow-up within 2 years of surgery,
therefore, it was difficult to evaluate the precise recurrence
rates.
Discussion
A review of previous literature of ameloblastoma in the chil-
dren and adolescents showed wide variation in age selection,
therefore, it is difficult to evaluate the precise incidence
because different authors have defined the upper age limit
such as under 20 years, 18 years.911 The incidence of 13.9%
(37/267) of ameloblastoma in children in this study is simi-
lar the incidence of 14.6% reported by Olaitan and Adekeye
and the 12.2% found by Kahn.1,10 However, Huang et al. and
Keszler and Dominguez reported a relatively low prevalence
of ameloblastoma (6.8% and 8.7%, respectively),3,7 while
Ord et al. and Al-Khateeb and Ababneh reported high rates
(28.9% and 38.5%, respectively).11,12
In our study, the malefemale ratio was 1.6:1. This was
similar to that reported by Daramola et al. (1.7:1),9 Olaitan
551
and Adekeye (1.5:1) and Huang et al. (1.5:1),1,3 but showed
a deviation from the findings of Kahn (0.9:1) and Ord et al.
(1:1.75).10,11 Some authors also have reported equal gender
distribution.5,7,13 In literature review, the distribution among
males and females were almost identical: 53.6% males and
46.4% female, a ratio was 1.16:1. The average age was 14.8
years in our series, this finding was consistent with the data
from the literature review (14.5).
About 91.9% of ameloblastomas in our series were found
in adolescents (1120 years old) and only 8.1% were found
under 11 years old. These findings are similar to previous
reports. Arotiba et al. reported 10 (12.7%) cases,6 while Ord
et al. and Chidzonga found none in their series.11,13 These
results also consistent with only 10.8% under 11 years old in
literature review.
The site distribution in the published literature showed
that the ameloblastomas have a marked predilection for the
mandible (96.6%). This was similar to other studies. The
molar-ramus region was the most common site followed by
the symphyseal region. Painless swelling was the most com-
mon complaint of the patients in this study (54.1%).
Radiologically, it has been reported that the unilocular
ameloblastomas tend to occur in younger age groups.3,10
Our findings confirmed this tendency (56.8%). However, the
results of literature review revealed that multilocular radiolu-
cent lesions (56.7%) predominated over unilocular lesions
(43.3%).
Ameloblastomas are currently classified into three types:
(a) solid or multicystic, the most frequent form and radio-
logically characterized by multilocular aspect, (b) unicystic,
radiologically characterized by unilocular aspect and (c)
extraosseous or peripheral type. Our cases included the solid
and unicystic ameloblstomas. Some studies reported that
the unicystic ameloblastoma is the most commonly type in
children,11,14 whereas in our study the solid multicystic type
(75.7%) predominated over unicystic type (24.3%). This was
consistent with the relevant literature, the rate of solid type
(63.1%) was higher than unicystic type (36.9%). By corre-
lating radiographic findings with histologic type, we found
that 16 (57.1%) cases of solid tumor manifested with uniloc-
ular radiolucent lesions, whereas minor of unicystic tumors
presented in multilocular radiolucent lesions (4/16, 25%).
Some publications also showed similar results.3,5,10 There-
fore, when the ameloblastomas appear as unilocular lesions
radiographically, the diagnosis of solid type also should be
considered. And in order to avoid the high recurrence rates
by conservative treatment, a biopsy is recommended.
The unicystic ameloblastoma is thought to have recur-
rence potential, but to be less aggressive than the solid type.
Previous literature reported that enucleation alone may be
sufficient to eradicate the tumor completely. However, current
studies revealed that the third histologic subtype of uni-
cystic ameloblastoma (a cystic lining showing intraluminal
and intramural proliferations) was aggressive and should be
treated more radically as a solid ameloblastoma.15 There-
fore, recommend relatively conservative treatment in the
552 J. Zhang et al. / British Journal of Oral and Maxillofacial Surgery 48 (2010) 549554

Table 1
Reported series cases of ameloblastomas in children and adolescents.
Literature data Daramola et al.9 Keszler and Dominguez7 Kahn10 Chidzonga13 Olaitan and Adekeye1 Takahashi et al.5
Reported year 1975 1986 1989 1996 1996 1998
Cases 16 8 38 20 30 6
Incidence 16/70, 22.9% 8/92, 8.7% 38/311, 12% 20/117, 17.1% 30/206, 14.6% 6/27, 22%
Age (years)
Range 517 415 719 1118 NS17 815
10 3 3 2 0 NS 2
1120 13 5 36 20 NS 4
Mean 13.4 10.8 14.8 15.5 NS 12.3
Sex
Male 10 4 18 10 18 3
Female 6 4 20 10 12 3
M/F ratio 1.7:1 1:1 0.9:1 1:1 1.5:1 1:1
Location
Maxilla 1 0 0 1 0 0
Mandible 15 8 38 19 30 6
Most common site Symphyseal Molar-ramus Molar-ramus Symphyseal Body (14/30, Angle-ramus
(7/16, 43.8%) (4/8, 50%) (29/36, 80.6%) (7/19, 35.8%) 46.7%) (3/6, 50%)
Radiography
Multilocular 11 0 10 17 23 2
Unilocular 5 8 26 3 7 4
Not known 2
Treatment
Conservative 4 8 (1 lost) 29 (13 lost) 0 13 6
Recurrence NS 3 2 Nil 5
Radical resection 12 0 7 (2 lost) 20 16 0
Recurrence NS 1 NS Nil
Not treatment Nil Nil 2 Nil 1 Nil
Histologic pattern
Solid NS NS 6 20 NS 6
Unicyst NS NS 31 0 NS 0
Peripheral 1 0 0
Follow-up Lost 5m4y (1 lost) 6m19y (18 lost) Lost 6m9y 4.4y11.8y

Literature data Ord et al.11 Al-Khateeb and Ababneh12 Arotiba et al.6 Huang et al.3 Total Present study
Reported year 2002 2003 2005a 2007 19752007 2009
Cases 11 10 79 15 233 37
Incidence 11/38, 28.9% 10/26, 38.5% 79/360, 21.9% 15/223, 6.8% 233/1470, 15.9% 37/267, 13.9%
Age (years)
Range 1219 920 619 917 420 518
10 0 1 10 1 22 3
1120 11 9 69 14 181 34
Mean 15.5 16 14.7 13.7 14.5 14.4
Sex
Male 4 4 45 9 125 23
Female 7 6 34 6 108 14
M/F ratio 1:1.75 1:1.5 1.3:1 1.5:1 1.16:1 1.6:1
Location
Maxilla 1 0 4 1 8 0
Mandible 10 10 75 14 225 37
Most common site Angle-ramus Molar-ramus Posterior area Body-angle-ramus Posterior area Body-angle-ramus
(5/11, 45.5%) (5/10, 50%) (31/73, 42.6%) (10/15, 66.6%) (19/37, 51.4.%)
 J. Zhang et al. / British Journal of Oral and Maxillofacial Surgery 48 (2010) 549554 553

Table 1 (Continued)
Literature data Ord et al.11 Al-Khateeb and Arotiba et al.6 Huang et al.3 Total Present study
Ababneh12
Radiography
Multilocular NS 5 47 5 120 16
Unilocular NS 5 24 10 92 21
Not known 6 8
Treatment
Conservative 9 (1 lost) 5 18 11 103 29
Recurrence 4 Nil NS 3 10
Radical resection 2 (1 lost) 5 57 4 123 8
Recurrence Nil NS Nil Nil
Not treatment Nil Nil 4 Nil 7 Nil
Histologic pattern
Solid 3 4 65 7 111 28
Unicyst 8 6 12 8 65 9
Peripheral 0 0 0 0 1
Follow-up 5m7y 4y13y (1 lost) 1m5y (most lost) 2y17y 3m6y

NS: not stated.

a Included two ameloblastic carcinoma.

initial presentation, such as enucleation and curettage fol-
lowed by physiochemical treatment or peripheral ostectomy
in the mandible and localized resection in maxilla, and reserve
more aggressive therapy for any recurrences.
Solid ameloblastoma as a locally aggressive tumor can
lead to a high recurrence rates (7590%) following conserva-
tive treatment.9,11 The decision to do initial radical, extensive
surgery or conservative procedures treatment in children
always poses a dilemma.16 The current opinion favors radical
resection for decreasing recurrence rates.17,18 However, in the
pediatric group, management of solid ameloblastomas often
need more consider the patients age, tumor size, anatomic
location, jaw growth and if it is an initial presentation or a
recurrence. In our cases, many cases were treated by conser-
vative method owing to the patient age, and radical treatment
could result in deformity and dysfunction of the jaw that are
bound to influence both the physical and psychological devel-
opment of the child in later life. Moreover, tumor size and
unilocular appearance would also influence the decision of
surgeons. These factors could increase the risk of recurrence.
Preoperative confirmation of histological type by means of
incisional biopsy or confirmation during surgical procedures
by surgical procedures by means of frozen section would help
clinicians select more suitable treatment modalities, which
may help to reduce the recurrence rate. If radical resection has
to be performed, the immediate bone graft is recommended
in order to prevent an obvious facial deformity.
Postoperative follow-up is very important because more
than 50% recurrences occur within 5 years of treatment.3,19
Our cases also confirmed the tendency. Many reports reveal
the recurrence time is between 1 and 15 years, and 25 years is
the most common.10,17 For the most accurate recurrence rate
findings, all cases should be routinely follow-up a long time
because of the insidious biological behavior of ameloblas-
toma. However, it was a consistent problem for clinicians to
follow the patients an enough long time (i.e. 20 years). There-
fore, data of the long-term follow-up could not acquire in our
series.
Conict of interest statement
I, the undersigned author, certify that I have no financial or
personal relationships (e.g. employment, consultancies, stock
ownership, honoraria, etc.) that pose a conflict of interest in
connection with the submitted article.
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