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Journal of Cranio-Maxillo-Facial Surgery xxx (2011) 1e7

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Journal of Cranio-Maxillo-Facial Surgery


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The pattern and occurrence of ameloblastoma in adolescents treated


at a university teaching hospital, in Kenya: A 13-year study
F.M.A. Butt a, *, S.W. Guthua b, D.A. Awange b, E.A.O. Dimba b, F.G Macigo c
a

Dept of Human Anatomy, University of Nairobi, P.O. Box 25361-00603, Nairobi, Kenya
Dept of Oral & Maxillofacial Surgery, Oral Pathology and Oral Medicine, School of Dental Sciences, University of Nairobi, Kenya
c
Dept of Periodontology/Community & Preventive Dentistry, School of Dental Sciences, University of Nairobi, Kenya
b

a r t i c l e i n f o

a b s t r a c t

Article history:
Paper received 20 July 2010
Accepted 1 March 2011

Ameloblastoma presenting in the adolescent age group is rare with few studies documenting their
occurrence.
Aim: The aim of this study was to carry out an analysis of the pattern and occurrence of ameloblastoma in
those less than 20 years of age.
Materials and method: Patients from the University of Nairobi Dental teaching Hospital treated for
ameloblastoma were included in the study over a 13-year period. The study highlights the demographic,
clinic-radiographic and histologic features of benign locally aggressive lesions.
Results: A total of 127 patients were recorded of which, 27 (21.3%) were below the age of 20 years; no case
was reported below the age of 10 years. 18.5% were below the age of 14 years and 81.5% were 15e19 years
old. The gender predilection was w1:1. All of the tumours occurred in the mandible, with radiographic
features of a multilocular radiolucencies (85.2%); and a fewer unilocular lesions (14.8%). The management is in a staged-wise approach: resection and/or disarticulation with temporary reconstruction using
mandibular stainless steel or titanium plates and delayed bone grafting.
Conclusion: The occurrence of ameloblastoma can mimic an odontogenic cyst, clinicians therefore need to
be vigilant when examining adolescents so that conservative treatment is started early in order to reduce
the subsequent morbidity.
2011 European Association for Cranio-Maxillo-Facial Surgery.

Keywords:
Ameloblastoma
Adolescents
Management

1. Introduction
Ameloblastoma is an aggressive benign slow growing epithelial
odontogenic tumour and comprises of only 1% of all tumours and
cysts of the jaws (Posnick, 2000). It is the most clinically signicant
odontogenic tumour. A prospective study done in Tanzania of
odontogenic tumours reported, ameloblastoma occurring with the
highest frequency followed by odontogenic myxoma (Simon et al.,
2005).
Ameloblastoma occurs in three different clinicoradiographic
patterns, the conventional intraosseous solid/multicystic (86%),
unicystic (13%) and peripheral (1%) (Neville et al., 2002). Three
histopathological variants of unicystic are luminal, intraluminal
and mural (Ackermann et al., 1988). The common histological
patterns include the follicular and plexiform patterns as the most
common. However, less common histopathologic patterns, acanthomatous, granular cell, desmoplastic and basal cell types have also
* Corresponding author.
E-mail address: fawzia_butt@yahoo.co.uk (F.M.A. Butt).

been reported (Neville et al., 2002). The conventional solid ameloblastoma is encountered over a wider age range being commonly
reported in the 3rde5th decade of life.
The occurrence of ameloblastoma in younger age group is
considered a rarity and it accounts for approximately 10e15% of all
reported cases of ameloblastoma (Keszler and Dominduez, 1986;
Ueno et al., 1986). The tumour is even less common in children
younger than 10 years of age and relatively uncommon in the
10e19-year-old age group with no signicant gender predilection.
A higher frequency in Blacks has been reported whereas other
studies show no racial predilection. About 85% of conventional
ameloblastomas occur in the mandible, most often in the body and
angle-ascending ramus area (Neville et al., 2002). About 15% occur
in the maxilla, usually in the posterior region. The most typical
radiographic feature is that of a multilocular radiolucency (soap
bubble) appearance accompanied by resorption of the roots of
teeth. The unicystic ameloblastoma is common in younger patients
with 50% occurring in the second decade of life.
There is a paucity of information especially from East Africa and
Kenya, regarding the incidence of ameloblastomas in the young

1010-5182/$ e see front matter 2011 European Association for Cranio-Maxillo-Facial Surgery.
doi:10.1016/j.jcms.2011.03.011

Please cite this article in press as: Butt FMA, et al., The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching
hospital, in Kenya: A 13-year study, Journal of Cranio-Maxillo-Facial Surgery (2011), doi:10.1016/j.jcms.2011.03.011

F.M.A. Butt et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2011) 1e7

population of less than 20 years. These tumours cause disgurement of the face, leading to poor self-esteem and interruption in
schooling. In addition, it also causes difculty in maintaining oral
hygiene, as well as feeding leading to malnutrition. This paper
highlights the age, gender, site predilection, histopathological and
radiographic features of ameloblastoma in patients aged 10e19
years seen at the University of Nairobi (UON) Dental Hospital. The
resulting information will assist surgeons in swift and efcient
management of such tumours in the young age group before they
reach a large size.
2. Materials and method
The data for this audit was obtained from the records of all the
cases of ameloblastomas treated at the UON Dental teaching
Hospital over a period of 13 years (1996e2009). This is a centre of
excellence for Oral and Maxillofacial Surgical training in East Africa.
The majority of such cases present to the department of Oral and
Maxillofacial surgery for management from all over the country.
The inclusion criterions were patients in the adolescent age group
(between the ages of 10 and 19 years of age), histopathological
conrmation of ameloblastoma and those with complete records.
Treatment consisted of marginal resection (and disarticulation
where necessary) with subsequent reconstruction using stainless
steel or titanium plates. The records were examined by one
investigator (Butt F.M.A) and the following information sought;
demographic characteristics such as age, gender, site of lesion,
radiographic and histopathological information. Radiographic
ndings were recorded from Orthopantomograms (OPGs),
Computed Tomographic (CT)-scans and 3-D reformatted images.
Histopathology slides retrieved from the laboratory were examined
by two independent Oral pathologists. Patients with incomplete
records and those with a disputed histopathologic ndings were
excluded from the study. Post-operatively the patients are under
close up for the next 15e20 years to observe for any recurrences.

were noted to have occurred in the mandible with none in the


maxilla. Only ve (18.5%) of the 27 patients were under than 14
years of age, while 22 (81.5%) of them were between the ages of
15e19 years. None of the patients were under 10 years old. The ratio
of males to females in the 10e14 and 15e19-year-old age brackets
was w1:1 similar to that in the total population (Table 2). The
youngest patient was a 10-year-old and the oldest 19 years of age.
The most frequently affected age group of patients with ameloblastoma was 18 and 19 years (44.4%), with the modal age group
being 18 years (25.9%).
Extra-oral features noted were facial swelling with asymmetry.
Intra-orally, there was cortical expansion, mobility of teeth and
occlusal derangement (Fig. 1). 16 patients had involvement of the
body/ramus with extension into coronoid and condyle, seven had
only the body and ramus affected and the remaining four had the
symphyseal and body areas affected (Fig. 2). Common radiological
features exhibited by the majority (85.2%) were the typical soap
bubble (multilocular) appearance that is characteristic of solid
ameloblastomas and the minority (14.8%) were unilocular radiolucencies. In addition there was root resorption and tooth
displacement. In 11 (40.7%) patients, there was an associated
impacted tooth (Fig. 3). A computed tomogram (CT) formatted as an
axial image showed bucco-lingual expansion and perforation of the
lingual cortex (Fig. 4). The treatment modality consisted of

Table 1
Age range of ameloblastoma in the study population.
Age bracket (years)

Males

Females

Males females (%)

10e19
20e29
30e39
40e49
50e59
60e69
70e79
Total

14
23
12
10
0
2
1
64

13
21
15
5
5
3
1
63

27
44
27
15
5
5
2
127

(21.3)
(34.6)
(21.3)
(11.8)
(3.9)
(3.9)
(1.6)
(100)

3. Results
During the 13-year period, a total of 127 patients were treated
for ameloblastoma of the oral and maxillofacial region. The male to
female ratio was w1:1. The majority of patients (90.6%) were within
the age bracket 10e49 years. The modal age group was 20e29 years
(Table 1). 27 (21.3%) patients were under 20 years of age and of
these 14 were males and 13 females giving a male: female ratio
w1:1. The average age of presentation was 16.1 years. All the lesions

Table 2
Distribution of adolescents with ameloblastoma by age and gender.
Age (years)

Male

Female

Total (%)

10e14
15e19
Total

3
11
14

2
11
13

5 (18.5)
22 (81.5)
27

Fig. 1. (A) Clinical features: extra-oral e facial swelling (B) intra-orally e bucco-lingual expansion.

Please cite this article in press as: Butt FMA, et al., The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching
hospital, in Kenya: A 13-year study, Journal of Cranio-Maxillo-Facial Surgery (2011), doi:10.1016/j.jcms.2011.03.011

F.M.A. Butt et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2011) 1e7

resection and reconstruction using non-vascularized iliac bone


graft secured with stainless steel or titanium plates. 16 (59.3%)
patients had resection with disarticulation due to the extension of
the tumour into the coronoid and condyle (Fig. 5).
Three histopathological variants of ameloblastoma were noted
in our study; follicular (77.8%), plexiform (18.5%) and desmoplastic
(3.7%) (Fig. 6). The male to female ratio of the follicular and plexiform was w1:1; there was only one patient (female) with the
desmoplastic type (Fig. 7). Each of the subtypes exhibited some
degree of microcystic degeneration within the ameloblastic islands;
this did not dominate the overall histological characteristics.
For patients who presented with large, extensive tumours
involving the condyle, coronoid and overlying soft tissues, resection
with disarticulation was done regardless of it being unilocular or
multilocular. It is in our protocol to try and eliminate the tumour in
our rst attempt and adhering to the practice of leaving a 2 cm
safety margin of cancellous bone after resection as has been
advocated (Muller and Slootweg 1985). Reconstruction was done
immediately with a condylar prosthesis on a stainless steel or
titanium plate (Fig. 8). In cases where there was spread of the
tumour from the mandible into the soft tissues, they were sacriced to prevent the development of peripheral ameloblastoma.
Excess facial skin was contoured for cosmesis before closure. There
were two patients who developed post-operative infection which
was resolved with course of broad spectrum intravenous antibiotics. The patients who have been followed so far have not
presented with recurrence of the ameloblastoma (Fig. 9).

Fig. 2. Schematic representation of the lesions involving the mandible. All the lesions
were unilateral. (Note: shaded area represent anatomical regions involved).

4. Discussion
In 1955, Waldron and Small documented a review of 1,036
ameloblastomas in which the average patient age was 38.9 years,
with only 2.2% under 10 years old and 8.7% between 10 and 19 years
old (Small and Waldron, 1955). Reichart in his biological prole of
3677 cases reported that in the developing countries, ameloblastomas occur in younger patients (average age 27.7 years)
compared to those from industrialized countries (average
age 39.1 years). As per the racial distribution the average age of
Blacks was 28.7 years compared to 39.9 years in Caucasians and
41.2 years in Asians (Reichart et al., 1995) Keszler and Dominquez
reported that 8.7% less than 16 years of age, whereas Kahn found
12.2% of ameloblastomas were in patients younger than 19 years
(Kahn, 1989; Keszler and Dominduez, 1986). Mehlisch et al. found
12.6% in those aged less than 20 years but, only one patient was
under 10 years of age, while Sandler et al. found 15% of their
patients under 20 years old and 3% to be under 10 years old
(Mehlisch et al., 1972; Sandler et al., 1983). Patients from Asian and
African countries show a higher percentage of children with ameloblastoma to be younger than 20 years with 18.2% for Japanese
patients and 19.7% for Thai patients (Sirichitara and
Dhiravarangkura, 1984; Ueno et al., 1986). A recent study done in
China documented a series of 27 patients of which only 8.1% were
under the age of 11 years and 91.9% were between the ages of
11 and 20 years (Zhang et al., 2010). In Nigeria, 25% of children
under the age of 20 years show an increased frequency of ameloblastoma (Arotiba et al., 1997). In Zimbabwe, Chidzonga studied
117 patients over a period of 10 years who were treated for ameloblastoma and reported 17.1% were under 18 years of age, the
youngest being 11 years: the average age at presentation being
15.5 years (Chidzonga, 1996).
In our study the incidence of ameloblastoma in the 10e19 years
old relative to all cases of ameloblastoma in all age groups was
21.3%. In comparison to other studies done in Africa, it shows
a similarity to the one reported by Arotiba et al. (21.9%), and a bit
higher than the 17.1 % reported by Chidzonga (Chidzonga, 1996;
Arotiba et al., 2005). The gender predilection shows a male to
female ratio of 1:1, which is similar to that reported by Arotiba as
1.3:1. Our nding of equal gender distribution has also been
reported by other authors (Arotiba et al. 2005). Available statistical
evidence suggests that this tumour is more common in the Black
population; in our study all the patients were Blacks of African
decent (Shear and Singh, 1978; Sawyer et al., 1985).
The rarity of this tumour in the younger age group is demonstrated by our study ndings in which only 18.5% were younger
than 14 years of age with the majority being in the older group, and
the youngest patient being 10 years. This is in accordance with

Fig. 3. OPG: (A) Unilocular radioluscency with an impacted tooth, involvement of condyle and coronoid. (B) Multilocular radioluscency of the anterior mandible.

Please cite this article in press as: Butt FMA, et al., The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching
hospital, in Kenya: A 13-year study, Journal of Cranio-Maxillo-Facial Surgery (2011), doi:10.1016/j.jcms.2011.03.011

F.M.A. Butt et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2011) 1e7

Fig.4. A 3-D CT formatted and axial view shows ameloblastoma with expansion of the bony cortex, tooth impaction and perforation of the lingual cortex.

Fig. 5. Ameloblastoma having expanded the (A) buccal cortex (intra-operative) (B) and (C) resected specimen its radiograph: illustrating ameloblastoma causing expansion of the
coronoid process covered by temporalis muscle (white arrow) and the X-ray highlighting the multilocular lesion with total destruction of the condyle process (blue arrow).

Fig.6. Distribution of ameloblastoma in adolescents by histopathologic variants.

previous reports of the low occurrence of this tumour in the rst


decade of life. In our study we had two 10-year-old patients (7.4%)
which is higher than the 4.7 % reported by Arotiba et al., and
Adekeye whereas, Chidzonga reported none in his group (Adekeye,
1980; Chidzonga, 1996; Arotiba et al., 2005). The most frequently
affected age group of patients with ameloblastoma was between 18
and 19 years (44.4%), the majority being 18 years (25.9%) This is
close to the one reported in Nigeria in which the modal age was 17
years (17.5%) (Arotiba et al., 2005).
The most prevalent site of occurrence of ameloblastoma is the
mandible in all races (Potdar, 1969; Huffman and Thatcher, 1974;
Adekeye, 1980; Olaitan and Adekeye, 1997). Reichart observed
that in Blacks, ameloblastomas affected the incisor region of the
mandible signicantly more often than in the white population

(Reichart et al., 1995). The anatomic location of ameloblastoma in


our study was mainly in the posterior ramus (85.2%). The unilocular
type was less common (14.8%) mainly located in the ramus and
body of the mandible and associated with an impacted tooth.
However in Whites the unicystic subtype has been reported as the
most common in those less than 20 years of age (Robinson and
Martinez, 1977; Leider et al., 1985; Zhang et al., 2010).
Histopathologically, some studies have shown that the plexiform type of ameloblastoma is more common than the follicular
type in children and is thought to be less aggressive hence
a conservative treatment can be considered (Shear and Singh, 1978;
Sawyer et al., 1985; Matsuo and Ueno, 1991). In contrast, our ndings showed the follicular (77.8%) to be most common followed by
the plexiform (18.5%) and then the desmoplastic (3.7%) The mean
age of patients presenting with the follicular and plexiform was 16
years and there was only one patient, a 15-year-old boy, with the
desmoplastic type. These results are consistent with those of
another study done in Kenya by Vilembwa et al. who in a study of
clinicopathologic features of ameloblastoma, reported, that
patterns of the follicular and plexiform types were predominant
(Sirichitara and Dhiravarangkura, 1984; Vilembwa et al., 2008).
The management of ameloblastoma in the younger age group
can be very challenging despite using a reconstruction plate, as this
has a profound effect on the subsequent growth dynamics of the
craniofacial skeleton, dentition and soft tissues that ensues.
Conservative management for unicystic ameloblastomas involves
enucleation, curettage and use of physico-chemical methods
(dredging and carnoys solution). However, high recurrence rates of
up to 60% have been noted which is similar to the rates when solid
and multicystic ameloblastomas are enucleated (Small and
Waldron, 1955; Ueno et al., 1986; Ueno et al., 1989; Li et al., 2000;
Lau and Samman, 2006; Vilembwa et al., 2008).
In our study population, most of the ameloblastomas are at an
advanced state and these lesions present with mural invasion and

Please cite this article in press as: Butt FMA, et al., The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching
hospital, in Kenya: A 13-year study, Journal of Cranio-Maxillo-Facial Surgery (2011), doi:10.1016/j.jcms.2011.03.011

F.M.A. Butt et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2011) 1e7

Fig. 7. Microscopy of the histologic variants of ameloblastoma.

possible involvement of cancellous bone. This hardly leaves room


for conservative methods which are indicated for small sized and
unicystic tumours. Interestingly, it has been reported that ameloblastomas in patients from the developing countries are larger
compared to those from the industrialized countries with Blacks
having larger dimensions than Caucasians (Reichart et al., 1995). In
the majority of cases extensive surgery is the management of
choice in our centre. Lesions smaller in size are accessed using an
intra-oral approach. For large tumours, extending to the condyle
and coronoid, an extra-oral approach is used. In our teaching
hospital patients are encouraged to adhere to a regular long-term
follow-up in order to detect any recurrences. A staged-reconstruction method using a non-vascularized free bone graft is
preferred, whereby the primary reconstruction with a bone graft is
secured with a mandibular reconstruction plate as shown in our
studies. A one-stage reconstruction using a vascularized bone graft
is often lengthy and costly for our patients. In addition to
challenging socioeconomic conditions, difculties arise in close
follow-up, given the distances patients have to travel to hospitals

and the cost for multiple procedures and their poor patient
compliance. Due to limited resources and general poverty, this
forces the surgeon to resect and reconstruct in the shortest time
possible. These are some of the main reasons against adopting
conservative measures involving decompression and enucleation
with peripheral ostectomy for paediatric and/or adolescents
patients as reported by in recent study on Taiwanese patients
(Huang et al., 2007).
Post-operative difculties for these young individuals include,
interference with craniofacial growth dynamics and function
(mastication and speech), disturbance in psychological well-being
and their progression in education. So far, there have been no
recurrences in this study: this could probably be attributed to our
treatment protocol as mentioned earlier.
It is recommended that multi-centre prospective studies should
be done in our region to record the histopathological types of
ameloblastoma and age of presentation with detailed emphasis on
post-operative craniofacial growth dynamics of those who are less
than 14 years of age.

Please cite this article in press as: Butt FMA, et al., The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching
hospital, in Kenya: A 13-year study, Journal of Cranio-Maxillo-Facial Surgery (2011), doi:10.1016/j.jcms.2011.03.011

F.M.A. Butt et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (2011) 1e7

Fig. 8. Treatment: extensive surgery involving mandibulectomy with disarticulation and reconstruction using a stainless steel plate.

5. Conclusion
The occurrence of ameloblastoma can mimic an odontogenic
cyst, clinicians therefore need to be vigilant when managing
adolescents with such lesions so that conservative approaches
involving decompression and/or enucleation and peripheral
ostectomy can be considered. As long as the lesions are detected
early, that is before the destruction of the lower border, perforation
of the buccal and lingual cortices and involvement of the condyle or
coronoid regions of the mandible, much of the anatomy can be
preserved during surgery and this will reduce the subsequent
morbidity affecting young patients.
Funding
Sources of support: none
Conict of interest statement
None
Acknowledgement
The authors would like to thank the Dean of the University of
Nairobi Dental Hospital for permitting us to conduct this study, the
laboratory technologists, Mr. J O Gichana and Ms. A K Limo for slide
preparation and photography and the administrative staff Serah
Mzenge and Charity W. Mungai for assisting the investigator to
access the medical records.
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Fig. 9. Showing orthopantomogram of an 18-year-old patient at different ages. (A) Preoperative (B) 2-months post-operative with bone graft (C) 4-years post-operative
showing complete healing of the bone graft.

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Please cite this article in press as: Butt FMA, et al., The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching
hospital, in Kenya: A 13-year study, Journal of Cranio-Maxillo-Facial Surgery (2011), doi:10.1016/j.jcms.2011.03.011

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Please cite this article in press as: Butt FMA, et al., The pattern and occurrence of ameloblastoma in adolescents treated at a university teaching
hospital, in Kenya: A 13-year study, Journal of Cranio-Maxillo-Facial Surgery (2011), doi:10.1016/j.jcms.2011.03.011

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