Escolar Documentos
Profissional Documentos
Cultura Documentos
com
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.
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
Results
with unilocular radiolucent lesions, whereas minor of uni- and Adekeye (1.5:1) and Huang et al. (1.5:1),1,3 but showed
cystic tumors presented in multilocular radiolucent lesions a deviation from the findings of Kahn (0.9:1) and Ord et al.
(4/16, 25%). (1:1.75).10,11 Some authors also have reported equal gender
Postoperative follow-up period ranged from 3 months to distribution.5,7,13 In literature review, the distribution among
6 years. Of the total of 10 recurrences, 9 patients had one males and females were almost identical: 53.6% males and
recurrence, 1 with two recurrences. Histologically, nine were 46.4% female, a ratio was 1.16:1. The average age was 14.8
solid type, one was unicystic type. Twenty-seven patients years in our series, this finding was consistent with the data
(73%) were lost to follow-up after 2 years. 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
Review of the literature under 11 years old. These findings are similar to previous
reports. Arotiba et al. reported 10 (12.7%) cases,6 while Ord
Ameloblastoma was rare in the pediatric population and only et al. and Chidzonga found none in their series.11,13 These
10 relevant publications were included in this study (Table 1). results also consistent with only 10.8% under 11 years old in
A total of 233 patients (233/1470, 15.9%), 125 were male and literature review.
108 were female, a ratio of 1.16:1. The mean age was 14.5 The site distribution in the published literature showed
years (range from 4 to 20 years). The mandible was the most that the ameloblastomas have a marked predilection for the
common site of occurrence in all races; 225 (96.6%) tumors mandible (96.6%). This was similar to other studies. The
were encountered in the mandible and 8 (3.4%) in the maxilla, molar-ramus region was the most common site followed by
with an overall mandible:maxilla ratio of 28.1:1. the symphyseal region. Painless swelling was the most com-
Majority of the tumor (89.2%, 181 of 203) were found in mon complaint of the patients in this study (54.1%).
patients between 11 and 20 years old, only 10.8% (22 of 203) Radiologically, it has been reported that the unilocular
under 11 years old. Mandibular posterior region was the most ameloblastomas tend to occur in younger age groups.3,10
commonly involved, followed by the symphyseal region. Our findings confirmed this tendency (56.8%). However, the
Radiologically, 56.7% (120 of 212) of ameloblastomas were results of literature review revealed that multilocular radiolu-
multilocular and 43.4% (92 of 212) were unilocular (19 cases cent lesions (56.7%) predominated over unilocular lesions
were not recorded and 2 were ameloblastic carcinoma). His- (43.3%).
tologically, solid type occupied 63.1% (111 of 176), unicystic Ameloblastomas are currently classified into three types:
type was 36.9% (65 of 176). (a) solid or multicystic, the most frequent form and radio-
Seven patients had no treatment, the remaining were man- logically characterized by multilocular aspect, (b) unicystic,
aged surgically. Of 226, 123 (54.4%) patients were treated radiologically characterized by unilocular aspect and (c)
with radical resection, while 103 (45.6%) patients under- extraosseous or peripheral type. Our cases included the solid
went conservative surgical management. The recurrence rate and unicystic ameloblstomas. Some studies reported that
after radical therapy was lower than conservative therapy. the unicystic ameloblastoma is the most commonly type in
The follow-up period varied from 1 month to 19 years. Most children,11,14 whereas in our study the solid multicystic type
patients were lost to follow-up within 2 years of surgery, (75.7%) predominated over unicystic type (24.3%). This was
therefore, it was difficult to evaluate the precise recurrence consistent with the relevant literature, the rate of solid type
rates. (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-
Discussion ular radiolucent lesions, whereas minor of unicystic tumors
presented in multilocular radiolucent lesions (4/16, 25%).
A review of previous literature of ameloblastoma in the chil- Some publications also showed similar results.3,5,10 There-
dren and adolescents showed wide variation in age selection, fore, when the ameloblastomas appear as unilocular lesions
therefore, it is difficult to evaluate the precise incidence radiographically, the diagnosis of solid type also should be
because different authors have defined the upper age limit considered. And in order to avoid the high recurrence rates
such as under 20 years, 18 years.911 The incidence of 13.9% by conservative treatment, a biopsy is recommended.
(37/267) of ameloblastoma in children in this study is simi- The unicystic ameloblastoma is thought to have recur-
lar the incidence of 14.6% reported by Olaitan and Adekeye rence potential, but to be less aggressive than the solid type.
and the 12.2% found by Kahn.1,10 However, Huang et al. and Previous literature reported that enucleation alone may be
Keszler and Dominguez reported a relatively low prevalence sufficient to eradicate the tumor completely. However, current
of ameloblastoma (6.8% and 8.7%, respectively),3,7 while studies revealed that the third histologic subtype of uni-
Ord et al. and Al-Khateeb and Ababneh reported high rates cystic ameloblastoma (a cystic lining showing intraluminal
(28.9% and 38.5%, respectively).11,12 and intramural proliferations) was aggressive and should be
In our study, the malefemale ratio was 1.6:1. This was treated more radically as a solid ameloblastoma.15 There-
similar to that reported by Daramola et al. (1.7:1),9 Olaitan 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
initial presentation, such as enucleation and curettage fol- follow the patients an enough long time (i.e. 20 years). There-
lowed by physiochemical treatment or peripheral ostectomy fore, data of the long-term follow-up could not acquire in our
in the mandible and localized resection in maxilla, and reserve series.
more aggressive therapy for any recurrences.
Solid ameloblastoma as a locally aggressive tumor can
lead to a high recurrence rates (7590%) following conserva-
Conict of interest statement
tive treatment.9,11 The decision to do initial radical, extensive
surgery or conservative procedures treatment in children
I, the undersigned author, certify that I have no financial or
always poses a dilemma.16 The current opinion favors radical
personal relationships (e.g. employment, consultancies, stock
resection for decreasing recurrence rates.17,18 However, in the
ownership, honoraria, etc.) that pose a conflict of interest in
pediatric group, management of solid ameloblastomas often
connection with the submitted article.
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 References
could result in deformity and dysfunction of the jaw that are
bound to influence both the physical and psychological devel- 1. Olaitan AA, Adekeye EO. Clinical features and management of
ameloblastoma of the mandible in children and adolescents. Br J Oral
opment of the child in later life. Moreover, tumor size and Maxillofac Surg 1996;34:24851.
unilocular appearance would also influence the decision of 2. Rapidis AD, Andressakis DD, Stavrianos SD, Faratzis G, Arnogiannaki-
surgeons. These factors could increase the risk of recurrence. Liappi N, Lagogiannis GA, et al. Ameloblastomas of the jaws: clinico-
Preoperative confirmation of histological type by means of pathological review of 11 patients. Eur J Surg Oncol 2004;30:9981002.
incisional biopsy or confirmation during surgical procedures 3. Huang IY, Lai ST, Chen CH, Chen CM, Wu CW, Shen YH. Surgical
management of ameloblastoma in children. Oral Surg Oral Med Oral
by surgical procedures by means of frozen section would help Pathol Oral Radiol Endod 2007;104:47885.
clinicians select more suitable treatment modalities, which 4. Reichart PA, Philipsen HP, Sonner S. Ameloblastoma: biological profile
may help to reduce the recurrence rate. If radical resection has of 3677 cases. Eur J Cancer B: Oral Oncol 1995;31:8699.
to be performed, the immediate bone graft is recommended 5. Takahashi IK, Miyauchi K, Sato K. Treatment of ameloblastoma in
in order to prevent an obvious facial deformity. children. Br J Oral Maxillofac Surg 1998;36:4536.
6. Arotiba GT, Ladeinde AL, Arotiba JT, Ajike SO, Ugboko VI, Ajayi O.
Postoperative follow-up is very important because more Ameloblastoma in Nigerian children and adolescents: a review of 79
than 50% recurrences occur within 5 years of treatment.3,19 cases. J Oral Maxillofac Surg 2005;63:74751.
Our cases also confirmed the tendency. Many reports reveal 7. Keszler A, Dominguez FV. Ameloblastoma in children. J Oral Maxillo-
the recurrence time is between 1 and 15 years, and 25 years is fac Surg 1986;44:60913.
the most common.10,17 For the most accurate recurrence rate 8. Ueno S, Nakamura S, Mushimoto K, Shirasu R. A clinicopathologic
study of ameloblastoma. J Oral Maxillofac Surg 1986;44:3615.
findings, all cases should be routinely follow-up a long time 9. Daramola JO, Ajagbe HA, Oluwasanmi JO. Ameloblastoma of the jaws
because of the insidious biological behavior of ameloblas- in Nigerian children: a review of sixteen cases. Oral Surg Oral Med Oral
toma. However, it was a consistent problem for clinicians to Pathol 1975;40:45863.
554 J. Zhang et al. / British Journal of Oral and Maxillofacial Surgery 48 (2010) 549554
10. Kahn MA. Ameloblastoma in young persons: a clinicopathologic anal- 15. Philipsen HP, Reichart PA. Unicystic ameloblastoma. A review of 193
ysis and etiologic investigation. Oral Surg Oral Med Oral Pathol cases from the literature. Oral Oncol 1998;34:31725.
1989;67:70615. 16. Ghandhi D, Ayoub AF, Pogrel MA, MacDonald G, Brocklebank LM,
11. Ord RA, Blanchaert Jr RH, Nikitakis NG, Sauk JJ. Ameloblastoma in Moos KF. Ameloblastoma: a surgeons dilemma. J Oral Maxillofac Surg
children. J Oral Maxillofac Surg 2002;60:76270. 2006;64:10104.
12. Al-Khateeb T, Khansa T, Ababneh KT. Ameloblastoma in young Jor- 17. Feinberg SE, Steinberg B. Surgical management of ameloblastoma. Cur-
danians: a review of the clinicopathologic features and treatment of 10 rent status of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol
cases. J Oral Maxillofac Surg 2003;61:138. Endod 1996;81:3838.
13. Chidzonga MM. Ameloblastoma in childrenthe Zimbabwean expe- 18. Pogrel MA, Montes DM. Is there a role for enucleation in the manage-
rience. Oral Surg Oral Med Oral Pathol Oral Radiol Endod ment of ameloblastoma? Int J Oral Maxillofac Surg 2009;38:80712.
1996;81:16870. 19. Olaltan AA, Arole G, Adekeye EO. Recurrent ameloblastoma of the
14. Leider AS, Eversole LR, Barkin ME. Cystic ameloblastoma: a clinico- jaws: a follow-up study. Int J Oral Maxillofac Surg 1998;27:456
pathologic analysis. Oral Surg Oral Med Oral Pathol 1985;60:62430. 60.