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Unicystic ameloblastoma

Article  in  Journal of dental sciences · December 2014

DOI: 10.1016/j.jds.2012.03.028

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Journal of Dental Sciences (2014) 9, 407e411

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CASE REPORT

Unicystic ameloblastoma
Ming-Hsuan Hsu a,c, Meng-Ling Chiang a,c*, Jyh-Kwei Chen b,c

a
Department of Pediatric Dentistry, Chang Gung Memorial Hospital, Taipei, Taiwan
b
Department of Oral and Maxillofacial Surgery, Chang Gung Memorial Hospital, Taipei, Taiwan
c
College of Medicine, Chang Gung University, Taoyuan, Taiwan

Received 27 February 2012; Final revision received 23 April 2012

Available online 4 May 2013

KEYWORDS Abstract Unicystic ameloblastomas are a rare variant of ameloblastomas, which usually
computed occur in younger populations. They are characterized by slow growth and being relatively
tomography; locally aggressive, with the main site of origin being the posterior portion of the mandible.
recurrence; Late recurrence following surgical management is relatively common and is related to the his-
unicystic tological type, the site of origin, and the initial treatment modality. This case report describes
ameloblastoma a unicystic ameloblastoma occurring in the right posterior mandible of an 8-year-old girl. She
presented with progressive swelling of the right lower deciduous molar region for 3 months.
Panorex imaging showed a well-defined unilocular radiolucency surrounding the impacted per-
manent first molar, root resorption of the deciduous second molar, and mesial displacement of
the partially formed second bicuspid. Computed tomography revealed expansion of both
buccal and lingual cortical plates of the right posterior mandible by the cystic lesion with in-
clusion of the permanent lower first molar. It was initially diagnosed as a dentigerous cyst and
was treated by enucleation and removal of the permanent first molar and the deciduous sec-
ond molar. The partially formed second premolar and the permanent second molar were left
undisturbed. A histopathological examination of the specimen showed a mural variant of a uni-
cystic ameloblastoma. After surgery, healing of the right mandibular bone defect was unevent-
ful, as shown by follow-up panoramic radiography. No recurrence was detected at an 18-month
follow-up. We suggest that the long-term follow-up is mandatory because late recurrence of
unicystic ameloblastomas has been reported.
Copyright ª 2013, Association for Dental Sciences of the Republic of China. Published by
Elsevier Taiwan LLC. All rights reserved.

Introduction
* Corresponding author. Department of Pediatric Dentistry, Chang
A unicystic ameloblastoma was first described by Robinson
Gung Memorial Hospital, 6th Floor, 199 Tung-Hwa North Road,
and Martinez in 1977.1 Although it is a variant of amelo-
Taipei 105, Taiwan.
E-mail address: [email protected] (M.-L. Chiang). blastomas, it has a relatively benign biologic behavior and

1991-7902/$36 Copyright ª 2013, Association for Dental Sciences of the Republic of China. Published by Elsevier Taiwan LLC. All rights reserved.
http://dx.doi.org/10.1016/j.jds.2012.03.028
408 M.-H. Hsu et al

better response to conservative treatment, and this makes

it a distinguishable entity. It accounts for 15% of all intra-
osseous ameloblastomas, and often affects the younger
population with half of the cases occurring in the second
decade of life.1e8 Unicystic ameloblastomas have a slight
male predilection and frequently originate from the pos-
terior mandible.4,7,8
Unicystic ameloblastomas are characterized as a slow-
growing and relatively locally aggressive cystic lesion.6
Radiographically, the lesions commonly show expansive
unilocular radiolucencies with a well-demarcated border.
Approximately 50e80% of cases are associated with an
impacted or unerupted tooth.7 Therefore, the clinical and
Figure 1 Panoramic radiograph showing a well-defined uni-
radiographic presentations of unicystic ameloblastoma are
locular radiolucent lesion partially surrounding an impacted
sometimes indistinguishable from those of dentigerous
permanent first molar, root resorption of the deciduous second
cysts.2e4,7
molar, and mesial displacement of the partially formed second
Three histological types are recognized according to the
bicuspid.
degree of ameloblastomatous epithelial extension, namely
luminal, intraluminal, and mural types.2 This classification
has a direct bearing on their biological behavior, treatment, Enucleation of the lesion under general anesthesia was
and prognosis.9 Compared to solid and multicystic amelo- performed. During surgery, the cystic lesion, which
blastomas, unicystic ameloblastomas are believed to be less enclosed a permanent lower first molar, was easily sepa-
aggressive and respond more favorably to conservative rated from the surrounding bone since it had an evident
management including enucleation,1,3,5 curettage, and capsule. The deciduous first and second molars were also
marsupialization.10e12 More-aggressive surgical interventions extracted. However, the partially formed second bicuspid
such as resection should be deferred until recurrence occurs. and permanent lower second molar were left undisturbed.
Here, a unicystic ameloblastoma that occurred in the right The entire specimen was then submitted for histopatho-
posterior mandible of an 8-year-old girl is described. The logic examination.
lesion was enucleated, and no recurrence was detected after Gross examination revealed a monocytic lesion. On low-
an 18-month follow-up. power microscopic view, a cystic lesion mainly lined by a
thin layer of nonkeratinizing stratified squamous epithelium
was seen. There was minimal inflammation in the thick
Case report fibrous connective tissue wall. In the focal area, the lining
epithelium grew downward into the underlying connective
This 8-year-old girl together with her mother visited our tissue (Fig. 3A). This invaded epithelium demonstrated a
pediatric dentistry department to seek treatment of basal layer of columnar cells with hyperchromatic nuclei
progressive swelling in the right posterior mandible for that showed reverse polarity and basilar cytoplasmic
3 months. An extraoral examination revealed obvious facial vacuolization (Fig. 3B). The suprabasal epithelial cells were
asymmetry. An intraoral examination showed marked loosely cohesive and resembled a stellate reticulum. Thus
expansion of the buccal cortical plate of the right posterior the final pathological diagnosis was a mural-type unicystic
mandible extending from the deciduous molars to the ameloblastoma (Fig. 3C).
permanent first molar area and the inferior border of the The 18-month follow-up panoramic radiograph showed
mandible. All permanent first molars except the right healing of the right mandibular bone defect and no recur-
mandibular first molar were present in the oral cavity. The rence of the unicystic ameloblastoma after surgery (Fig. 4).
patient’s medical and family histories were essentially
unremarkable.
Panoramic radiography showed a well-defined unilocular Discussion
radiolucent lesion partially surrounding the impacted per-
manent first molar, root resorption of the deciduous second CT, first introduced in the early 1970s, is a very valuable
molar, and mesial displacement of the partially formed tool for lesion diagnosis and treatment planning. The su-
second bicuspid (Fig. 1). Computed tomographic (CT) scans periority of CT over conventional radiographic examinations
revealed expansion of both buccal and lingual cortical in the detection and delineation of intraosseous lesions and
plates of the right posterior mandible by the cystic lesion. their impact on adjacent tissues was suggested by many
The permanent lower first molar was included within the studies.2,13,14 Use of CT in the differential diagnosis, how-
lesion, while the lesion did not involve the partially formed ever, is not extensively emphasized. Since CT scans are
second bicuspid or second deciduous molar (Fig. 2A and B). commonly undertaken nowadays to evaluate pathological
After an intravenous injection of contrast medium, the processes and assess surgical management, its additional
lesion was thought to be a cyst, since it exhibited little value in diagnosis is sometimes overlooked.15 A major
enhancement and did not look like the surrounding soft advantage of CT scans is the reproducible measurement of
tissues such as muscles (Fig. 2C). Based on clinical and radiodensity, presented in a scale of values called Houns-
radiographic findings, a working diagnosis of a dentigerous field units (HU). In our case, the possibility of air contained
cyst was made. within the lesion was excluded by the noncontrast
Unicystic ameloblastoma 409

Figure 2 Computed tomography (CT) of the facial bone. (A) Three-dimensional CT reconstruction revealing a round lesion with
inclusion of the permanent right lower first molar and perforated buccal cortical bone. (B) Axial CT slice without contrast
enhancement displaying an expansive osteolytic lesion of 2.5  4.0  4.0 cm. The permanent right lower first molar is enclosed
within the lesion. (C) Axial CT slice with contrast enhancement showing a hypodense lesion, suggestive of a cystic lesion.

examination, because the lesion was hyperdense compared angiogenesis is positively correlated with the CT attenua-
to that of air (e1000 HU). After an intravenous injection of tion value.15 At this point, the cystic nature of the lesion
contrast medium, there was little enhancement in the was evident, but a further differential diagnosis was not
lesion, suggestive of a cystic lesion, since tumor feasible due to the fact that limited investigations have

Figure 3 Histological photographs of a mural-type unicystic ameloblastoma. (A) Low-power view showing a cystic lesion mainly
lined by a thin layer of nonkeratinizing stratified squamous epithelium. There was minimal inflammation in the thick fibrous
connective tissue wall. In a focal area, the lining epithelium grew downward into the underlying connective tissue. (B) Invaded
epithelium demonstrating a basal layer of columnar cells with hyperchromatic nuclei that showed reverse polarity and basilar
cytoplasmic vacuolization. (C) Suprabasal epithelial cells loosely cohesive and resembling stellate reticulum. (Hematoxylin-eosin
stain, original magnification: A, 10; B, 25; C, 50.)
410 M.-H. Hsu et al

and surgical morbidity.11 Marsupialization is reported to be

useful as preliminary management and as a more-effective
treatment modality for cystic lesions in teenaged patients
as well.10,12 Nakamura et al10 reported a series of 24 uni-
cystic ameloblastomas treated by marsupialization and
found regression of the lesion to less than half the initial
size in 16 lesions. Therefore, when planning treatment of
unicystic ameloblastomas, patient’s esthetic problems,
masticatory function, facial growth, quality of life, and
potential morbidity caused by the surgical intervention
should be taken into account.
Regarding unicystic ameloblastomas, recurrence
following treatment is reported at w7e25%1,12,19 and is
Figure 4 Eighteen-month follow-up panoramic radiograph
related to the histological type, site of origin, and initial
showing healing of the right mandibular bone defect and no
treatment modality.4,17 Ackermann et al2 classified uni-
recurrence of the unicystic ameloblastoma. The second
cystic ameloblastomas into three histologic subgroups:
bicuspid has erupted to the occlusal level, and the second
luminal type, in which the tumor is confined to the lining
molar has emerged into the oral cavity.
epithelium of the cyst; intraluminal type, in which nodular
proliferation of neoplastic epithelium projects into the
been conducted regarding the density patterns of unicystic lumen; and mural type, in which ameloblastomatous
ameloblastomas, odontogenic keratocysts, and dentigerous epithelium in either a follicular or plexiform pattern in-
cysts.16 vades the connective tissue wall. Among the three sub-
Biopsy of a large pericoronal cystic lesion is usually types, the mural type has the highest recurrence rate
necessary, because this type of lesion may include a den- since the epithelium penetrates and breaches the fibrous
tigerous cyst, odontogenic keratocyst, unicystic or con- wall with a high potential to invade the adjacent cancel-
ventional ameloblastoma, ameloblastic fibroma, lous bone.11 Li et al8,20 reported a higher recurrence rate
adenomatoid odontogenic tumor, or odontogenic fibroma. of 35.7% for the mural type and a lower recurrence rate of
The cystic cavities of unicystic ameloblastomas are not al- 6.7% for the luminal and intraluminal types.7 More-radical
ways uniformly characteristic and are often partly outlined management therefore should be given for the mural
with a nonspecific epithelium similar to the lining of den- type.5,7,8,18,19 Conservative interventions are generally
tigerous cysts. Biopsies consisting exclusively of such preferred for unicystic ameloblastomas in the mandible
epithelium may be unable to reflect the true nature of the but are not suggested for those in the maxilla, because of
entire lesion.3,7,8 This kind of biopsy may lead to an the spongy osteoarchitecture of the maxilla which facili-
incorrect diagnosis and subsequent inadequate treatment. tates spread of the tumor and the close proximity to vital
By contrast, unicystic ameloblastomas often clinically and structures such as the orbit, pterygomaxillary fossa, and
radiographically mimic cystic lesions. A lack of a biopsy cranium.3,8 More-aggressive interventions in the form of
along with a diagnosis solely dependent on clinical and resection basically eliminate the risk of recurrence, but
radiographic findings may give rise to considerable diag- they are usually not the most favorable treatment mo-
nostic problems and thus render the lesion either over- dality for unicystic ameloblastomas.12,17,19 We suggest
estimated or underestimated.5,6 In this case, an incisional that relatively conservative therapies can initially be
biopsy was not undertaken, and the lesion was regarded as applied for unicystic ameloblastomas, with more-
a dentigerous cyst. Only after enucleation could the aggressive approaches being reserved for later
definitive diagnosis of a unicystic ameloblastoma be made. recurrence.11
Fortunately, unicystic ameloblastomas have less-aggressive Long-term follow-up is mandatory for unicystic amelo-
biological behavior and a better prognosis even after con- blastomas since recurrence may take place years after
servative surgical treatment. However, we still suggest that removal.8,11 More than 50% of cases recurred within 5 years
an incisional biopsy be performed before surgery for a large after the operation.6 Frequent postsurgical radiographic
pericoronal radiolucent lesion, because other more- examinations favor early detection of recurrence.
aggressive lesions such as odontogenic keratocysts or con-
ventional ameloblastomas may be encountered.
Unicystic ameloblastomas compare favorably with their
Conflicts of interest
solid counterparts in terms of clinical behavior and
response to treatment.1,3,12 Accordingly, conservative The authors declare that there are no conflicts of interest
treatment is suggested, especially in younger populations, that could influence their work.
in light of the devastating impacts on the developing jaw,
masticatory function, facial growth, and psychosocial References
aspects.4e6,10,11,17 Porgrel et al18 advocated that enucle-
ation followed by curettage and liquid nitrogen cryospray 1. Robinson L, Martinez MG. Unicystic ameloblastoma: a prog-
or Carnoy’s solution cauterization would be appropriate for nostically distinct entity. Cancer 1977;40:2278e85.
unicystic ameloblastomas. In extensive lesions, marsupial- 2. Ackermann GL, Altini M, Shear M. The unicystic amelo-
ization may be an alternative treatment, because it is easy blastoma: a clinicopathological study of 57 cases. J Oral Pathol
to perform and safe, and can reduce the size of the lesion 1988;17:541e6.
Unicystic ameloblastoma 411

3. Isacsson G, Andersson L, Forsslund H, Bodin I, Thomsson M. 13. Crusoe-Rebello I, Oliveira C, Campos PS, Azevedo RA, dos
Diagnosis and treatment of the unicystic ameloblastoma. Int J Santos JN. Assessment of computerized tomography density
Oral Maxillofac Surg 1986;15:759e64. patterns of ameloblastomas and keratocystic odontogenic tu-
4. Bisinelli JC, Ioshii S, Retamoso LB, Moyses ST, Moyses SJ, mors. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;
Tanaka OM. Conservative treatment of unicystic amelo- 108:604e8.
blastoma. Am J Orthod Dentofacial Orthop 2010;137:396e400. 14. Hertzanu Y, Mendelsohn DB, Cohen M. Computed tomography
5. Zhang J, Gu Z, Jiang L, et al. Ameloblastoma in children and of mandibular ameloblastoma. J Comput Assist Tomogr 1984;8:
adolescents. Br J Oral Maxillofac Surg 2010;48:549e54. 220e3.
6. Huang IY, Lai ST, Chen CH, Chen CM, Wu CW, Shen YH. Surgical 15. Hayashi K, Tozaki M, Sugisaki M, Yoshida N, Fukuda K,
management of ameloblastoma in children. Oral Surg Oral Med Tanabe H. Dynamic multislice helical CT of ameloblastoma and
Oral Pathol Oral Radiol Endod 2007;104:478e85. odontogenic keratocyst: correlation between contrast
7. Philipsen HP, Reichart PA. Unicystic ameloblastoma. A review enhancement and angiogenesis. J Comput Assist Tomogr 2002;
of 193 cases from the literature. Oral Oncol 1998;34:317e25. 26:922e6.
8. Li TJ, Wu YT, Yu SF, Yu GY. Unicystic ameloblastoma: a clini- 16. Konouchi H, Asaumi J, Yanagi Y, et al. Usefulness of contrast
copathologic study of 33 Chinese patients. Am J Surg Pathol enhanced-MRI in the diagnosis of unicystic ameloblastoma.
2000;24:1385e92. Oral Oncol 2006;42:481e6.
9. Kawamura JY, de Magalhaes RP, Sousa SC, Magalhaes MH. 17. Hong J, Yun PY, Chung IH, et al. Long-term follow up on
Management of a large dentigerous cyst occurring in a six-year- recurrence of 305 ameloblastoma cases. Int J Oral Maxillofac
old boy. J Clin Pediatr Dent 2004;28:355e7. Surg 2007;36:283e8.
10. Nakamura N, Higuchi Y, Tashiro H, Ohishi M. Marsupialization of 18. Pogrel MA, Montes DM. Is there a role for enucleation in the
cystic ameloblastoma: a clinical and histopathologic study of management of ameloblastoma? Int J Oral Maxillofac Surg
the growth characteristics before and after marsupialization. 2009;38:807e12.
J Oral Maxillofac Surg 1995;53:748e56. 19. Lee PK, Samman N, Ng IO. Unicystic ameloblastomaduse of
11. Li TJ, Kitano M, Arimura K, Sugihara K. Recurrence of unicystic Carnoy’s solution after enucleation. Int J Oral Maxillofac Surg
ameloblastoma: a case report and review of the literature. 2004;33:263e7.
Arch Pathol Lab Med 1998;122:371e4. 20. Li T, Wu Y, Yu S, Yu G. Clinicopathological features of unicystic
12. Nakamura N, Higuchi Y, Mitsuyasu T, Sandra F, Ohishi M. ameloblastoma with special reference to its recurrence.
Comparison of long-term results between different approaches Zhonghua Kou Qiang Yi Xue Za Zhi 2002;37:210e2.
to ameloblastoma. Oral Surg Oral Med Oral Pathol Oral Radiol
Endod 2002;93:13e20.

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