Saudi Journal of Pathology and Microbiology (SJPM) ISSN 2518-3362 (Print)

Scholars Middle East Publishers ISSN 2518-3370 (Online)

Dubai, United Arab Emirates
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Adenomatoid Odontogenic Tumor of Mandible: A Rare Case Report

Dr. Suman Kumari1*, Dr. Deepti Agarwal2, Dr. Parveen Kundu2, Dr. Swaran Kaur3, Dr. Kulwant Singh2
1
Post graduate student, Department of Pathology, Bhagat Phool Singh Government Medical College for Women,
Khanpur kalan, Sonepat, Haryana, India
2Associate Professor, Department of Pathology, Bhagat Phool Singh Government Medical College for Women, Khanpur
kalan, Sonepat, Haryana, India
3
Professor & Head, Department of Pathology, Bhagat Phool Singh Government Medical College for Women, Khanpur
kalan, Sonepat, Haryana, India

Abstract: Adenomatoid Odontogenic Tumor is a rare tumor constituting 3-7% of all

Case Report odontogenic tumors. It is a benign neoplasm which arises from the odontogenic
epithelium. We present a case of 22 year female patient with a well defined, unilocular
*Corresponding author radiolucent lesion containing an impacted tooth seen radiographically. On microscopic
Dr. Suman Kumari examination, sections comprise of cyst wall composed of fibrous connective tissue with
areas of haemorrhage. Whorls and lobules of spindle shaped cells were seen on luminal
Article History aspect with interspersed duct like structures. Based on the above histopathological
Received: 02.10.2018 features final diagnosis of Adenomatoid Odontogenic Tumor was made. The treatment
Accepted: 10.10.2018 of choice is conservative surgical removal through simple curettage or enucleation.
Published: 30.10.2018 Recurrence is very rarely seen.
Keywords: Odontogenic, radiolucent, adenomatoid.
DOI:
10.21276/sjpm.2018.3.10.7 INTRODUCTION
Philipsen et al., [1] proposed the term “Adenomatoid Odontogenic Tumor”
(AOT). It is a benign neoplasm which arises from the odontogenic epithelium
accounting for 3-7% of all odontogenic tumors [2]. The tumor is common in younger
age group between 10-30 years and around two-thirds of all cases are reported when
patient is between 10-19 years of age [2, 3, 4]. The incidence is uncommon in patients
older than 30 years. It commonly occurs in the anterior portion of the jaw (95%) and is
found twice as often in the maxilla (65%) than in the mandible. Females are affected
about twice as often as males [2, 3, 5].

AOT has three clinical presentations: intra- Radiographically, a well defined, unilocular radiolucent
osseous (associated with impacted teeth and also known lesion containing an impacted tooth was seen. Any
as “follicular”; 70% of cases); intra-osseous (present internal septation or sclerotic area was not seen (Figure-
between erupted teeth and also known as “extra- 1). The provisional diagnosis of dentigerous cyst was
follicular”; 25% of cases); and extra-osseous (on the made. The lesion was surgically removed and sent for
gingiva, also known as “peripheral”; 5% of cases) [5]. histopathological examination. The specimen received
The treatment of choice is conservative surgical was grossly, globular in shape, firm in consistency with
removal through simple curettage or enucleation [6, 7]. an embedded tooth. Cut section showed a cystic cavity
Recurrence is very rarely seen [6-9]. containing tooth with papillary growth like structures
lining the cavity (Figure-2). On microscopic
CASE REPORT examination, sections comprised of cyst wall composed
A 22 year female patient presented to the of fibrous connective tissue with areas of haemorrhage.
Department of Dentistry of our institute with chief Whorls and lobules of spindle shaped cells were seen on
complaint of pain in left lower jaw anterior region for luminal aspect with interspersed duct like structures
past 5-6 months. On examination, intraorally a swelling (Figure-3a &b). Based on the above histopathological
was palpated in the region of lower left incisor. The features final diagnosis of Adenomatoid Odontogenic
swelling was firm in consistency and non-tender. Tumor was made.

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Suman Kumari et al., Saudi J. Pathol. Microbiol., Vol-3, Iss-10 (Oct, 2018): 354-356

Fig-1: Panoramic radiograph showing impacted permanent mandibular right canine in a well-defined
radiolucency, expansion and thinning of the inferior border of the mandible, displacement of the anterior teeth

Fig-2: Gross appearance of grey brown to tan cystic tumor mass with embedded root of canine teeth

Fig-3: Adenomatoid odontogenic tumor. (a) Low-power view demonstrating a capsule surrounding the
tumor.(20X, H&E) (b) Higher magnification showing the duct-like epithelial structures. (100X, H&E)

DISCUSSION demarcated radiolucent lesion of 2.5cm diameter in the

The differential diagnosis of jaw swelling in a anterior mandibular region associated with an impacted
young patient include both non neoplastic and permanent canine. The tumor progressively enlarged
neoplastic causes. The common non neoplastic causes over a period of 5-6 months.
are an apical cyst, dentigerous cyst, calcifying
odontogenic cyst, odontogenic keratocyst, and central Since the lesion was well-demarcated and
giant cell granuloma [2, 10, 11], whereas common radiolucent and associated with the crown of impacted
neoplastic causes are an adenomatoid odontogenic teeth like in this case, possibility of apical cyst,
tumor, unicystic ameloblastoma, calcifying epithelial calcifying odontogenic cyst, odontogenic keratocyst,
odontogenic tumor (CEOT), ameloblastic fibroma, and and central giant cell granuloma was ruled out.10,14,15
ameloblastic fibroodontoma [2, 11, 12]. Further an apical cyst is usually associated with an
endodontic-treated or mortified pulptooth. Calcifying
Adenomatoid odontogenic tumor (AOT) is an odontogenic cyst, odontogenic keratocyst, and giant cell
odontogenic epithelial tumor commonly affecting granuloma were ruled out because usually they are not
young female patients between 10 and 19 years of age. related to the crown of an impacted tooth, and they are
The maxilla being the more common site compared to mostly multilocular. Dentigerous cyst, unicystic
the mandible. The size of the lesion ranges from 2 to ameloblastoma, ameloblastoma, and ameloblastic
7cm with a slow growing pattern which results in a fibroma are most frequent in the posterior region of the
painless expansion of the jaws. The canine is the most mandible and are associated in most cases with the third
common tooth which is impacted in AOT [3, 10, 13]. In molar. However, adenomatoid odontogenic tumor
this case, a 22-year-old female presented with a well-

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Suman Kumari et al., Saudi J. Pathol. Microbiol., Vol-3, Iss-10 (Oct, 2018): 354-356
occurs mostly in the anterior maxillary region and is 4. Wood, N. K., Goaz, P. W., & Alling, C. C.
associated in 60% of cases with a canine [14, 15]. (1991). Differential diagnosis of oral lesions. St.
Louis: Mosby.
Mostly, AOTs are the central follicular type 5. More, C. B., Das, S., Gupta, S., & Bhavsar, K.
and appear as well-demarcated radiolucent lesions. (2013). Mandibular adenomatoid odontogenic
AOT usually surrounds an unerupted tooth, and it looks tumor: Radiographic and pathologic
as a corticated radiolucency with small radiopacities, correlation. Journal of natural science, biology,
but there are cases where the lesion has no radiopaque and medicine, 4(2), 457.
component, and in such cases, a dentigerous cyst is the 6. Saluja, R., Kaur, G., & Singh, P. (2013).
preferred differential diagnosis. However, an AOT Aggressive adenomatoid odontogenic tumor of
often appears to envelop the crown as well, when we mandible showing root resorption: A histological
divide the specimen to show the relation of the lesion to case report. Dental research journal, 10(2), 279.
the tooth, unlike the dentigerous cyst which does not 7. Baskaran, P., Misra, S., Kumar, M. S., & Mithra,
surround the roots [8, 13, 15]. In this case, the lesion R. (2011). Adenomatoid odontogenic tumor–A
surrounded the entire crown and overlapped the root of report of two cases with histopathology
the lateral incisor. Few cases of AOT, like this case, correlation. Journal of clinical imaging science, 1.
were described in the literature in association with a 8. Vasudevan, K., Kumar, S., &
mandibular canine. Vijayasamundeeswari, S. V. (2012). Adenomatoid
odontogenic tumor, an uncommon
The histological typing of the WHO defined tumor. Contemporary clinical dentistry, 3(2), 245.
the AOT “like a tumor of odontogenic epithelium with 9. Jindwani, K., Paharia, Y. K., & Kushwah, A. P. S.
duct-like structures and with varying degrees of (2015). Surgical management of peripheral variant
inductive change in the connective tissue. The tumor is of adenomatoid odontogenic tumor: A rare case
well encapsulated and shows an identical benign report with review. Contemporary clinical
behaviour [7, 13].” The treatment of choice was a dentistry, 6(1), 128.
surgical management of this tumor. Enucleation of 10. Marx, R. E., & Stern, D. (2012). Oral and
tumor along with the associated impacted tooth and Maxillofacial Pathology: A Rationale for Diagnosis
simple curettage is the treatment of choice. and Treatment, Quintessence Publishing Co,
Conservative treatment is adequate because the tumor is Hanover Park, IL, USA, 2nd Edition.
not locally invasive, is well encapsulated, and can be 11. Daryani, D., & Gopakumar, R. (2011). Central
easily separated from the bone [3]. giant cell granuloma mimicking an adenomatoid
odontogenic tumor. Contemporary clinical
CONCLUSION dentistry, 2(3), 249.
AOTs are usually asymptomatic lesions that 12. Bulut, E., Tasar, F., Akkocaoglu, M., & Ruacan, S.
sometimes may cause cortical expansion and (2001). An adenomatoid odontogenic tumor with
displacement of the adjacent teeth. Early diagnosis of unusual clinical features. Journal of oral
and early enucleation prevents an excessive destruction science, 43(4), 283-286.
of bone and it should be differentiated from close 13. Swasdison, S., Dhanuthai, K., Jainkittivong, A., &
differential diagnosis of dentigerous cyst. Philipsen, H. P. (2008). Adenomatoid odontogenic
tumors: an analysis of 67 cases in a Thai
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