Reminder of important clinical lesson
CASE REPORT
1
Department of Oral Pathology,
Pacific Dental College &
Hospital, Udaipur, Rajasthan,
India
2
Department of Oral &
Maxillofacial Pathology, KM
Shah Dental College &
Hospital, Vadodara, Gujarat,
India
3
Department of Oral &
Maxillofacial Pathology,
Darshan Dental College &
Hospital, Udaipur, Rajasthan,
India
4
Department of Conservative
Dentistry & Endodontics,
Armed Dental Cops,
Chandimandir (WC),
Panchkula, Haryana, India
Correspondence to
Professor Bhari Sharanesha
Manjunatha;
drmanju26@hotmail.com
To cite: Astekar M,
Manjunatha BS, Kaur P,
et al. BMJ Case Rep
Published online: [please
include Day Month Year]
doi:10.1136/bcr-2013-
200316
Astekar M, et al. BMJ Case Rep 2014. doi:10.1136/bcr-2013-200316
Histopathological insight of complex odontoma
associated with a dentigerous cyst
Madhusudan Astekar,1 Bhari Sharanesha Manjunatha,2 Prabhpreet Kaur,3
Jappreet Singh4
SUMMARY histopathology in the proper diagnosis and hence
Odontomas and dentigerous cysts are common findings treatment and further prognosis.
for practicing dental professionals. However,
simultaneous occurrence of pathologies like odontoma CASE PRESENTATION
and dentigerous cyst are uncommon and their diagnosis A 17-year-old male patient reported to the depart-
based on the radiographic appearance alone is a ment of oral medicine with pain in the upper left
challenge to overcome. They together are a potential for
front tooth region since 10–15 days. The pain was
complications like attaining large size, root resorption, mild, dull and non-radiating in nature. It occurred
destruction of the jaw bones and sometimes neoplastic on applying pressure and was relieved by itself a
changes like ameloblastoma. This paper presents a case few seconds after the removal of pressure.
of complex odontoma associated with dentigerous cyst Clinically, the patient was healthy with unre-
in relation to a retained deciduous tooth in the maxillary markable medical history. On inspection, a diffuse,
anterior region and confirming its diagnosis reddish-pink coloured swelling having a smooth
histopathologically. surface causing obliteration of the labial mucosa
and vestibule region of 21, 22 and 63. The retained
deciduous canine was present in the oral cavity.
BACKGROUND INVESTIGATIONS
Odontomas are non-aggressive, hamartomatous Intraoral periapical radiograph revealed the pres-
developmental malformations or lesions of odonto- ence of dense, calcified, amorphous, irregularly
genic origin, which consists of enamel, dentin, shaped radio-opaque mass of varying density meas-
cementum and pulpal tissue.1 2 These lesions are uring approximately 1 cm in diameter. This radio-
composed of more than one type of tissue and for opacity was surrounded by a radiolucency having
this reason, have been called as composite odonto- well-defined borders measuring approximately
mas.3 4 They can be considered as slow-growing
benign tumours showing non-aggressive behaviour.5
The aetiology of odontomas is not clearly estab-
lished but genetic and environmental factors such
as local trauma and infection have been proposed.6
They are classified as complex, when the calcified
tissue presents as an irregular mass composed
mainly of mature tubular dentin or compound if
there is superficial anatomic similarity to even rudi-
mentary teeth.5 7 Complex odontomas are less
common than compound in the ratio of 1 : 2.7
Whereas compound odontomas occur slightly
more often anterior to the mental foramen,
complex odontomas occur more often posterior to
the mental foramen. However, each type may occur
in any location in either jaw without a sex predilec-
tion. It is doubtful whether new odontomas rise
after the age of 25 years.8
They occur more often in the permanent
dentition.9
We report a case of complex odontoma with
dentigerous cyst. The association of the lesion with
a retained deciduous tooth, an impacted permanent
tooth, the unusual site of occurrence and most
importantly its association with a dentigerous cyst
makes it worth presenting. Here we lay emphasis Figure 1 Intraoral periapical radiograph showing
on the histopathological diagnosis to rule out dense, amorphous, irregularly shaped radio-opacity
various other possibilities considered clinically and surrounded by a well-defined radiolucency in the apical
radiographically bringing out the essence of region of 22, 63 and 24.
1
 Reminder of important clinical lesson

Figure 2 Panoramic view showing an irregularly shaped radio-opacity

associated with a well-defined radiolucency in the second quadrant.

1.5 cm×2 cm in the apical region of 22, 63 and 24 (figure 1).

In the panoramic view, radio-opacity enclosed within the radio- Figure 4 Photomicrograph showing the cystic wall with homogenous
lucent lesion is seen in the upper left quadrant. Impacted eosinophilic-stained masses seen in the lumen (H&E, ×10
canine, that is, 23 can also be appreciated here (figure 2). magnification).

DIFFERENTIAL DIAGNOSIS
connective tissue containing small endothelial lined vascular
The mixed radiolucent-radio-opaque appearance will closely
spaces and extravasated RBCs. Rests of odontogenic epithelium
mimic that of a related lesion, the ameloblastic fibro-odontoma.
were evident along with the presence of calcified tissue in the
Odontomas if superimposed over roots may also suggest a
vicinity (figure 3). Photomicrograph shows epithelial lining and
cementoblastoma. In addition, complex odontomas bear a
calcified tissue seen at a higher magnification (figure 4). There
radiographic resemblance to osteoblastomas, ossifying fibromas
was the presence of mature dental tissues like enamel, dentin
and even osteomas. Differential diagnosis made on the basis of
and cementum arranged as unstructured sheets. Components of
clinical and radiological examination were ameloblastic
enamel organ were present. Large mature tubular dentin was
fibro-odontoma, calcified odontogenic cyst, pindborg’s tumour
apparently seen enclosed in clefts or hollow structures which
and complex odontoma with dentigerous cyst.
contained immature enamel or enamel matrix. Small
After correlating the clinical and radiographic findings, a pro-
eosinophilic-stained islands of epithelial ghost cells undergoing
visional diagnosis of complex odontoma with cystic changes was
made.

TREATMENT
Complete surgical excision was performed under local anaesthe-
sia and tissue was sent for histopathological examination.
The macroscopic features of the excisional biopsy specimen
showed a single bit of hard tissue with attached soft tissue
which was rectangular in shape, whitish in colour with irregular
surface texture, measured about 1 cm×0.6 cm with no colour
changes on pressing. The given H&E-stained soft tissue and
decalcified (5% nitric acid) hard tissue section showed one bit
of soft tissue with thin, non-keratinised cuboidal or flattened
epithelial cell lining and underlying loosely arranged fibrous

Figure 5 Photomicrograph showing homogenously stained tissue

with varying intensities. Eosin-stained odontogenic components with
Figure 3 Photomicrograph showing the cystic lining with calcified adjacent clear spaces are visible representing the components of
masses present within the lumen of the cyst (H&E, ×4 magnification). complex odontoma (H&E, ×4 magnification).

2 Astekar M, et al. BMJ Case Rep 2014. doi:10.1136/bcr-2013-200316


Figure 6 Photomicrograph showing randomly placed unstructured
sheets of eosin-stained hard tissue component (H&E, ×10
magnification).
keratinisation were visible (figure 5). Small amount of support-
ing fibrous connective tissue stroma representing fibrous capsule
was also present (figure 6). Under high-power view, homogen-
ous H&E-stained calcified tissues were visible in the background
of loose connective tissue. Inflammatory cells can also be appre-
ciated (figure 7). The overall features were suggestive of
Non-Inflammatory Dentigerous Cyst with Complex Odontoma,
a clinical pathological correlation.
DISCUSSION
According to 1992 WHO classification of odontogenic tumours,
there are four odontogenic tumours with mixed tissue, that is, ame-
loblastic fibroma, complex odontoma, compound odontoma and
ameloblastic fibro-odontoma.5 The term odontoma was coined by
Paul Broca in 1867.10 11 Odontomas are hamartomas of aborted
tooth formation, of which there are two general types. One type,
which forms multiple small tooth-like structures, is called the com-
pound odontoma. The other type forms an amorphous calcified
mass and is called the complex odontoma. Both types may get
somewhat large (up to 6 cm) but will reach a maximum size and
cease growth.8 Clinically odontomas are either complex or com-
pound and are classified as: (1) intraosseous: odontomas occur
inside the bone and may erupt (erupted odontomas) into the oral
cavity and (2) extraosseous: odontomas occurring in the soft tissue
covering the tooth bearing portion of the jaws.5
Figure 7 Photomicrograph showing H&E-stained hard tissues present
in the background of connective tissue (H&E, ×40 magnification).
Astekar M, et al. BMJ Case Rep 2014. doi:10.1136/bcr-2013-200316
Reminder of important clinical lesson
The odontoma seems to result from budding of extraodonto-
genic epithelial cells from the dental lamina. This cluster of cells
forms a large mass of tooth tissue that may be deposited in an
abnormal arrangement but consists of normal enamel, dentin,
cementum and pulp.12 Hitchin suggested that odontomas are
inherited through a mutant gene or interference, possibly post-
natal, with genetic control of tooth development.13–15 They
arise from the odontogenic epithelium, which produces enamel,
and odontogenic mesenchyme, which produces dentin via
odontoblast differentiation. Because they are composed of cell
types and form products of both cell types, they have previously
been termed ‘composite’ odontomas. They represent an attempt
to duplicate tooth formation but in a distorted fashion.8
The dentigerous cyst, also known as the follicular cyst arises
from the follicle of a tooth germ or unerupted tooth or rarely an
odontoma, enclosing the same within it. It develops by the cystic
degeneration of the epithelial component of the enamel organ and
the resultant fluid accumulation between the reduced enamel epi-
thelium and enamel of the tooth.16 Although odontomas may be
found in any tooth bearing region of the jaws, complex odonto-
mas are frequently located in the premolar and molar region of
both jaws.9 The present case reportedly showed complex odon-
toma in the anterior region of the maxilla. An odontoma fre-
quently is situated between the crown of an unerupted tooth and
the crest of the ridge, effectively blocking the tooth’s eruption.12
In the case reported, the odontoma was noted coronal to an
impacted canine and apical to a retained deciduous canine. The
histopathological analysis of the present case reported the presence
of small amount of acellular fibrous connective tissue stroma at the
periphery. Dentigerous cyst arising from the fibrous connective
tissue lining of the odontoma has been rarely reported.17 Cases of
complex odontomas associated with dentigerous cyst have also
been reported in the literature.18
Learning points
▸ Cystic transformation or development from the capsule is
well recognised in situations such as ameloblastomas
originated from a dentigerous cyst. Otherwise, despite
literature reports, dentigerous cysts arising from odontomas
are very rare and could lead to misdiagnosis.
▸ Here the dentigerous cyst is seen to arise from the fibrous
connective tissue lining of the odontoma. There is a
potential for attaining large size, tooth resorption,
destruction of the jaw bones and may show neoplastic
changes like ameloblastomas or carcinoma within isolated
segments of the cyst wall.
▸ The potential complications justify its radiographic screening,
complete enucleation and confirming the diagnosis by
histopathological evaluation for better prognosis.
Competing interests None.
Patient consent Obtained.
Provenance and peer review Not commissioned; externally peer reviewed.
REFERENCES
1 Perumal CJ, Mohamed A, Singh A, et al. Erratum to: sequestrating giant complex
odontoma: a case report and review of the literature. J Maxillofac Oral Surg
2011;10:344–8.
2 Kavitha NL, Venkateswarlu M, Geetha P. Radiological evaluation of a large complex
odontoma by computed tomography. J Clin Diagn Res 2011;5:1307–9.
3
 Reminder of important clinical lesson

3 Zoremchhingi JT, Varma B, Mungara J. A compound composite odontoma
associated with unerupted permanent incisor—a case report. J Indian Soc Pedod
Prev Dent 2004;22:114–17.
4 Baldawa RS, Khante KC, Kalburge JV, et al. Orthodontic management of an
impacted maxillary incisor due to odontoma. Contemp Clin Dent 2011;2:37–40.
5 Masthan KMK, Rajkumari S, Deepasree M, et al. Neoplasms associated with
odontogenic cysts. J Dent Oral Hyg 2011;3:123–30.
6 Wanjari SP, Tekade SA, Parwani RN, et al. Dentigerous cyst associated with multiple
complex composite odontomas. Contemp Clin Dent 2011;2:215–17.
7 Gokkulakrishnan SS, Singh M, Singh KT. A rare case of odontome in a 65-year-old
lady. Natl J Maxillofac Surg 2010;1:67–70.
8 Marx RE, Stern D. Odontogenic tumors: hamartomas and neoplasms. In: Oral and
maxillofacial pathology—a rationale for diagnosis and treatment. 1st edn.
Quintessence Publishing Co, Inc, 2003:678–9.
9 Yildirim-Oz G, Tosun G, Kiziloglu D, et al. An unusual association of odontomas
with primary teeth. Eur J Dent 2007;1:45–9.
10 Singh S, Singh M, Singh I, et al. Compound composite odontoma associated with an
unerupted deciduous incisor—a rarity. J Indian Soc Pedod Prev Dent 2005;23:146–50.
11 Tejasvi MLA, Babu BB. Erupted compound odontomas: a case report. J Dent Res,
Dent Clin, Dent Prospects 2011;5:33–6.
12 Wood NK, Goaz PW, Lehnert JF. Mixed radiolucent-radiopaque lesions associated
with teeth. In: Wood NK, Goaz PW, eds. Differential diagnosis of oral and
maxillofacial lesions. 5th edn. St. Louis, MO: Mosby, 1997:423.
13 Chandra S, Bagewadi A, Keluskar V, et al. Compound composite odontoma
erupting into the oral cavity: a rare entity. Contemp Clin Dent 2010;1:123–6.
14 Shekar S, Rao RS, Gunasheela B, et al. Erupted compound odontoma. J Oral
Maxillofac Pathol 2009;13:47–50.
15 Mehta D, Raval N, Udhani S, et al. An unusual case report of erupted odontoma.
Case Rep Dent 2013;2013:570954.
16 Singh V, Dhasmana S, Mohammad S, et al. The odontomas: report of five cases.
Natl J Maxillofac Surg 2010;1:157–60.
17 Neville BW, Damm DD, Allen CM, et al. Odontogenic cysts and tumors. In: Oral and
maxillofacial pathology. 3rd edn. Saunders: Elsevier, 2002:724–6.
18 Rajendran R. Cysts and tumors of odontogenic origin. In: Rajendran R,
Sivapathasundharam B, eds. Shafe’s textbook of oral pathology. 7th edn. New
Delhi: Elsevier, 2012:259.

Copyright 2014 BMJ Publishing Group. All rights reserved. For permission to reuse any of this content visit
http://group.bmj.com/group/rights-licensing/permissions.
BMJ Case Report Fellows may re-use this article for personal use and teaching without any further permission.
Become a Fellow of BMJ Case Reports today and you can:
▸ Submit as many cases as you like
▸ Enjoy fast sympathetic peer review and rapid publication of accepted articles
▸ Access all the published articles
▸ Re-use any of the published material for personal use and teaching without further permission
For information on Institutional Fellowships contact consortiasales@bmjgroup.com

Visit casereports.bmj.com for more articles like this and to become a Fellow

4 Astekar M, et al. BMJ Case Rep 2014. doi:10.1136/bcr-2013-200316