[Downloaded free from http://www.jiaomr.in on Tuesday, February 14, 2023, IP: 182.253.133.

211]

Case Report

Compound-complex odontoma: A case report

of a rare variant
Nishath Khanum, Mahesh Mysore Shivalingu, Naresh Lingaraju, Srisha Basappa
Department of Oral Medicine and Radiology, Farooqia Dental College and Hospital, Mysore, Karnataka, India

ABSTRACT

The odontoma is a benign tumor containing all the various component tissues of the teeth. It is the most common odontogenic
tumor representing 67% of all odontogenic tumors. Odontomas are considered to be developmental anomalies (hamartomas)
rather than true neoplasms. Based on the degree of morphodifferentiation or on the basis of their resemblance to normal
teeth, they are divided into compound and complex odontomas. The compound odontoma is composed of multiple, small
tooth-like structures. The complex odontoma consists of a conglomerate mass of enamel and dentin, which bears no
anatomic resemblance to a tooth. They are usually diagnosed on routine radiological examinations in the second decade of
life and are often slow growing and non-aggressive in nature. Here, we report a case of rare, unusually large, compound-
complex odontoma, located in the left anterior maxilla of a 13-year-old male patient.
Key words: Compound complex odontoma, hamartoma, odontogenic tumor, radiopacities

Introduction the time of diagnosis being 14 years, odontomas are

usually detected during the first two decades of life.[3]

T
he term odontoma was used earlier to describe
any odontogenic origin tumour. In 1867, Paul The World Health Organization (WHO) classifies
Broca coined the term odontoma. He defined the odontomas into compound and complex odontomas.
term odontoma as ‘tumors formed by the overgrowth Complex odontomas are less common than the compound
of transitory or complete dental tissues.[1] Because variety, and the latter occurs more commonly in the
the odontogenic cells do not reach the normal state maxilla, having a predilection for the incisor-canine
of morphodifferentiation, the enamel and dentin are region without gender bias.[2] The complex odontoma is
formed in an abnormal pattern. Although unknown, more common in the mandibular posterior teeth region
the etiology is said to include local trauma or infection. and has a female predilection.[4] However, rarely, lesions
A gene mutation or postnatal interference with the may show features of both compound and complex
genetic control of tooth development is also one of the odontomas. The treatment of choice is surgical removal
suggested causes.[2] Odontomas are slow growing and of the lesion, followed by histopathological examination
often non-aggressive in nature. With the mean age at to confirm the diagnosis.

Access this article online

Quick Response Code:
Case Report
Website:
www.jiaomr.in A 13-year-old male patient visited the department of
Oral Medicine and Radiology, with a chief complaint
DOI: of swelling in the upper left back teeth region since
10.4103/0972-1363.155668 1 year. The swelling was gradually and constantly
increasing in size to reach the present size. No history of

Address for correspondence: Dr. Nishath Khanum, Department of Oral Medicine and Radiology, Farooqia Dental College and
Hospital, Mysore - 570 021, Karnataka, India. E-mail: nishkhan7@yahoo.co.in
Received: 22-08-2014 Accepted: 29-03-2015 Published: 22-04-2015

Journal of Indian Academy of Oral Medicine & Radiology | Oct-Dec 2014 | Vol 26 | Issue 4 463
[Downloaded free from http://www.jiaomr.in on Tuesday, February 14, 2023, IP: 182.253.133.211]

Khanum N et al.: Compound-complex odontoma

pus, blood or watery discharge or any other secondary and inferiorly to the alveolar crest, medially to the distal
changes were noted. root surface of 22 and laterally to the mesial surface of
26. The missing premolars were pushed distally near
On local extraoral examination, a solitary, ill-defined the mesial root surface of 26 and the canine was placed
swelling was present in the left middle 1/3rd of the face. near the infra-orbital margin [Figure 2]. The occlusal
On intraoral hard tissue examination, 23, 24 and 25 were radiograph also showed a solitary, well-defined, mixed
clinically missing and 63 was retained with pre-shedding radiopaque-radiolucent lesion measuring about 3 ×
mobility. There was a well-defined solitary oval swelling 3 cm in the left posterior maxilla, surrounded by a thin
present in relation to the left buccal alveolar bone, measuring radiolucent rim. The impacted 24 and 25 were displaced
about 2 × 3 cm, extending from the distal aspect of 22 to the distally and palatally in relation to 26 [Figure 3].
mesial aspect of 26. The mucosa over the swelling was
normal with no pus, blood or watery discharge. There was Based on the history and clinical examination, a provisional
no evidence of ulcer or growth noted on the mucosa. On diagnosis of dentigerous cyst was made. Differential
palpation, the swelling was non-tender, hard in consistency diagnosis included adenomatoid odontogenic tumor,
with a smooth surface and well-defined borders. Expansion odontoma, and keratocystic odontogenic tumor. Based
of the buccal cortical plate was noted [Figure 1]. on the radiographic appearance a diagnosis of complex
odontoma was made. The tumor was surgically removed
The panoramic radiograph (OPG) revealed a well- under local anesthesia [Figure 4]. The gross specimen
defined mixed radiopaque-radiolucent lesion measuring consisted of an irregular mass of calcified tissue as well as
about 3 × 3 cm in diameter in the left maxilla, surrounded multiple teeth-like structures [Figure 5]. The post-operative
by a thin radiolucent rim. The lesion extended superiorly OPG revealed complete enucleation of the tumor [Figure 6].
up to the floor of the maxillary sinus and the nasal cavity
On histopathological examination, the sections revealed
calcified structures like enamel, dentin, and cementum
which were intermingled with pulp-like tissues in few

Figure 2: Preoperative OPG showing the mixed radiopaque-radiolucent

Figure 1: Intraoral photograph lesion

Figure 3: Maxillary occlusal view Figure 4: Intraoperative photograph showing the exposed odontoma

464 Journal of Indian Academy of Oral Medicine & Radiology | Oct-Dec 2014 | Vol 26 | Issue 4
[Downloaded free from http://www.jiaomr.in on Tuesday, February 14, 2023, IP: 182.253.133.211]

Khanum N et al.: Compound-complex odontoma

areas. The calcified structures were arranged regularly The present case is unique in that it has combined
in few areas, whereas, in few areas it was haphazardly features of both compound and complex odontoma.
arranged. Few odontogenic islands were seen in a Very few cases of compound - complex odontoma have
fibro-vascular connective tissue [Figures 7 and 8]. been reported in literature.[5] In a review of 160 cases
Based on the appearance of the gross specimen and the of odontomas by Tekkesin et al., of all the investigated
histopathological examination a diagnosis of compound- cases, 99 were complex, 57 were compound, and only
complex odontoma was made, which is a rare entity. four were mixed odontomas. This further confirms
the rare occurrence of the mixed odontoma, that is the
compound - complex odontoma.[6]
Discussion
Although the etiology of odontomas remains unknown,
Rather than being true neoplasms, odontomas are local trauma, infection, family history and genetic
hamartomatous lesions or malformations. [2,3] They mutation have been suggested as possible causes.
constitute 22% of all odontogenic tumours of the jaw Compound odontomas usually do not cause any bony
making them the most common benign odontogenic expansion, whereas complex odontomas often cause
tumours.[4] Majority of the complex odontomas reported slight or even marked bony expansion. Pathological
in literature measure around 1-2 cm in diameter. changes such as impaction, malpositioning, aplasia,
However, in the present case, the odontoma measured malformation and devitalization of the adjacent teeth
about 3 × 3 cm, which is very rare. The anterior maxilla is can be caused by 70% of the odontomas.[7] The lesions
the common site of occurrence of compound odontomas, are usually asymptomatic and are invariably discovered
whereas the complex odontomas frequently occur in the on routine radiographic examinations. The radiological
posterior mandibular region.[2,4] appearance of odontomas depends on their stage of
development and degree of mineralization and is divided
into three stages.[2] Since the present case had a mixed
radiopaque radiolucent lesion, we considered the present
lesion to be in the intermediate stage of development.

Figure 5: Specimen after surgical removal Figure 6: Post-operative OPG

Figure 7: Histopathological section showing enamel and dentin-like Figure 8: Histopathological section showing haphazardly arranged
structure enamel and dentin

Journal of Indian Academy of Oral Medicine & Radiology | Oct-Dec 2014 | Vol 26 | Issue 4 465
[Downloaded free from http://www.jiaomr.in on Tuesday, February 14, 2023, IP: 182.253.133.211]

Khanum N et al.: Compound-complex odontoma

Conclusion 2. Reichart PA, Philipsen HP. Odontogenic Tumors and Allied

Lesions. 3rd ed. London: Quintessence; 2004. p. 141-53.
Cases of large complex odontomas erupting into the oral 3. Neville BW, Damm DD, Allen CM, Bouquot JF. Odontogenic cysts
and tumors. In: Neville BW, Damm DD, Allen CM, Bouquot JF.
cavity and also infected complex odontomas have been
Oral and Maxillofacial Pathology. 2 nd ed. Philadelphia: WB
reported. When odontomas occur in association with Saunders; 2002. p. 633-42.
missing teeth, it is a rare occurrence. Early diagnosis will 4. Wood NK, Goaz PW. Differential Diagnosis of Oral and
facilitate removal of the cause of eruption disturbances, Maxillofacial Lesions. 5th ed. Singapore: Harcourt Brace and Co
which is important in the developing dental arch. For Asia Pvt Ltd; 1998. p. 289-314.
early detection of odontomas, panoramic radiography 5. Prabhakar C, Haldavnekar S, Hegde S. Compound-Complex
and a proper clinical examination are important. odontoma — An important clinical entity. J Int Oral Health
Complications can be prevented by surgical excision 2012;4:49-53.
followed by histopathological analysis. A careful follow- 6. Soluk Tekkesin M, Pehlivan S, Olgac V, Aksakallı N, Alatli C.
up of the case, both clinically and radiographically Clinical and histopathological investigation of odontomas:
Review of the literature and presentation of 160 cases. J Oral
is necessary to assess the eruption of the unerupted
Maxillofac Surg 2012;70:1358-61.
or impacted teeth. The odontomas have a favorable
7. Kaneko M, Fukuda M, Sano T, Ohnishi T, Hosokawa Y.
prognosis with rare chances of relapse. Microradiographic and microscopic investigation of a rare case of
complex odontoma. Oral Surg Oral Med Oral Pathol Oral Radiol
References Endod 1998;86:131-4.

1. Shafer WG, Hine MK, Levy BM. Tumors and cysts of odontogenic How to cite this article: Khanum N, Shivalingu MM, Lingaraju N,
origin. In: Shafer WG, Hine MK, Levy BM, editors. A Textbook Basappa S. Compound-complex odontoma: A case report of a rare
of Oral Pathology. 4th ed. Philadelphia: WB Saunders Co; 1993. variant. J Indian Acad Oral Med Radiol 2014;26:463-6.
p. 258-317. Source of Support: Nil, Conflict of Interest: None declared.

466 Journal of Indian Academy of Oral Medicine & Radiology | Oct-Dec 2014 | Vol 26 | Issue 4