The Saudi Dental Journal
Volume 22, Issue 3 , Pages 145-149, July 2010

Compound composite odontoma with unusual number of denticles – A rare entity

  • Urvashi Sharma

      Affiliations

    • Department of Pedodontics and Preventive Dentistry, Dr. Harvansh Singh Judge Institute of Dental Sciences and Hospital, Punjab University, Sector 25, Chandigarh, India
    • Corresponding Author InformationCorresponding author. Address: House No. 433, Sector 37-A, Chandigarh, India. Tel.: +91 172 2691433; fax: +91 172 2724912.
    • ,
  • Rahul Sharma

      Affiliations

    • Department of Oral and Maxillofacial Surgery, Dr. Harvansh Singh Judge Institute of Dental Sciences and Hospital, Punjab University, Sector 25, Chandigarh, India
    • ,
  • Anubha Gulati

      Affiliations

    • Department of Oral Pathology, Dr. Harvansh Singh Judge Institute of Dental Sciences and Hospital, Punjab University, Sector 25, Chandigarh, India
    • ,
  • Renu Yadav

      Affiliations

    • Department of Oral Pathology, Dr. Harvansh Singh Judge Institute of Dental Sciences and Hospital, Punjab University, Sector 25, Chandigarh, India
    • ,
  • Krishan Gauba

      Affiliations

    • Department of Pedodontics and Preventive Dentistry, Dr. Harvansh Singh Judge Institute of Dental Sciences and Hospital, Punjab University, Sector 25, Chandigarh, India

Received 5 September 2009; received in revised form 7 November 2009; accepted 25 December 2009.

Article Outline

Abstract 

Compound odontoma is stated to be a hamartomatous lesion rather than a true odontogenic tumour. It has an unknown etiology and often suspected when there are retained deciduous teeth in children. Early detection and surgical enucleation of the tumour is recommended to prevent impaction of unerupted teeth. In this index case, multiple denticles or rudimentary teeth, numbering 37 were enucleated from the maxillary anterior region of a 17-year old male, which makes this case unusual. Evidence of concrescence, fusion and dilaceration were observed in the denticles enucleated, the size of which varied from 4mm to 12.5mm.

Keywords: Odontoma, Compound odontoma, Odontogenic tumours, Denticles

 

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1. Introduction 

Odontomas are mixed odontogenic tumours in which both the epithelial and mesenchymal components undergo functional differentiation and form enamel and dentin (Budnick, 1976, Shafer, 1993). They are hamartomatous lesions rather than true neoplasms (Budnick, 1976, Shafer, 1993) WHO classifies odontomas into: compound and complex. The compound odontoma is a malformation in which all the dental tissues are in a more orderly pattern than in the complex odontoma so that the lesion consists of many tooth-like structures. In each tooth-like structure, enamel, dentin, cementum and pulp are arranged as in the normal tooth (Budnick, 1976, Shafer, 1993, Philipsen et al., 1997, Neville et al., 1995).

In a literature review of 38 cases of compound odontoma by Amado-Cuesta et al. the number of denticles varied from 4 to 28. The sheer number of denticles extracted, numbering 37, makes the present case unique as such a high number has not been reported before. The extracted denticles exhibited concrescence, fusion and dilaceration.

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2. Case history 

A 17-year old boy reported with the complaint of a small upper right tooth associated with painless, hard swelling above it. An intra-oral examination revealed a retained deciduous right lateral incisor. A well-defined hard, bony swelling was seen in relation to the retained deciduous tooth on both the labial and palatal aspect (Fig. 1). It was non-tender and the overlying gingiva was of normal colour and consistency. The medical and family history was non-contributory.

An intra-oral periapical radiograph and an upper anterior occlusal film (Fig. 2) revealed multiple tooth-like structures of different shapes and sizes in relation to the root of the deciduous maxillary right lateral incisor. They were surrounded by a well-circumscribed radiolucent zone. The permanent maxillary right lateral incisor was displaced and lay labially in relation to the apical third of the root of permanent maxillary right central incisor.

  • View full-size image.
  • Figure 2. 

    Maxillary anterior occlusal radiograph showing the compound odontoma, the retained deciduous right lateral incisor and the unerupted permanent right lateral incisor.

Upon the clinical and radiographic findings, a provisional diagnosis of a compound composite odontoma was made. It was decided to extract the deciduous maxillary right lateral incisor and to surgically enucleate the tumour.

Under local anaesthesia, the deciduous maxillary right central incisor was extracted. A labial mucoperiosteal flap was elevated, the thin bone overlying the odontoma was removed.

Numerous denticles, numbering 37, were enucleated with their fibrous capsule. Evidence of concrescence was seen in six groups of denticles. Fusion and dilaceration was also noticed in some (Fig. 3). The size of the denticles varied from 4mm to 12.5mm. The unerupted permanent right lateral incisor was noticed (Fig. 4). The flap was closed and healing was uneventful.

  • View full-size image.
  • Figure 3. 

    Enucleated multiple rudimentary tooth-like structures with a soft-tissue lining along with the extracted deciduous right lateral incisor on the top left corner.

Histopathologically, the sections revealed miniature teeth containing dentin with dentinal tubules, pulp, cementum and periodontal ligament-like tissue (Figure 5, Figure 6). The enamel cap on the denticles was removed during decalcification. Most of these denticles had a single root and a root canal. Some of them were surrounded by a compressed connective tissue stroma.

By correlating the clinical, radiological and histopathological findings, a definite diagnosis of compound odontoma was made. It was arranged for the patient to follow-up the management of the impacted permanent lateral incisor.

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3. Discussion 

Odontomas are classified as benign, mixed, calcified odontogenic tumours. The absolute incidence of odontogenic tumours varies from 0.002% to 0.1% (Yeung et al., 2003) out of which odontomas constitute about 22% of all odontogenic tumours of the jaws (Saadettin et al., 2007, Cildir et al., 2005) Paul Broca had first described odontoma in 1867. However, literature includes odontomas in a separate category of developmental malformations (Budnick, 1976, Shafer, 1993, Neville et al., 1995, Yeung et al., 2003, Soames and Southam, 1998).

Although the etiology of odontoma is unknown (Shafer, 1993), yet cases have related odontoma to local trauma (Saadettin et al., 2007), infection (Saadettin et al., 2007) and genetics (Cildir et al., 2005). It arises from an exuberant proliferation of the dental lamina or its remnants and is termed laminar odontome (Chuong and Kaban, 1985) or forms as a result of multiple schizodontia i.e. a locally conditioned hyperactivity of dental lamina (Philipsen et al., 1997). It may also be associated with the Gardner’s syndrome of intestinal polyposis (Cawson et al., 2001) and the rare odontoma-dysphagia syndrome (Bader, 1967). Compound odontomas are twice as commonly observed as the complex (Neville et al., 1995) and commonly lie in the maxillary incisor-canine region.

Odontomas may have a racial predilection with a higher reported incidence of about 65% of all odontogenic tumours in Caucasians (Regezi et al., 1978) and only 6–6.7% in the Chinese (Yeung et al., 2003). The incidence is low in the Africans (Ladeinde et al., 2005) where as the majority of the odontogenic tumours in North America are the odontomas (Regezi et al., 1978). This could be suggestive of genetic or environmental susceptibility of the tumour or could be related to insufficient resources to detect asymptomatic cases (Marcela et al., 2007).

Odontomas are often seen in the permanent dentition and very rarely associated with the primary teeth (Katz, 1989, Tomizawa et al., 2005). A mean age of detection at 14.8years for compound odontoma and 16years for complex odontoma was found (Budnick, 1976). Compound odontomas are equally distributed among males and females whereas the complex one has a 60% predilection for women (Neville et al., 1995, Kaugers et al., 1989).

Radiographically, odontoma appears as an irregular radio-opacity or denticles surrounded by a radiolucency with or without a bony expansion. There is usually a retained deciduous tooth associated with an unerupted permanent tooth. The patient is usually asymptomatic.

Odontomas often come to clinical attention by causing over-retention, impaction (Saadettin et al., 2007, Kamakura et al., 2002) and delayed eruption of both primary (Yeung et al., 2003, Cildir et al., 2005, Tomizawa et al., 2005, Singh et al., 2005) and permanent teeth (Budnick, 1976, Kaugers et al., 1989). Despite interfering with eruption, there is usually no resorption of adjacent tooth roots. Kaugers in a series of cases found 48% of odontomas in association with an unerupted tooth and 28% cases in conjunction with a dentigerous cyst (Kaugers et al., 1989). Cases of devitalization of adjacent teeth with compound odontomas have also been reported (Saadettin et al., 2007). Erupted odontomas (Amailuk and Grubor, 2008) can become carious and lead to abscess formation (Kaban and Troulis, 1990). Odontomas are usually well-encapsulated. Recurrence is usually not observed if the lining epithelium is removed intact (Kaban and Troulis, 1990).

In a review of 38 cases of compound odontoma, the number of denticles varied from 4 to 28 (Amado-Cuesta et al., 2003). The sheer number of denticles extracted, numbering 37, makes this case unique as such a high number has not been reported before. The extracted denticles exhibited concrescence, fusion and dilaceration. Timely detection and surgical enucleation of odontoma followed by curettage is recommended to prevent complications such as over-retention, impaction and delayed eruption of permanent teeth.

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References 

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PII: S1013-9052(10)00047-7

doi:10.1016/j.sdentj.2010.04.009

The Saudi Dental Journal
Volume 22, Issue 3 , Pages 145-149, July 2010

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