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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 6  |  Issue : 2  |  Page : 61-64

Compound odontome with 64 denticles: An exceptional case report


1 Department of Pedodontics and Preventive Dentistry, Dr. R. Ahmed Dental College and Hospital, Kolkata, West Bengal, India
2 Department of Dentistry, Burdwan Medical College and Hospital, Bardhaman, West Bengal, India

Date of Submission29-Jul-2020
Date of Acceptance10-Aug-2020
Date of Web Publication31-Oct-2020

Correspondence Address:
Dr. Shiladitya Sil
Burdwan Medical College and Hospital, Khosbagan, Purba Bardhaman - 713 104, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohr.ijohr_17_20

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  Abstract 


Odontomes are among the most common odontogenic tumors that consists of dental tissues organized in a disorderly pattern. The World Health Organization classification defines odontome as malformations in which all the dental tissues are represented. Here, we report a case of 12-year-old male who reported with chief complaint of a swelling in the upper right front tooth region since 4 months. Clinically, a diffuse, hard swelling was appreciated. Radiographic examination revealed irregular heterogeneously radio-opaque mass with peripheral radiolucency. Root resorption of 15 and impaction of 13 and 14 was noted along with missing lateral incisor. Surgical enucleation was done and 64 denticles were removed. No recurrence was reported. Most odontomes are asymptomatic and detected during routine dental examination. Hence, addressing the patient's primary complaint and conducting proper clinical and radiological examination may help in the early detection and intervention of such lesions with minimal complication.

Keywords: 64 denticles, compound odontomes, missing lateral incisor, routine examination


How to cite this article:
Saha N, Sil S, Ghosh S. Compound odontome with 64 denticles: An exceptional case report. Indian J Oral Health Res 2020;6:61-4

How to cite this URL:
Saha N, Sil S, Ghosh S. Compound odontome with 64 denticles: An exceptional case report. Indian J Oral Health Res [serial online] 2020 [cited 2021 Dec 6];6:61-4. Available from: https://www.ijohr.org/text.asp?2020/6/2/61/299702




  Introduction Top


Paul Brocain was the first person to describe odontomes in 1867.

The World health organization (WHO) histological typing of odontogenic tumors classifies odontoma under benign odontogenic tumors containing ectomesenchyme, with or without dental hard tissue formation. Odontomes are regarded as hamartomas, rather than neoplasm.[1]

Several factors have been associated in the pathogenesis of odontomes such as mutant gene, focal infection, trauma, and Gardener's Syndrome. Although there is acceptable maturation of ameloblasts and odontoblasts, due to abnormal morphodifferentiation, the pattern of laying down enamel and dentin is disturbed resulting in the formation of odontomes.[2]

Odontomes are classified into complex and compound variants. In complex odontome, there is disorganized orientation of enamel, dentin, and cementum and in compound odontome there is an organized orientation of the same.[3]

Commonly seen in second to third decade of life, complex odontomes are prevalent among the males in the posterior region of mandible while compound odontomes are usually seen among females in anterior maxilla.[3],[4]

A rare case of compound odontome associated with missing maxillary lateral incisor (LI) and more than 60 teeth like structures is reported here.


  Case Report Top


A 12-year-old male reported to our outpatient department with swelling in the upper right front tooth region since the past 4 months, without any history of pain or sudden increase in size. He also complained of delayed eruption of the corresponding permanent tooth in that region. There was no relevant medical/family history and no history of trauma. Proper immunization schedule was followed. Extraction of 53 and 54 was done about a year back secondary to gross mutilation.

On intraoral examination, mild rotation was appreciated in 11, missing 12, 13, 14 with erupted 15, 16, and 17. On inspection, diffuse swelling was seen involving 12, 13, 14 region with loss of vestibular depth. Overlying mucosa appeared thinned out without any ulcerations or discharging sinus. On palpation, a bony hard mass was appreciated with diffuse outline and irregular texture. No tenderness was elicited [Figure 1]. Intraoral periapical radiograph (IOPAR), true maxillary occlusal radiograph, and orthopantomogram (OPG) were advised.
Figure 1: The initial presentation of the swelling with missing 12, 13, and 14

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IOPAR revealed heterogeneously radio-opaque mass with irregular outline in relation to 12, 13 and 14 region. The occlusal radiograph revealed similar findings along with impacted 13 [Figure 2]. OPG revealed an irregular heterogeneous radiopacity resembling multiple teeth like structures with peripheral radiolucent band. Impacted 13, 14 could be appreciated in the maxillary sinus. The lesion caused the rotation of 11 and partial root resorption of 15 [Figure 3]. A provisional diagnosis of compound odontome was given.
Figure 2: The intraoral periapical radiograph (left) and true maxillary occlusal radiograph (right). Radiographs reveal a heterogeneously radio-opaque mass with irregular outline and a thin radiolucent band around the lesion

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Figure 3: The orthopantomogram that revealed an irregular calcified mass with heterogeneous radiopacity resembling multiple teeth like structures with peripheral radiolucent band. Impacted 13, 14 could be appreciated in the maxillary sinus

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Surgical excision was planned after obtaining informed consent. A mucoperiosteal flap was raised in 12, 13, 14 region, a bone window was made [Figure 4] and approximately 64 teeth like calcified masses were removed [Figure 5]. Complete enucleation was performed. Postoperative healing was uneventful. No recurrence was appreciated on subsequent follow-ups.
Figure 4: The irregular white lesion exposed during the procedure (left) and the approximated margins of the lesion by cat gut suture (right)

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Figure 5: The enucleated denticles and tooth-like structures

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  Discussions Top


Among the odontogenic tumors, odontomes constitute about 22% making them one of the most common jaw tumors. The incidence of compound odontome ranges between 9% and 37% and complex odontome between 5% and 30%.[4],[5]

In the WHO classification, the lesions that contain enamel and dentin of normal appearance are-ameloblastic fibro-odontome (AFO), ameloblastic odontome (AO), complex and compound odontomes.[6]

Slootweg commented that there was considerable doubt as whether AFO, AO, and Complex Odontomes each represent a separate entity or stages of maturation with complex odontome being the end point for which they manifest at an advanced age.[7]

Compound odontomes occur due to hyperactivity of the dental lamina, thus manifesting in younger individuals. In our case report, the extraction of 53, 54 could have triggered hyperactivity of the dental lamina. The mean age of detection of complex odontomes is 20.3 years while for compound odontomes, it is 14.8 years.[7]

Odontomes are known to be associated with over retention or early exfoliation of the deciduous teeth and/or delayed eruption of the permanent teeth, impaction, pathological tooth displacement with or without root resorption. Our case report was associated with displaced 11, root resorption of 15, delayed eruption and missing LI.[8]

Most of the reported odontomes are usually seen on the right side, including our case report. No explanation could be obtained from literature regarding this particular finding.[2],[3]

Odontomes are asymptomatic and discovered during radiographic examination after patient's primary complaint. However, sometimes may be associated with pain, bony expansion, facial asymmetry.[8] This case report was associated with slight disfigurement but no pain.

Rarely, odontomes lead to pathological developments such as dentigerous cyst and calcifying odontogenic cyst. There are no prior signs that can warn clinicians but when cystic changes take place, the odontome usually have faster growth potential and tends to attain huge size.[9] No cystic development was appreciated in our case report.

The presence of missing LI in compound odontomes is a unique entity. Literature reports that trauma in the form of dental extraction can cause dilaceration and malformed permanent tooth.[10] Similar history of prior extraction was reported in our case that could have contributed to missing LI.

In this case report, 13, 14 was impacted and was expected to erupt following removal of the lesion. However, follow-up is necessary to monitor the tendency of the impacted tooth to erupt. If they fail to erupt after 3 months, surgical exposure of the teeth may be considered with or without orthodontic traction.[11]

Surgical exposure followed by complete enucleation is the preferred management of odontomes. Care should be taken to preserve the surrounding vital structures. Odontomes have minimum recurrence rate following complete removal.

There is great variation regarding the number of denticles removed from odontomas. Literature reports an average of 4–37 denticles per lesion; although as many as 232 denticles have also been reported.[12] Around 64 denticles were enucleated from our patient which along with the missing LI makes this case report a unique one.

Being asymptomatic, most odontomes remain undetected. Clinicians are advised to meticulously perform routine dental examination, especially in children with eruption disturbances, impaction, and pathological tooth displacement with root resorption, to facilitate early detection and minimize the postoperative interventions.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Kramer IR, Pindborg JJ, Shear M. The WHO histological typing of odontogenic tumours. A commentary on the Second Edition. Cancer 1992;70:2988-94.  Back to cited text no. 1
    
2.
Rajendran R, Sivapathasundharam B. Shafer's Textbook of Oral Pathology. 6th ed. India: Elsevier; 2009.  Back to cited text no. 2
    
3.
Satish V, Maganur C Prabhadevi, Sharma R. Odontome: A brief review. Int J Clin Ped Dent 2011;4:177-85.  Back to cited text no. 3
    
4.
Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors: Analysis of 706 cases. J Oral Surg 1978;36:771-8.  Back to cited text no. 4
    
5.
Philipsen HP, Reichart PA, Praetorius F. Mixed odontogenic tumors and odontomas. Considerations on interrelationship. Review of literature and presentation of 134 new cases of odontomas. Oral Oncol 1997;33:86-7.  Back to cited text no. 5
    
6.
Pindborg JJ, Kramer IR, Torloni H. Histological typing of odontogenic tumors, jaw cysts and allied lesions. In: International Histological Classification of Tumors. Geneva: World Health Organization; 1970. p. 29-30.  Back to cited text no. 6
    
7.
Slootweg PJ. An analysis of the interrelationship of the mixed odontogenic tumors–ameloblastic fibroma, ameloblastic fibro-odontoma, and the odontomas. Oral Surg Oral Med Oral Pathol 1981;51:266-76.  Back to cited text no. 7
    
8.
Zerener T, Sencimen M, Bayar GR, Kan B, Ayberk H, Arici G. Immediate reconstruction using autogenous bone graft in huge compound odontome. Oral Health Dent Manag 2015;15:243-6.  Back to cited text no. 8
    
9.
Kim KS, Lee HG, Hwang JH, Lee SY. Incidentally detected odontoma within a dentigerous cyst. Arch Craniofac Surg 2019;20:62-5.  Back to cited text no. 9
    
10.
Amailuk P, Grubor D. Erupted compound odontoma: Case report of a 15-year-old Sudanese boy with a history of traditional dental mutilation. Br Dent J 2008;204:11-4.  Back to cited text no. 10
    
11.
Amado Cuesta S, Gargallo Albiol J, Berini Aytés L, Gay Escoda C. Review of 61 cases of odontoma. Presentation of an erupted complex odontoma. Med Oral 2003;8:366-73.  Back to cited text no. 11
    
12.
Pokharel S, Li Z. Teen Gets 232 “Teeth” Removed in Mumbai. Available from: http://edition.cnn.com/2014/07/25/world/asia/indiaabnormal-teeth/. [Last retrieved on 2014 Jul 25].  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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