|Year : 2011 | Volume
| Issue : 6 | Page : 56-60
Solitary odontodysplasia: A rare entity
D Gurunathan1, S Tandon2, R Radha Krishnan3, M Kalra4
1 Department of Pedodontics, Saveetha Dental College, Chennai, India
2 Department of Pedodontics, Manipal College of Dental Sciences, Manipal, Karnataka, India
3 Department of Oral Pathology, Manipal College of Dental Sciences, Manipal, Karnataka, India
4 Department of Oral and Maxillofacial Pathology, SGT Dental College, Gurgaon, Haryana, India
|Date of Web Publication||12-Dec-2011|
Department of Pedodontics, Saveetha Dental College, 162, Poonamalle High Road, Chennai - 77
| Abstract|| |
Odontodysplasia is a rare, non-hereditary developmental anomaly affecting dental tissues derived from both the mesoderm and ectoderm which results in a bizarre clinical and radiographic appearance. Regional odontodysplasia describes the segmental and localized nature of the condition. Odontodysplasia occurring in a single tooth is a rare occurrence. A case of Solitary odontodysplasia in an eleven year and half old male whose chief complaint was the absence of eruption of permanent maxillary right central incisor teeth is presented. Clinical, radiographic and histological findings of a single tooth, odontodysplasia are described in this case report.
Keywords: Histological findings, radiological findings, solitary odontodyslasia, permanent tooth, single tooth odontodysplasia
|How to cite this article:|
Gurunathan D, Tandon S, Krishnan R R, Kalra M. Solitary odontodysplasia: A rare entity. J Indian Soc Pedod Prev Dent 2011;29:56-60
|How to cite this URL:|
Gurunathan D, Tandon S, Krishnan R R, Kalra M. Solitary odontodysplasia: A rare entity. J Indian Soc Pedod Prev Dent [serial online] 2011 [cited 2013 May 18];29:56-60. Available from: http://www.jisppd.com/text.asp?2011/29/6/56/90743
| Introduction|| |
Odontodysplasia is a rare, nonhereditary developmental anomaly affecting dental tissues derived from both the mesoderm and ectoderm, which results in a bizarre clinical and radiographic appearance. , The term "odontodysplasia" was coined by Zagarelli et al. in 1963 to describe a peculiar dental anomaly of an unknown etiology.  Pindborg used the term "regional odontodysplasiaregional odontodysplasia" (RO) in 1970 to describe the segmental and localized nature of the condition. 
RO affects both the primary and permanent dentition in the maxilla and mandible, or both jaws. Generally, it is localized to one arch, and the maxillary arch is affected more often than the mandibular arch. The lesions appear more commonly in anterior segment.  Though the condition most often affects only one quadrant, cases with bilateral or multiquadrant involvement have also been reported.  The criteria for diagnosis of RO are primarily clinical and radiographic, sometimes supplemented by histological features. 
Clinically, a tooth affected by RO appears hypocalcified and hypoplastic.  Radiographically, there is a lack of contrast between the enamel and dentin, both of which are less radiopaque than their unaffected counterparts. Additionally, enamel and dentin layers are thin, giving the teeth a "ghost-like" appearance. ,,,
The dental follicles and pulp chambers are noticeably enlarged and root apices are open. Teeth affected by odontodyspalsia must be observed over a period of time for eruption, and development of maxilla and mandible. The chosen method of treatment for each case should be performed from time to time as per the needs during long-term observation.
This paper reports a rare case of odontodysplasia of a single unerupted tooth. Since limited literature exists on such cases, the clinical and radiographic characteristics of odontodysplasia are discussed in this article, describing the anomaly with certain unique findings and treatment options that need to be considered with specific reference to pediatric patients. Informed consent to publish this article was obtained from the child and his parents.
| Case Report|| |
An11˝-year-old boy presented at the Pediatric Dentistry Department, Manipal College of Dental Sciences, Manipal, India, with the complaint of an unerupted upper right front permanent tooth. The patient's mother had a normal pregnancy and delivery; family histories regarding congenital disea. The patients' mother had a normal pregnancy and delivery. Family history regarding congenital disease and dental anomalies were unremarkable. Patient's medical history was noncontributory, with no congenital or acquired disease. According to the mother, there was a history of trauma to the upper anterior region of the child when he was 1-2 years old and the primary tooth corresponding to missing permanent tooth has turned mobile. This mobile primary right central incisor was extracted as advised by the dentist.
Extraoral examination revealed no significant abnormalities such as facial asymmetry, nevi or birth mark on the face. On intraoral examination, all teeth were normal with a good oral hygiene except for the missing upper right central incisor. The gingival tissue covering the missing tooth was bluish in color and softer than the adjacent area [Figure 1].
Periapical radiograph showed impacted permanent right upper central incisor surrounded by periapical radiolucency suggestive of an enlarged dental follicle. The enamel and dentin layer of the central incisor was very thin. The demarcation between them was not discernable and pulp chamber was wide, all of which gave rise to a "ghost-like" appearance. Root was short and apical end was distally curved [Figure 2].
|Figure 2: Panoramic radiograph showing normal development of maxillary and mandibular arch except maxillary right central incisor|
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Panoramic and occlusal radiograph revealed a delay in the development of the maxillary right central incisor as compared to the left central incisor. All other teeth exhibited normal thickness of enamel and dentin [Figure 3] and [Figure 4].
|Figure 3: Occlusal radiograph showing odontodysplasia of maxillary right central incisor|
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|Figure 4: Intraoral periapical radiograph showing odontodysplasia of maxillary right central incisor|
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The treatment advised was surgical removal of the maxillary right permanent central incisor. In the case of the patient described herein, the diagnosis was quite clear prior to surgery, based on the radiographic appearance and the age of the patient. The surgically removed tooth exhibited a crown that was hypoplastic with pitted enamel and a short root that had a distal curvature in the apical third [Figure 5].
The specimen, consisting of tooth and soft tissue, was sent for a histopathologic examination. Histological examination of the removed teeth showed irregular dentin with large areas of interglobular dentin and widened predentin. This confirmed the clinical diagnosis of odontodysplasia [Figure 6] and [Figure 7].
|Figure 6: Photomicrograph of decalcified dentin showing a mixture of interglobular dentin and amorphous material (×20)|
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|Figure 7: Photomicrograph showing follicular tissue surrounding the crown, exhibiting focal collection of basophilic enamel-enamel-like calcifications suggestive of disturbed enamel formation (×40)|
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After healing of the alveolar ridge, an esthetic and functional acrylic space maintainer was installed. The patient was kept under observation and periodic recall was advised to monitor the development of dentition and craniofacial growth [Figure 8].
| Discussion|| |
The case discussed here presents an interesting feature of odontodysplasia affecting only a single tooth. In general, this dental anomaly affects groups of teeth, , in a single quadrant with central and lateral incisors affected more frequently than the posterior teeth,  across the midline  or all the teeth may be involved.  This condition occurs in females more often than in males unlike in the present case.  The etiology of RO is uncertain and numerous factors have been suggested such as local trauma, infection, local ischemia, irradiation, activation of latent viruses in the odontogenic epithelium and can be associated with the presence of nevus, hemangiomas and hydrocephaly. , Although many theories have been suggested for the etiology of odontodysplasia, most of the cases reported do not attribute the occurrence to any specific cause. ,, Significantly, the present case gives a history of trauma to the corresponding primary tooth during the age of development of permanent central incisor, which could have resulted in the anomaly. This is in contrary to the case reported by Kappadia et al, which does not attribute trauma to the occurrence of RORO. In the permanent dentition, teeth affected by odontodysplasia usually are not erupted or can be partially erupted, which is similar to present case.  Gingival tissue in the affected area may appear enlarged, fibrous or may be hyperemic and associated with a fistula.  However, in this case, the gingiva covering the tooth was bluish in color and soft in texture, indicating accumulation of venous blood. Although dentinal dysplasia, amelogenesis and dentinogenesis imperfecta show some similarities to RO, these conditions affect the entire dentition in contrast to the segmental involvement seen in RO.  In the current case,, the presence of pericoronal radiolucency represents an enlarged follicle similar to that reported by Gardner in in 1973.  On the other hand, radiographically, unerupted teeth affected by RO may be incorrectly diagnosed as undergoing root resorption or being an odontoma.  A dilacerated dilacerated tooth appears coronally foreshortened on a radiograph unlike an odontodysplastic tooth.  But in the present case, all the radiographic features of right permanent central incisor gave a clear picture of odontodysplasia i.e. a "ghost tooth" "ghost tooth" appearance. The present case was differentiated from Turners hypoplasia in that tooth affected by Tuners hypoplasia has a local enamel hypoplastic defect in the crown, and radiographically only the enamel might be affected with normal dentin and pulp chambers.  The anomalous tooth was localized in a single quadrant, had hypoplastic crown, delayed eruption and "ghost tooth appearance" on radiograph, all of which are typical features of odontodysplasia.
Treatment of odontodysplasia has given rise to controversy. These cases require a continuous and multidisciplinary approach. Consultations between pedodontic, prosthodontic and orthodontic specialties are are necessary in the case of odontodysplasia. Most clinicians advocate extracting the affected teeth as soon as possible and inserting a prosthetic replacement , because the longer the affected teeth are retained, the higher are the instances for development of pathologies pathologies like formation of follicular cysts or odontogenic tumors. ,,, On the contrary, some prefer to retain and conservatively manage these teeth until skeletal growth is complete as long as they are free of infection.  The non-infected teeth help maintain the alveolar bone, avert the need for a removable prosthesis and eliminate the psychological effects of premature tooth loss. , It has been proposed that orthodontic traction is effective for odontodysplastic tooth when the root development of the impacted teeth progresses to a reasonable extent.  However, selection of method and timing appear to be critical factors in the treatment of odontodysplasia.
In the present case, when the affected tooth was compared with the corresponding tooth on the opposite side, the calcification of the affected tooth was less and the tooth had an underdeveloped root. Hence, waiting for the eruption of the affected tooth was considered to be unfruitful. Orthodontic extrusion of the tooth in the current case is difficult because of atypical shape of crown and hypoplastic nature,, which make it difficult to bond the orthodontic brackets to the tooth.  Moreover, because of the short root, orthodontic forces must be very low to avoid root resorption and undermining bone resorption.  Even if the tooth erupts, the malformed morphology of the tooth will be uneathetic. Therefore, surgical removal of the affected tooth followed by replacement of the missing tooth with removable acrylic prosthesis and later by permanent rehabilitation was a preferred method in this case. Parents of the patient were concerned about the delayed eruption, waiting period for the eruption and unesthetic appearance, hence immediate replacement of the tooth was insisted by the parents.
During surgical exposure, the tooth was visible as soon as the soft tissue was excised. This could be could be attributed to the presence of a large follicle which was similar to the case reported by Hamdan.  The extracted tooth exhibited a small crown with yellowish brown, hypoplastic pitted enamel with an irregular incisal surface.  The enamel was soft in consistency and was easily removed by probe.  The root of the affected tooth was small and incompletely formed with curvature in apical third, which is similar to that reported by Futabo.  The provisional diagnosis of odontodysplasia of a single tooth was confirmed by histological examination of the tooth.
Histologically, follicular tissue surrounding the crown, exhibiting focal collection of basophilic enamel-enamel-like calcifications suggestive of disturbed enamel formation was seen. Decalcified dentin showing a mixture of interglobular dentin and amorphous material was also identified. 
An acrylic removable partial denture was given to maintain esthetics, achieve space preservation, and to avoid supraeruption of opposing tooth and lessen the psychological trauma of early tooth loss. An implant was proposed after completion of pubertal growth, but with care taking into consideration the low-low-density bone around the affected tooth. , The patient was placed on periodic recall to monitor the growth and development of the maxilla which was satisfactory.
This case report is considered unique and rare because it presents with the condition affecting only one tooth unlike cases reported by previous authors. Hence, the term "solitary odontodysplasia" is suggested. Another interesting feature of the present case is that local trauma might be an attributable etiology for the solitary odontodysplasia.
| Conclusion|| |
The presentation of this case helps pediatric dentists to review special clinical and radiographic features of odontodysplasia. The treatment of odontodysplasia depends on the degree of dental involvement and is performed in accordance with the functional and esthetic needs of each case. In addition,, it is important to provide continuous dental supervision and case management.
| References|| |
|1.||Hamdan MA, Sawair FA, Rajab LD, Hamdan AM, Al-Oamri IK. Regional odontodysplasia: A review of the literature and report of a case. Int J Paediatr Dent 2004;14:363-70. |
|2.||Cahuana A, Gonzalez Y, Palma C. Clinical management of regional odontodysplasia. Pediatr Dent 2005;27:34-9. |
|3.||Zegarelli EV, Kutscher AH, Applebaum E, Archard HO. Odontodysplasia. Oral Surg Oral Med Oral Pathol 1963;16:187-93. |
|4.||Pindborg JJ. Pathology of Dental Hard tissues. Copenhaen: Munkgard; 1970. p. 120-3. |
|5.||Lustmann J, Klein H, Ulmansky M. Odontodysplasia. Report of two cases and review of the literature. Oral Surg Oral Med Oral Pathol 1975;39:781-93. |
|6.||Vaikuntam J, Tatum NB, McGuff HS. Regional odontodysplasia: Review of the literature and report of a case. J Clin Pediatr Dent 1996;21:35-40. |
|7.||Spini TH, Sargenti-Neto S, Cardoso SV, Souza KC, de Souza SO, de Faria PR, et al. Progressive dental devolpment in regional odontodysplasia. Oral Surg Oral Med Oral Path Oral Radiol Endod 2007;104:40-5. |
|8.||Ozer L, Cetiner S, Ersoy E. Regional odontodysplasia: Report of a case. J Clin Pediatr Dent 2004;29:45-8. |
|9.||Pandis N, Polido C, Bell WH. Regional odontodysplasia. A case associated with asymmetric maxillary and mandibular development. Oral Surg Oral Med Oral Pathol 1991;72:492-6. |
|10.||Sadeghi EM, Ashrathi MH. Regional odontodysplasia. Clinical, pathological and theraputic considerations. J Am Dent Assoc 1981;102:336-9. |
|11.||Kahn MA, Hinson RL. Regional odontodysplasia. Case report with etiologic and treatment considerations. Oral Surg Oral Med Oral Pathol 1991;724627. |
|12.||Rosa MC, Marcelino GA, Belchior RS, Souza AP, Parizotto SC. Regional odontodisplasia: Report of case. J Clin Pediatr Dent 2006;30:333-6. |
|13.||Crawford PJ, Aldred MJ. Regional odontodysplasia: A bibliography. J Oral Pathol Med 1989;18:251-63. |
|14.||Hanks PA, Williams B. Odontodysplasia: Report of two cases. Pediatr Dent 1998;20:199-203. |
|15.||Oncag O, Eronat C, Sen BH. Regional odontodysplasia: A case report. J Clin Pediatr Dent 1996;21:41-6. |
|16.||Kappadi D, Ramasetty PA, Rai KK, Rahim AM. Regional odontodysplasia: An unusual case report. J Oral Maxillofac Pathol 2009;13:62-6.J Oral Maxillofac Pathol 2009;13:62-6. |
|17.||Ansari G, Reid JS, Fung DE, Creanor SL. Regional odontodysplasia: Report of four cases. Int J Paediatr Dent 1997;7:107-13. |
|18.||Cho S-Y. Conservative management of regional odontodysplasia: Case report. J Can Dent Assoc 2006;72:735-8. |
|19.||Gardner DG, Sapp JP. Regional oontodysplasia. Oral Surg 1973;35:351-65. |
|20.||Amado Cuesta S, Gargallo Albiol J, Berini Ayte´s L, Gay Escoda C. Review of 61 cases of odontoma. Presentation of an erupted complex odontoma. Med Oral 2003;8:366-73. |
|21.||Asokan S, Rayen R, Muthu MS, Sivakumar N. Crown dilacerations of maxillary right permanent central incisor-A case report. J Indian Soc Pedod Prev Dent 2004;22:197-200. |
|22.||Goaz PW, White SC. Oral radiology-principles and interpretation. 2nd ed. St. Louis: Mosby Co.; 1987. p. 433-5. |
|23.||Yuan SH, Liu PR, Childers NK. An alternative restorative method for regional odontodysplasia: Case report. Pediatr Dent 1997;19421-4. |
|24.||Courson F, Bdeoui F, Danan M, Degrange M, Gogly B. Regional odontodysplasia: Expression of matrix metalloproteinases and their natural inhibitors. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:60-6. |
|25.||Gomes MP, Modesto A, Cardoso AS, Hespanhol W. Regional odontodysplasia: Report of a case involving two separate affected areas. ASDC J Dent Child 1999;66:203-7. |
|26.||Melamed Y, Harnik J, Becker A, Shapira J. Conservative multidisciplinary treatment approach in an unusual odontodysplasia. ASDC J Dent Child 1994;61:119-24. |
|27.||Fearne J, Willias DM, Brook AH. Regional odontodysplasia: A clinical and histological evaluation. J Int Assoc Dent Child 1986;17:21-5. 1986;17-21. |
|28.||LIazwa F. Regional odontodysplasia: Long term observation of a case on the mandibular left side. Pediatr Dent J 2010;20:103-9.Pediatr Dent J 2010:20:103-9. |
|29.||Tervonen SA, Stratmann U, Mokrys K, Reichart PA. Regional odontodysplasia: A review of the literature and report of four cases. Clin Oral Investig 2004;8:45-51. |
|30.||Van der Wal JE, Rittersma J, Baart JA, van der Waal I. Regional odontodysplasia: Report of three cases. Int J Oral Maxillofac Surg Surg1993;22:356-8. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]