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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 5  |  Issue : 3  |  Page : 195-197

Regional odontodysplasia in the primary dentition associated with eruption failure


1 Department of Pedodontics and Preventive Dentistry, Dr. R. Ahmed Dental College and Hospital, Kolkata, West Bengal, India
2 Department of Orthodontics, Dr. R. Ahmed Dental College and Hospital, Kolkata, West Bengal, India
3 Department of Oral and Maxillofacial Pathology, Dr. R. Ahmed Dental College and Hospital, Kolkata, West Bengal, India

Date of Web Publication5-Aug-2016

Correspondence Address:
Dr. Santanu Mukhopadhyay
Department of Pedodontics and Preventive Dentistry, Dr. R. Ahmed Dental College and Hospital, Kolkata - 700 014, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-344X.187838

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  Abstract 

Regional odontodysplasia (RO) is an uncommon nonhereditary developmental anomaly of dental hard tissues derived from ectoderm and mesoderm. The condition is more common in females, may affect primary and permanent dentitions with the maxilla, involves twice as frequently as mandible. The exact etiology of RO is unknown. Diagnosis is usually made by clinical and radiographic findings, sometimes supplemented with histopathologic examination. Clinically, teeth are hypoplastic with surface pits and grooves, and have brownish or yellowish discoloration. On the radiograph, there is reduced radiodensity of enamel and dentin with a lack of contrast between them. In this article, we described a case of a 3½-year-old girl with RO. The left maxillary quadrant was affected. The patient also showed eruption failure. Treatment of RO is, to a great extent, individualized. As the condition was asymptomatic, the present case was managed conservatively.

Keywords: Ghost teeth, primary dentition, regional odontodysplasia


How to cite this article:
Mukhopadhyay S, Roy P, Halder M. Regional odontodysplasia in the primary dentition associated with eruption failure. Int J Health Allied Sci 2016;5:195-7

How to cite this URL:
Mukhopadhyay S, Roy P, Halder M. Regional odontodysplasia in the primary dentition associated with eruption failure. Int J Health Allied Sci [serial online] 2016 [cited 2021 Feb 27];5:195-7. Available from: https://www.ijhas.in/text.asp?2016/5/3/195/187838


  Introduction Top


Regional odontodysplasia (RO) also known as odontogenesis imperfecta, odontogenic dysplasia, and nonhereditary amelogenesis imperfecta, and ghost teeth is an uncommon localized developmental anomaly of teeth in which dental tissues derived from both the ectoderm and mesoderm are affected. Hatchin first reported this anomaly, and McCall and Wald in 1947 described the condition reporting only the radiographic features. [1],[2] In 1954, Rushton coined the term "shell teeth" to indicate the radiological features of this disease. [3] In addition, Zegarelli et al. used the term "odontodysplasia" and Pindborg added the prefix "regional" indicating that the condition frequently affects a segment or a region of a dental arch. [4]

The prevalence of this rare anomaly is not known. Till 2013, approximately 140 cases appeared in literature. [5] The condition is more common in females with no known racial or ethnic predilection. In this developmental anomaly, several teeth may be affected, although the degree of involvement may vary. Diagnosis of this nonhereditary disorder is made by clinical as well as radiographic examination and, sometimes, supplemented with microscopic examination of the affected teeth. [5],[6],[7],[8],[9],[10],[11],[12],[13] Clinically, teeth involved with RO are small, hypoplastic with surface pits, and grooves, and have brownish or yellowish discoloration. [10] The thin enamel is susceptible to caries and fracture. The eruption of teeth is often delayed or does not occur at all. Gingival swellings and periapical infection are also frequent findings of this disease. [11] Radiographically, affected teeth demonstrate decreased radiodensity of enamel and dentin, wide pulp chambers, short roots, and open apices. [12] Histopathologic characteristics of RO show hypomineralized and hypoplastic enamel, mixed areas of cellular, amorphous, and interglobular dentin. [6],[7],[8],[9] Cementum is relatively unaffected.

The present article describes clinical and radiographic findings of RO in a case of a 3½-year-old girl involving the left maxilla associated with eruption failure of several teeth.


  Case Report Top


A 3½-year-old girl reported to our department with the chief complaint of noneruption of upper teeth. There was no history of trauma, and her prenatal, birth, and family history were not significant. Extraoral examination of the patient showed a bilaterally symmetrical face. On intraoral examination, the patient presented with primary dentition. The mandibular dentition appeared normal as was the teeth on the maxillary right quadrant. On the left quadrant of the maxilla, a hypoplastic, partially erupted primary central incisor and the tip of the adjacent lateral incisor were seen [Figure 1]. Posteriorly, the occlusal surface of the left maxillary primary second molar was visible. Oral hygiene was fair, and no soft-tissue abnormality was detected.
Figure 1: Intraoral photograph of the patient showing hypoplastic left maxillary primary central incisor along with the tip of the adjacent lateral incisor

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Panoramic radiograph of the patient was taken. It revealed that all teeth in the maxillary left quadrant had thin faint outlines with a lack of contrast between the enamel and dentin. There was a reduced radiopacity of enamel and dentin giving rise to ghost-like appearance [Figure 2]. Radiographic examination indicated that the underlying permanent successors also had poor outlines with decreased radiopacity compared to the unaffected side.
Figure 2: Panoramic radiograph showing reduced radiodensity of enamel and dentin of teeth involved with regional odontodysplasia compared to the unaffected contralateral side

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Based on clinical and radiographic findings, a diagnosis of RO was made. The condition was explained to the parents. As the condition was asymptomatic, a conservative treatment approach was followed. Oral hygiene instructions were given, and regular follow-up visits were advised to monitor eruption of teeth.


  Discussion Top


The etiology of RO is not clearly known although several factors such as infection, trauma, circulatory disorder, teratogenic drugs, Rh incompatibility, neural damage, local somatic mutation, hyperpyrexia, nutritional deficiency, and idiopathic factors have been proposed. In addition, this anomaly has been found to be associated with hemangioma, epidermal nevus, vascular nevi, ectodermal dysplasia, hydrocephalus, hypophosphatasia, and gingival swelling. [6],[7],[8],[9],[10],[11],[12],[13],[14] In the present case, no definite cause was found.

RO may occur in the primary or permanent dentition with maxilla involves twice as frequently as the mandible. The condition is usually unilateral, rarely crosses the midline, and is more common in the anterior dentition. In most cases, the central and lateral incisors are involved. In addition, teeth may be affected in different degrees even in the same arch. In the present case, both dentitions on the maxillary left quadrant were affected. The involved teeth in RO are generally discolored, hypoplastic, or hypomineralized, and may be associated with gingival swelling or even abscess formation. Delayed or failure of eruption of teeth is also reported. [7],[8],[9],[10],[11],[12],[13] In the present case, the left primary maxillary central incisor was hypoplastic. Delayed eruption of the maxillary lateral incisor was also evident. However, enlarged pulp chamber with open apices and gingival abscesses were not expressed in the present case.

The dental follicle calcification is affected in RO. The enamel prisms are generally hypoplastic or hypocalcified with irregular appearance of enamel prisms. [9],[10] The dentin is also poorly mineralized. Coronal part of the dentin is fibrous and contains clefts which could lead to communication between the oral cavity and pulp. [9],[10] Radicular dentin is relatively normal in structure and calcification.

The present case demonstrated no clear-cut radiographic demarcation between the hypomineralized enamel and dentin. The primary as well as permanent teeth, on the maxillary left quadrant are less radiopaque compared to the unaffected side.

Clinically as well as radiographically, the present case was differentiated from amelogenesis imperfecta, dentinogenesis imperfecta, dentin dysplasia Type I and Type II, and hypophosphatasia as these developmental anomalies involve entire dentition.

Treatment of RO is somewhat controversial although a prolonged multidisciplinary approach is often advocated. [5],[6],[7],[13],[14],[15] As hypomineralized teeth are prone to caries and fracture, long-term retention of these teeth is not always possible. Therefore, extraction of the involved teeth and replacement of missing teeth with denture are indicated. [6],[7],[13],[14] Conversely, some clinicians prefer to retain the teeth in growing children to stimulate natural growth of the jaw bones. [5],[6],[7],[14] Management of RO generally depends on the age of the patient, degree of malformation, type of dentition affected, number and position of teeth affected, relevant medical history, and parental attitude regarding dental treatment. [7],[13] In the present case, the affected maxillary left primary central incisor was retained as it was asymptomatic. Furthermore, psychological trauma associated with extraction in a young child is avoided.


  Conclusion Top


RO is a developmental dental anomaly with distinct clinical and radiological features. Early diagnosis and intervention are necessary for a favorable prognosis. Treatment of RO usually requires a multidisciplinary approach although several factors such as age of the patient, degree of malformation, type of dentition, parental attitude, and relevant medical history should be considered while formulating a treatment plan. When affected teeth were not associated with pain or gingival swelling, conservative treatment is suggested.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Hatchin AD. Unerupted deciduous teeth in a youth aged 15½. Br Dent J 1934;56:631-3.  Back to cited text no. 1
    
2.
McCall JO, Wald SS. In: Clinical Dental Roentgenography. Philadelphia: WB Saunders Co.; 1947. p. 169-70.  Back to cited text no. 2
    
3.
Rushton MA. Odontodysplasia: "Ghost teeth". Br Dent J 1965;119:109-13.  Back to cited text no. 3
[PUBMED]    
4.
Zegarelli EV, Kutscher AH, Applebaum E, Archard HO. Odontodysplasia. Oral Surg Oral Med Oral Pathol 1963;16:187-93.  Back to cited text no. 4
[PUBMED]    
5.
Rashidian A, Afsharian Zadeh M, Azarshab M, Zarrabian T. Regional Odontodysplasia: Report of a case. J Dent (Shiraz) 2013;14:197-200.  Back to cited text no. 5
    
6.
Cho SY. Conservative management of regional odontodysplasia: Case report. J Can Dent Assoc 2006;72:735-8.  Back to cited text no. 6
    
7.
Cahuana A, González Y, Palma C. Clinical management of regional odontodysplasia. Pediatr Dent 2005;27:34-9.  Back to cited text no. 7
    
8.
Crawford PJ, Aldred MJ. Regional odontodysplasia: A bibliography. J Oral Pathol Med 1989;18:251-63.  Back to cited text no. 8
    
9.
Kinirons MJ, O′Brien FV, Gregg TA. Regional odontodysplasia: An evaluation of three cases based on clinical, microradiographic and histopathological findings. Br Dent J 1988;165:136-9.  Back to cited text no. 9
[PUBMED]    
10.
Fearne J, Williams DM, Brook AH. Regional odontodysplasia: A clinical and histological evaluation. J Int Assoc Dent Child 1986;17:21-5.  Back to cited text no. 10
[PUBMED]    
11.
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, 155.  Back to cited text no. 11
    
12.
Walton JL, Witkop CJ Jr., Walker PO. Odontodysplasia. Report of three cases with vascular nevi overlying the adjacent skin of the face. Oral Surg Oral Med Oral Pathol 1978;46:676-84.  Back to cited text no. 12
    
13.
Magalhães AC, Pessan JP, Cunha RF, Delbem AC. Regional odontodysplasia: Case report. J Appl Oral Sci 2007;15:465-9.  Back to cited text no. 13
    
14.
Babu NS, Smriti RJ, Pratima DB. Regional odontodysplasia: Report of an unusual case involving mandibular arch. Contemp Clin Dent 2015;6:237-9.  Back to cited text no. 14
    
15.
Mathew A, Dauravu LM, Reddy SN, Kumar KR, Venkataramana V. Ghost teeth: Regional odontodysplasia of maxillary first molar associated with eruption disorders in a 10-year-old girl. J Pharm Bioallied Sci 2015;7 Suppl 2:S800-3.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2]


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