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 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 4  |  Issue : 2  |  Page : 86-89

Regional odontodysplasia: A classical case report


1 Department of Pedodontics and Preventive Dentistry, People's College Of Dental Sciences and Research Centre, Bhopal, Madhya Pradesh, India
2 Private Practitioner, Bhopal, Madhya Pradesh, India

Date of Web Publication22-Oct-2013

Correspondence Address:
Ritika Malhotra
Department of Pedodontics and Preventive Dentistry, People's College of Dental Sciences and Research Centre, Bhopal - 462 037, Madhya Pradesh
India
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DOI: 10.4103/0976-433X.120186

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  Abstract 

Regional odontodysplasia (RO) is an uncommon developmental anomaly affecting a localized area of the dentition; which presents with distinctive clinical and radiographical findings. Clinically, the affected teeth are often grossly hypoplastic with abnormal morphology. They often tend to develop abscess soon after eruption. Radiographically, they have a typical "ghost-like" appearance. Although extractions are often required, in some milder cases the teeth may be retained for a long period. The treatment plan should be based on the degree of involvement as well as functional and esthetic needs in each case. This article describes a classical report of a 3.5-year-old female with RO.

Keywords: Ghost teeth, localized arrested tooth development, regional odontodysplasia


How to cite this article:
Malhotra R, Singla S, Shashikiran N D, Verma A. Regional odontodysplasia: A classical case report. SRM J Res Dent Sci 2013;4:86-9

How to cite this URL:
Malhotra R, Singla S, Shashikiran N D, Verma A. Regional odontodysplasia: A classical case report. SRM J Res Dent Sci [serial online] 2013 [cited 2019 Nov 13];4:86-9. Available from: http://www.srmjrds.in/text.asp?2013/4/2/86/120186


  Introduction Top


Regional odontodysplasia (RO) is an unusual, localized, nonhereditary developmental anomaly affecting the ectodermal and mesodermal dental components. The condition was first published by McCall et al., in 1947 [1] under the name of "arrested tooth development"; and the term "odontodysplasia" was coined by Zegarelli et al., in 1963. [2] Since it is commonly found to be affecting only a segment of the jaw, it was prefixed with the word "regional" by Crawford [3] ; and thus, RO became an accepted terminology. The other terms which can interchangeably be used with this condition are "ghost teeth", [4] "odontogenesis imperfecta", [5] "localized arrested tooth development", [3] "unilateral dental malformation", "amelogenesis imperfecta", [5] "non-herediteria segmentalis", and "familial amelodentinal dysplasia". [6]

Although genetic predisposition has been reported, [7] but the precise etiology of the condition has not been explained. Suggested etiological factors include somatic mutations affecting the dental lamina, localized trauma during pregnancy, forceps delivery, vascular disorders, pharmacological treatments during pregnancy, local or systemic viral infection, local traumas, nutritional or metabolic deficits, radiation, hyperpyrexia, local ischemia, and Rh incompatibility. [2],[3],[4],[7],[8],[9]

Though both the jaws may be affected; there is a 2:1 predilection of maxilla over mandible. [3] It is usually unilateral, with common involvement of anterior region. The anomaly is seen to affect both primary and permanent dentitions, which is usually apparent in one of the jaw and mostly in one quadrant.

The condition presents with a classical clinical, radiographic, and histological findings. Clinically, the abnormality involves the dental hard tissues derived from both the epithelial (enamel) and mesenchymal (dentine and cementum) components of tooth forming apparatus. Owing to which, the tooth is hypoplastic and hypocalcified, giving a typical yellow or yellowish-brown appearance to the enamel, and an abnormal morphology. [10] A frequent finding of RO is the presence of gingival swelling, periapical infections, and abscesses even in the absence of caries. [11],[12] Radiographically, a marked reduction in the radiodensity of the enamel and dentine can be seen, which leads to a poor contrast between them. Classically, a "ghost-like appearance" [13] (thus the name ghost teeth) of the teeth is observed. These teeth present with a shorter appearance, wide open apices, and a large pulp chamber. Histologically, the dental hard tissues are characterized as hypocalcified. Enamel prisms are irregularly distributed, with aprismatic and degenerative hypocalcified areas. [9] There is a marked reduction in the dentinal tubules, and areas of clefts can be seen within them. The pulp and dental follicle may contain calcifications of varying degrees. [14]

This paper reports a case of RO with its classical appearance clinically and radiographically.


  Case Report Top


A 3.5-year-old female patient reported in the Department of Pedodontics and Preventive Dentistry, People's College of Dental Science, Bhopal with the chief complaint of pain and swelling in the left upper back tooth region since 10 days. History revealed that the patient was delivered preterm by a forceps delivery with a breech presentation. The patient had an uneventful medical history.

Clinically, the child presented with a complete set of deciduous dentition [Figure 1], with the affected left maxillary primary first molar, associated with a gingival swelling. The deciduous molars were fractured, because of its hypoplastic and hypocalcified nature [Figure 2]. The deciduous left lateral incisor and canine were partially erupted, while the left deciduous central incisor appeared hypocalcified [Figure 1] and [Figure 2]. The teeth in the adjacent arches showed normal morphological features.
Figure 1: Clinical aspect of regional odontodysplasia case showing hypomineralized left deciduous central incisor, and partially erupted lateral incisor and canine

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Figure 2: Occlusal view, showing fractured left deciduous molars

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Panoramic [Figure 3] and periapical radiographs [Figure 4] and [Figure 5] showed longitudinal shortened teeth with lack of contrast between enamel and dentine. The affected teeth had wide pulp chambers with open apices and were smaller than normal in overall size. The main radiographic feature was a 'ghost' appearance. The succedaneous tooth germs appeared as initial hypocalcified clefts; suggesting the permanent tooth involvement. Other than the left maxillary hemiarch, other radiographic features showed normal appearance.
Figure 3: Panoramic view, showing initial hypocalcified crypts of the succedaneous permanent tooth germs

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Figure 4: Intraoral periapical radiograph of posterior region showing typical ghost appearance

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Figure 5: Intraoral periapical radiograph of anterior region showing typical ghost appearance

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Proper parent counseling was done, and only symptomatic treatment was given to the patient. After administration of systemic antibiotics, the primary first molar was extracted under local anesthesia.

Based upon the clinical and radiographical findings, the diagnosis of RO was made. Because of uncertain prognosis, the patient is kept on long-term follow-ups, to maintain the teeth in the arch, until full growth of jaw.


  Discussion Top


RO is a relatively uncommon, localized, nonhereditary developmental anomaly affecting the tooth morphogenesis by targeting the ectodermal and mesodermal components of the dental hard tissues. [3] It is often confused with other developmental anomalies affecting the tooth development like dentinal dysplasia, shell teeth, hypophosphotasia, dentinogenesis imperfecta, or amelogenesis imperfecta; but these disorders tend to affect the entire dentition, which is contradictory to RO.

The condition can involve both sets of the dentition. If the primary dentition is affected, most likely the permanent set of dentition is also affected. [15] The patient in the present report has an affected deciduous and permanent dentition which is visible radiographically. Since, there was no family history; the genetic predisposition in the present case can be ruled. Thus, the possible etiology could be local trauma due to forceps delivery. The present case can be described is a classic case of RO.

The literature reveals a female predilection (1.4:1) [3],[13],[14] and a left maxillary arch involvement over mandibular arch (2:1), which does not crosses the midline. [3],[9],[14] This is consistent with the findings of the present case; however, bilateral involvement [16] can also be seen. Although more commonly anterior region is affected, in the present case, all the teeth of the affected arch were equally affected. The affected teeth most often occur as a continuous series, although occasionally the anomaly will "skip" a tooth or group of teeth. [17]

The classical clinical presentation of partially erupted teeth with yellowish brown hypoplastic pitted appearance [3],[9],[10],[11] supports the previous reports. The tooth is thin, friable, and susceptible to trauma; which is attributable to its hypomineralization. Typically, gingival swelling is seen even in the absence of caries [11],[12] which is consistent with the present report.

Radiographically, the affected teeth showed a classical "ghost-like" appearance, with hypomineralized enamel and dentin, and the absence of clear demarcation between them. [3],[13] The teeth presented with a shorter appearance and a wide open apices with a large pulp chamber. This is in accordance with the previous published reports of the condition. [12],[13],[15] The succedaneous dental germs were also seen affected, suggesting the involvement of the permanent dentition.

The management of affected tooth in RO still remains controversial. [9] These cases require a continuous and a multidisciplinary approach. Some suggest the removal of the tooth with periapical pathologies followed by suitable prosthesis to restore function and esthetics, while other professionals suggest a restorative treatment wherever possible. Cahuna et al., [18] proposed autotransplantation as a treatment option in RO patients; however it is limited because of availability of an appropriate donor. Bulut et al., [19] suggested the use of endosseous implant for oral rehabilitation in patients with odontodysplasia, however, this treatment can be done at a later stage of life. Since there is no specific treatment projected for this condition, the treatment should be considered based upon the patient's age, compliance, attitude towards the dental treatment, and the degree of odontodysplasia.

In the present report, the conservative approach was not possible because of loss of the tooth structure and an open wide chamber, so we opted for removal of the involved tooth following the systemic administration of the antibiotics. The aim was to retain the anomalous teeth until skeletal growth was complete; so that the patient could be reassessed for dental implants and other rehabilitation methods.


  Conclusions Top


A case of classical presentation of RO is presented, with typical features and a possible etiology of local trauma caused by forceps delivery. As seen in this case, affected teeth showed different degrees of malformation. Although the tooth with periapical infection was extracted, others that were free from infection can be retained. The treatment plan should be based on degree of involvement as well as functional and esthetic needs in individual cases.

 
  References Top

1.McCall JO, Wall SS. Clinical dental roentgenology. 3 rd ed. Philadelphia: W B Saunders; 1952.  Back to cited text no. 1
    
2.Zegarelli EV, Kutscher AH, Applebaum E, Archard HO. Odontodysplasia. Oral Surg Oral Med Oral Pathol 1963;16:187-93.  Back to cited text no. 2
    
3.Crawford PJ, Aldred MJ. Regional odontodysplasia: A bibliography. J Oral Pathol Med 1989;18:251-63.  Back to cited text no. 3
    
4.Rushton MA. Odontodysplasia: "Ghost teeth". Br Dent J 1965;119:109-13.  Back to cited text no. 4
    
5.Claudhry AP, Wittich HC, Stickel FR, Holland MR. Odontogenesis imperfecta. Report of a case. Oral Surg Oral Med Oral Pathol 1961;14:1099-103.  Back to cited text no. 5
    
6.Mock D, Aidelbaum MR, Chapnick P. Familial amelodentinal dysplasia. Oral Surg Oral Med Oral Pathol 1986;61:485-91.  Back to cited text no. 6
    
7.Sadeghi EM, Ashrafi MH. Regional odontodysplasia: Clinical, pathologic, and therapeutic considerations. J Am Dent Assoc 1981;102:336-9.  Back to cited text no. 7
    
8.Hanks PA, Williams B. Odontodysplasia: Report of two cases. Pediatric Dent 1998;20:199-203.  Back to cited text no. 8
    
9.van der Wal JE, Rittersma J, Baart JA, van der Waal I. Regional odontodysplasia: Report of 3 cases. Int J Oral Maxillofac Surg 1993;22:356-8.  Back to cited text no. 9
    
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
    
11.Gomes MP, Modesto A, Cardoso AS, Hespanphool W. Regional odontodysplasia: Report of a case involving two separate affected areas. ASDC J Dent Child 1999;66:203-7.  Back to cited text no. 11
    
12.Kiniros MJ, O`Brien FV, Gregg TA. Regional odontodysplasia: An evaluation of three cases based on clinical, microradiographic and histopathologic findings. Br Dent J 1998;165:136-9.  Back to cited text no. 12
    
13.Melamed Y, Harnik J, Becker A, Shapira J. Conservative multidisciplinary treatment approach in an unusual odontodysplasia. ASDC J Dent Child 1994;61:119-24.  Back to cited text no. 13
    
14.Fanibunda KB, Soames JV. Odontodysplasia, gingival manifestations, and accompaying abnormalities. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1996;81:84-8.  Back to cited text no. 14
    
15.O'Neil DW, Koch MG, Lowe JW. Regional odontodisplasia: Report of case. ASDC J Dent Child 1990;57:459-61.  Back to cited text no. 15
    
16.Steiman HR, Cullen CL, Geist JR. Bilateral mandibular regional odontodysplasia with vascular nevus. Pediatr Dent 1991;13:303-6.  Back to cited text no. 16
    
17.Cho SY. Conservative management of regional odontodysplasia: Case report. J Can Dent Assoc 2006;72:735-8.  Back to cited text no. 17
    
18.Cahuna A, Gonzalez Y, Palma C. Clinical management of regional odontodysplasia. Pediatr Dent 2005;27:34-9.  Back to cited text no. 18
    
19.Bulut E, Guler AU, Sen Tunc E, Telcioglu NT. Oral rehabilitation with endosseous implants in a child with ectodermal dysplasia: A case report. Eur J Paediatr Dent 2010;11:149-52.  Back to cited text no. 19
    


    Figures

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


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