Regional odontodysplasia with supernumerary teeth ...

[Downloaded free from http://www.cjhr.org on Wednesday, September 28, 2016, IP: 81.83.62.154]

Case Report

Regional odontodysplasia with supernumerary teeth in pediatric patients: Coincident/new finding? Vela D. Desai, Rajeev Sharma, Swati Phore, Sudakshina Das Department of Oral Medicine and Radiology, Jaipur Dental College, Jaipur, Rajasthan, India

A B S T R A C T Regional odontodysplasia is a rare developmental dental anomaly affecting both primary and permanent dentition in any of the jaws with uncertain etiology. Clinically, affected tooth are impacted and if erupted are hypoplastic or hypocalcified. Radiographically, the affected teeth show a typical “ghost‑like” appearance. In this article, two such case reports are presented by the authors associated with supernumerary teeth in the same region. Keywords: Ghost‑like teeth, hypoplasia, odontodysplasia, regional

INTRODUCTION Regional odontodysplasia (RO) is an uncommon, nonhereditary developmental anomaly affecting dental tissues, derived from both the mesoderm and ectoderm.[1] The first report of this condition was published by McCall et al., in 1947, but the term “odontodysplasia” was introduced by Zegarelli et al., in 1963. Since then, various other terms have been suggested, such as RO, ghost teeth, odontogenesis imperfecta, localized arrested tooth development, unilateral dental malformation, amelogenesis imperfect, nonhereditaria segmentalis and familial amelodentinal dysplasia.[2] Females are slightly more affected (1.4:1) and have no ethnic predilection.[3] The anomaly is usually localized in one arch, with incidence higher in the maxilla. Rarely, all teeth of the same arch are affected. In cases, where both arches are involved, the presentation is usually unilateral. The affected teeth most often occur as a continuous series, Access this article online Quick Response Code: Website: www.cjhr.org DOI: 10.4103/2348-3334.158714

although occasionally the anomaly will “skip” a tooth or group of teeth. Eruption of the affected teeth is often delayed or failed.[4] Clinically, affected teeth have an abnormal morphology and a rough surface with defective mineralization.[5,6] The management of RO is somewhat controversial and revolves around the question of whether or not to remove the affected teeth. Although many clinicians prefer to extract the anomalous teeth as soon as the diagnosis is made, some would prefer to retain them as long as they are free of infection until the skeletal growth is complete.[3,4] These cases are interesting and rare. We present case reports of this uncommon anomaly with unique finding of additional supernumerary teeth (ST).

CASE REPORTS Case 1 A 12‑year‑old boy reported to the Oral Medicine and Radiology Department of Jaipur Dental College with the chief complaint of missing upper front tooth [Figure 1]. Patient did not give any history of previous trauma, and medical history was noncontributory. General physical examination was normal. There was no history of similar anomaly reported in either side of his family. Patient visited earlier with the same complaint of

Corresponding Author: Dr. Vela D. Desai, B‑406, Trimurty Apartments, Opposite BSNL Telecom Colony, Model Town, Malviya Nagar, Jaipur ‑ 302 017, Rajasthan, India. E‑mail: [email protected] 272

CHRISMED Journal of Health and Research /Vol 2/Issue 3/Jul-Sep 2015


Desai, et al.: Regional odontodysplasia with supernumerary teeth

missing teeth, and the panoramic radiograph [Figure 2] revealed impacted 11, 12, 13, 15, 16 and 17 in the upper right quadrant. But nothing much was done at that time as the patient returned only after 2 years. On intraoral examination, mixed dentition was present with 11,12,13,15 and 16 missing [Figure 3]. Associated alveolar mucosa was hyperplastic and covered by fibrous tissue. First premolar was rotated, and mild enamel hypoplasia was evident. Rest of the dentition was normal with relatively good oral hygiene. After informed consent, radiographic investigations were advised. Panoramic radiograph [Figure 4] showed impacted 11, 12, 13, 16 and an extra tooth‑like structure above the apices of 15 having similar radiodensity with that of adjacent teeth suggestive of ST (paramolar). It was better visualized in the intra oral periapical radiograph [Figure 5]. First premolar was horizontally displaced with incomplete root formation. There was malformed and retarded development of the teeth on the

right side of the maxilla, related to the age of the child with reduction in radiodensity of teeth as compared to the contralateral side. Malformed teeth with thin contours were appreciated in maxillary occlussal radiograph [Figure 6], with no distinction between enamel and dentin and had wide pulp chambers, giving “ghost‑like appearance”. Crowns of affected unerupted teeth were surrounded by radiolucent areas, probably representing enlarged dental follicles. Only an insignificant amount of root formation with open apices was visible radiographically with respect to 12, 14, 15, and 16 and with respect to 11, 13 and ST (paramolar) only crown formation was evident. With clinical and radiographical evaluation, a diagnosis of RO and ST (paramolar) in the same quadrant was made. Patient was explained about the treatment plan of extraction of the hypoplastic teeth followed by removable partial dentures that can be later replaced by implants after patient achieves complete skeletal growth. Oral prophylaxis and oral hygiene instructions were advised and followed up.

Figure 1: Case I - extraoral profile

Figure 2: Previous radiograph- impacted 11, 12, 13, 15, 16 and 17

Figure 3: Intraoral picture - missing 11, 12, 13, 15 and 16

Figure 4: Panoramic radiograph showing impacted 11, 12, 13, 16 and an extra tooth-like structure above the apices of 15


273



Case 2 A 9‑year‑old patient visited Dental Outpatient Department in Jaipur Dental College with the chief complaint of extra tooth in upper front region. Past dental, medical and family history was noncontributory. There was no history of trauma in the childhood.

thin radiopaque contours and no distinction between enamel and dentin. There were wide pulp chamber and incomplete root formation with respect to 21. Irregular root apex with respect to mesiodens was appreciated with periapical radiolucency suggestive of chronic periapical abscess.

On clinical examination, patient had mixed dentition with conical shaped tooth‑like structure in the midline suggestive of mesiodens present in the midline between 11 and 21 [Figure 7]. Left central incisor was rotated and showed mild yellowish tint as compared to adjacent teeth in the dental arch. There was no evidence of caries, deep fissures, pits or pockets notable. Rest of the dentition was normal with good oral hygiene.

On the basis of clinical and radiographical findings, provisional diagnosis of RO and conical mesiodens was made. Patient’s parents were informed about the anomaly and advised for extraction of mesiodens, orthodontic correction of rotated central incisor and were regularly followed up.

An intra‑oral periapical radiograph was taken for the same region after parent’s informed consent [Figure 8]. Radiographically, left central incisor was malformed with

Regional odontodysplasia is a relatively rare, nonhereditary, localized developmental anomaly of

Figure 5: Intraoral periapical radiograph showing extra tooth-like structure above the apices of 15

Figure 7: Case II - Rotated left central incisor with mild yellowish tint and mesiodens present in the midline between 11 and 21

274

DISCUSSION

Figure 6: Occlussal radiograph showing malformed teeth with thin contours, with no distinction between enamel and dentin, wide pulp chambers and giving “ghost-like” appearance

Figure 8: Malformed left central incisor with thin radioopaque contours and no distinction between enamel and dentin with wide pulp chamber and incomplete root formation, giving ghost-like appearance of the tooth




the dental hard tissues of a group of contiguous teeth. Many cases are probably misdiagnosed as malformed teeth or odontomas. It occurs in both deciduous and permanent dentitions, and it has a marked preference for the maxilla.[7] The etiology of RO is still unknown, and such conditions as viral infections, local trauma, vascular defects, irradiation, metabolic disturbance, rhesus incompatibility and medications during pregnancy have been suggested as possible causes. In the present cases also, etiologic factors could not be identified, and no systemic involvement was evident. 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.[8] A ST is one that is additional to the normal series and can be found in almost any region of the dental arch.[9] ST can be classified according to their location in the dental arch: Mesiodens, paramolar and distomolar.[10] The term mesiodens denotes a ST located between the maxillary central incisors and is the commonest form. A paramolar most commonly occurs in the interproximal space buccal to the upper second and third molars.[11] However, uncommon association with ST was observed in both the cases, which is a very rare finding and not reported in the literature till date. In second case, the involved tooth had incomplete root formation or the periapical radiolucency was leading to root resorption still remains dilemma. As there is no general agreement on the best treatment for these patients, dentists should consider such factors as the patient’s age, medical history, degree of involvement, the presence or absence of pathosis and the attitude and expectations of the child and parents. The treatment is aimed at improving function and esthetics, reducing the psychological impact of early tooth loss and facilitating normal jaw growth. If a

decision is made to retain the anomalous teeth, regular review is mandatory.[4]

CONCLUSION Unique clinical and radiographic features of RO are presented here, and the association of RO and ST in the same quadrant is very rare finding that is seen in both the cases. This could give a clue to the dentist at an early age. Also the association of ST in the same quadrant, which is also affected, is coincidental or associated finding needs to be further questioned and add to the present English literature.

REFERENCES 1.

Upadhyay V, Chaturvedi TP. Regional odontodysplasia crossing midline: A rare case report. Int J Clin Pediatr Dent 2011;4:159‑61. 2. Zegarelli EV, Kutscher AH, Applebaum E, Archard HO. Odontodysplasia. Oral Surg Oral Med Oral Patho 1963;16:187–93. 3. Kappadi D, Ramasetty PA, Rai KK, Rahim AM. Regional odontodysplasia: An unusual case report. J Oral Maxillofac Pathol 2009;13:62‑6. 4. Cho SY. Conservative management of regional odontodysplasia: Case report. J Can Dent Assoc 2006;72:735‑8. 5. Dagistan S, Miloglu O Goregen M, Harorli A. Regional odontodysplasia: A case report. J Dent Res Dent Clin Dent Prospects 2009;3:141-44. 6. Cahuana A, González Y, Palma C. Clinical management of regional odontodysplasia. Pediatr Dent 2005;27:34‑9. 7. Mehta DN, Bailoor D, Patel B. Regional odontodysplasia. J Indian Soc Pedod Prev Dent 2011;29:323‑6. 8. Magalhães AC, Pessan JP, Cunha RF, Delbem AC. Regional odontodysplasia: Case report. J Appl Oral Sci 2007;15:465‑9. 9. Fazliah SN. Supernumery tooth: Report of a case. Arch Orofac Sci 2007;2:54‑8. 10. Garvey MT, Barry HJ, Blake M. Supernumerary teeth – an overview of classification, diagnosis and management. J Can Dent Assoc 1999;65:612‑6. 11. David DS, Akhila R. Management of multiple impacted supernumery teeth in a non‑syndromic patient using CBCT. Dent Open Access J 2014;4:1‑2. Cite this article as: Desai VD, Sharma R, Phore S, Das S. Regional odontodysplasia with supernumerary teeth in pediatric patients: Coincident/ new finding?. CHRISMED J Health Res 2015;2:272-5. Source of Support: Nil. Conflict of Interest: No.


275