Single Tooth Odontodysplasia- Case Report With Review OfLiteratureAuthors:Kalra Manpreet 1 , Gurunathan. Deepa 2 , Radhakrishnan Raghu 3 , Dave Aparna 4 , Kanwardeep Singh Nanda 4 ,Madan Parul 41,4 Department of Oral and Maxillofacial Pathology, SGT <strong>Dental</strong> College Hospital and Research institute, Gurgaon,India2 Department of Pedodontic, Saveetha Institute of Medical and Technical Sciences, Chennai3 Department of Oral and Maxillofacial Pathology, Manipal college of <strong>Dental</strong> sciences Manipal, IndiaABSTRACTRegional odontodysplasia (RO) is a rare, non-hereditary anomaly of the dental hard tissues that results incharacteristic ghost like appearance of affected teeth. This paper reports a unique case in a 11yr year old boy with“single tooth” in maxillary arch affected instead of a segment or a quadrant/arch. Final diagnosis of RO was givenon the basis of typical radiological and histopathological investigations. Clinical management of cases RO has givenrise to controversy. The purpose of the article is to provide valuable information and review pathogenesis, specialclinical, radiographical, histological feature and various treatment alternatives for ROD.Key Words: Single tooth, odontodysplasia, ghost teeth, pathogenesis,treatment alternatives.INTRODUCTIONHitchin was first to recognize this conditionin 1934 1 , however it was McCall and Wald 2 whowere credited for publishing the first report ofodontodysplasia in 1947, in which they termed thecondition ‘arrested tooth development’. In 1954 theterm ‘shell teeth’ was introduced by Rushton 3 , whichthe author used to describe the radiographic findingsof the anomaly. In 1963, Zegarelli et al. 4 were thefirst to suggest the term ‘odontodysplasia’. The term‘regional’ was added because the condition affects agroup of several adjacent teeth in a particular segmentof the jaw.The etiology of RO is uncertain; numerousfactors have been suggested and considered as localtrauma, irradiation, hypophosphatasia, hypocalcemia,hyperpyrexia. Generally, the disturbance is localizedto one arch and the maxilla is involved twice as oftenas the mandible 5 . The left side of the maxilla is themost frequently affected site followed, in decreasingfrequency, by the maxillary right, mandibular rightand mandibular left regions 5 . The condition is usuallyunilateral, without tendency to cross the midline 6, 7 .However in some cases, RO has been seen to crossthe midline, particularly in lower jaw 6,8,9 . The numberof affected teeth is variable and the affected teethare usually in a continuous series. Sometimes thecondition has been seen to ‘skip’ a tooth or a group ofteeth 10 .45CASE REPORTAn eleven year old boy came to dentalpractitioner with the complaint of an uneruptedpermanent maxillary right central incisor. According tothe mother there was a history of trauma to the upperanterior region of the child when he was two year oldand the deciduous right maxillary central incisor hadturned mobile which was extracted as advised by thedentist. Predecessors were reported to be normal withno atypical appearance. No relevant medical historywas found and there were no similar cases in thefamily.On examination the patient had no facialasymmetry or other extra oral abnormalities. Onintraoral examination all teeth were present accordingto age and were normal with a good oral hygieneexcept for the missing upper right central incisor. Thegingival tissue covering the missing tooth was bluish incolour and softer than the adjacent area (Fig 1).Radiographic examination showed impactedpermanent right upper central incisor. The crown of theaffected tooth was surrounded by a large radiolucencysuggestive of an enlarged dental follicle. The teethhad thin radio-opaque contours with no distinctionbetween enamel and dentin, and wide pulp chambersgiving a ghost- like appearance. (Fig 2)On basis of clinical and the radiographic findings,a provisional diagnosis of RO was made.<strong>Malaysian</strong> <strong>Dental</strong> Journal Jan-Jun 2011 Vol 32 No 1© 2011 The <strong>Malaysian</strong> <strong>Dental</strong> <strong>Association</strong>
Single Tooth Odontodysplasia- Case Report With Review Of LiteratureUnder local anaesthesia and intravenous sedation,unerupted maxillary incisor was enucleated. Theenucleated tooth was of altered morphology, yellowishin colour with pitted enamel and short root that had adistal curvature in the apical third ( Fig 3).Following decalcification, the extracted teethwere prepared for microscopic examination in the usualmanner and stained with haematoxylin and eosin. Lightmicroscopic examination reveals poorly formed hardtissue. The enamel was irregularly mineralized andhypoplastic while dentin has large areas of interglobularsubstance. The dental follicle was hyperplastic andwas composed of dense fibrous connective tissuewith scattered collection of basophilic enamel likecalcification suggestive of enameloid conglomeratesand islands of odontogenic epithelium (Fig 4,5,6).Based on histologic features, the provisional diagnosisof RO was confirmed.Figure 3: Gross viewFigure 1: Frontal view of the patientFigure 4 :(4X magnification). Section shows poorlyformed hard tissue. The enamel is irregularlymineralized and hypoplastic while dentin has largeareas of interglobular substanceFigure 2: Occlusal view showing “ghost teeth”Figure 5: (40X magnification). Basophilic enamel-likecalcification called enamel conglomerates<strong>Malaysian</strong> <strong>Dental</strong> Journal Jan-Jun 2011 Vol 32 No 146