An Alternative Restorative Method for Regional Odontodysplasia: Case Report

An alternative restorative method for regional odontodysplasia: case report Shu-Hwa Yuan, DDS Perng-Ru Liu, DDS, MS Noel K. Childers, DDS, MS, PhD Abstract velopment, and reported only radiographic findings. The first comprehensive description of this condition A 5-year-old Caucasian male presented with early loss was by Hit~hin,~who published a description of of multiple deciduous teeth. All the characteristics were consistent with the diagnosis of regional odontodysplasia unerupted primary teeth. The term odontodysplasia (ROD).Significant initial findings included premature was introduced by Zegarelli, et aL4Numerous case re- loss of multiple primary mandibular teeth and some mal- ports and literature reviews have been published since formed permanent teeth. The affected teeth showed hy- then. An article by Crawford and Aldred provides a relatively complete bibliography of the condition.’ poplastic enamel and dentin, short roots, and wide pulp chambers, and were localized in the mandibular denti- Published cases suggest ROD occurs more fre- quently in females, with no racial predilection. The tion. Treatment objectives for this patient were to pro- condition can affect both primary and permanent den- vide improved esthetics, restored chewing function, and titions and can occur in the maxilla, the mandible, or space maintenance by the contruction of a temporary prosthetic restoration. However, with limited tooth sup- both. The maxilla is involved twice as often. Although the incisors and canines are most often involved, any port and an unusual occlusal pattern, it is difficult to obtain satisfactory retention and esthetics with tradi- tooth may be affected. When primary teeth are involved, the permanent successors are also usually af- tional prosthetic techniques. In this article we introduce an alternative method for fabricating a custom remov- fected.’ The condition tends to affect several adjacent teeth within a particular quadrant. Lustmann5 found able denture and discuss the prognosis of the malformed that affected teeth were located on both sides of the permanent dentition and further treatment plan. midline in only 11 of the 51 cases reported. ROD is usu- (Pediatr Dent 19:421-24,1997) ally seen in consecutive teeth, with no normal teeth interposed, although occasional skipped or minimally egional odontodysplasia (ROD) is a relatively affected teeth do occur in the permanent dentition. rare dental anomaly. The clinical manifestations Permanent first molars are affected only if the adjacent of odontodysplasia are:’ R primary molar exhibits ROD.6 Hypoplasia and hypocalcification of the enamel Multiple etiologies have been proposed, with defec- and dentin tive vascular function the most widely accepted. The Localized occurrence within a particular seg- association of vascular nevi in the facial skin overlying ment in either or both dentitions areas where ROD occurs suggests that a local vascular Discolored, small, and distorted affected teeth defect may be involved in the pathogenesis of ROD. with various surface markings (e.g., irregular These nevi are pale pink in color and disappear gradu- contour, shallow furrows, pitting, and grooves) ally with age.’ Delayed or failed eruption due to arrested root Other conditions which share some characteristics formation. with ROD include amelogenesis imperfecta, dentino- The radiographic features have consistently dem- genesis imperfecta, dentinal dysplasia types I and 11, onstrated thin and defective layers of enamel and den- shell teeth, rickets, and hypophosphatasia. However, tin, resulting in a faint, fuzzy outline, creating a ghost- it should be noted that, unlike ROD, all these conditions like appearance. The pulp chambers and canals are affect the entire dentition rather than a localized area. enlarged and the roots appear short and stubby with Turner’s teeth should be considered if only one or two open apices. True and false denticles may be seen in permanent teeth are affected.’ the pulp of affected teeth as well as adjacent, clinically Though many cases of ROD have been reported in normal teeth.’ the literature, few have addressed treatment of this The first report of this condition was published by condition. Usually, cases were treated by the extraction McCall and Wald2under the title of Arrested Tooth De- of the affected teeth and restoration with a temporary Pediatric Dentistry - 19:6,1997 American Academy of Pediatric Dentistry 421 partial denture. Prosthetic management of multiple cated ghost teeth for all permanent mandibular teeth missing primary teeth is difficult because of the dy- except the right first molar. These ghost teeth were namic condition of the primary dentition (e.g., growth, small, with thin layers of enamel and dentin, short exfoliation, and transition to permanent dentition). roots, and wide pulp chambers. The mandibular left Although dental implants are advocated for restoration second premolar appeared to be missing. of adult patients, they have not been proven suitable Treatment for growing children. Therefore, a removable denture is the best available treatment choice to establish chew- Treatment goals consisted of the establishment of ing function, normal vertical dimension, space preser- normal chewing function and improved esthetics and vation, and improved esthetics. This report introduces space management. The primary mandibular right sec- an alternative method of denture fabrication for pa- ond molar and left primary canine received pulpoto- tients with multiple missing primary teeth, abnormal mies and stainless-steel crowns to prevent pulp infec- tooth morphology, and unusual occlusal relationship. tion from excessive abrasion. Rather than extracting affected teeth, preventive root Alginate impressions were obtained for partial den- canal treatment and restoration with stainless-steel ture construction. The missing teeth were fabricated in crowns were done to preserve these teeth by protect- wax to establish optimal occlusion and esthetics on the ing them from infection. study cast. The wax-up model was duplicated and used to manufacture a template. The custom-made teeth Case report were fabricated by pouring tooth-colored acrylic resin History and chief complaint into the template (complete instructions for the fabrication of custom-made artificial teeth are available on A 5-year-old Caucasian male was referred from a request from the author). After the partial denture was private dentist to the pediatric dental clinic of the Uni- made, a layer of resin (approximately 0.5 mm) was re- versity of Alabama at Birmingham with complaints of moved on the tooth-bearing portion of the denture and premature loss of multiple primary teeth and malfor- relined with soft liner material. Because there were no mation of permanent teeth. The patient's private den- undercuts on the remaining primary mandibular teeth, tist reported that several hypoplastic primary teeth, wire clasp retainers were not practical. A thin layer of none of which had had significant caries, had been ex- interim resilient liner was added to surround the pri- tracted at multiple episodes of abscesses. At the time mary canine and the second molar for improved reten- of examination he was in good health with normal stat- tion. Additionally at delivery, a thin layer of soft liner ure and no physical abnormalities. He was the middle was added to the tissue side of the denture to further child with two dentally normal and healthy siblings. improve retention. No other member of his family had any identified The patient and parents were satisfied with the es- dental anomalies. thetics, function, and retention of the denture at deliv- Gestation had been normal with full-term delivery, ery and at each 3-month follow-up appointment (Figs however at birth a flat and reddish birthmark under 3a and 3b). At the 1-year follow-up, the mandibular the right side of the lower lip had been noted. The right first permanent medical history revealed no contributory serious ill- molar was found to nesses, other than antibiotic therapy for 2 weeks for have erupted with nor- an ear infection at 2 years of age. mal morphology. The Examination denture was modified Extraoral examination revealed a nonprominent with a reinforced wire, birthmark on the facial skin under the right lower lip. self-cured acrylic resin Intraoral clinical examination revealed a caries-free, to the right lingual pos- morphologically normal, full complement of primary Fig 1. Intraoral photograph of terior border of the maxillary teeth. All of the mandibular primary teeth the mandibular dentition. denture, and a ball were missing except the left canine and right second molar (Fig 1). The primary mandibular left canine had rough, hypoplastic yellow enamel and was moderately mobile. The primary mandibular right second molar had normal morphology with mild enamel hypoplasia. Full-mouth periapical radiographs and a panoramic radiograph (Fig 2) were obtained. Dental development from radiographs appeared age-appropriate and showed normal thickness of enamel and dentin in maxillary primary and permanent dentitions. However, excessive pulp horns and pulp stones were observed in Fig 2. Panoramic radiograph showing normal maxillary some primary maxillary teeth. The radiographs indi- dentition and affected mandibular teeth. 422 American Academy of Pediatric Dentistry Pediatric Dentistry -19:6,1997 clasp between the right second primary molar and the side suggesting a local vascular defect. This patient first permanent molar. The

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