Pre-eruptive coronal resorption and congenitally missing teeth in a patient with amelogenesis imperfecta: a case report.

Pre-Eruptive Coronal Resorption and Congenitally Missing Teeth in a Patient with Amelogenesis Imperfecta: A Case Report Ozkan Miloglua Osman Fatih Karaalioglub Fatma Caglayana Zeynep Duymus Yesilc Abstract This clinical report describes a male with autosomal recessive generalized hypoplastic amelogenesis imperfecta. This case is unusual in coronal resorptions prior to tooth eruption. This finding has been reported in some cases of autosomal recessive, autosomal dominant and X linked amelogenesis imperfecta (AI). In reported cases, the defects were usually small and occurred in a maximum of 2 teeth per person. In our case, pre-eruptive coronal resorptions affected three second molar teeth from both jaws. On the other hand; congenitally missing teeth and malocclusion were present in this case. Recall evaluations at 3 month intervals occurred for a period of 2 years and then prosthodontic management began. (Eur J Dent 2009;3:140-144) Key words: Amelogenesis imperfecta; Congenital missing teeth; Pre-eruptive coronal resorption; Malocclusion; Prosthetic restoration. Introduction Amelogenesis imperfecta (AI) is a developmental, often inherited disorder affecting dental enamel. It usually occurs in the absence of systemic features Research Assistant, Ataturk University, Faculty of Dentistry, Department of Oral Diagnosis and Radiology, Erzurum, Turkey. b Research Assistant, Ataturk University, Faculty of Dentistry, Department of Prosthodontics, Erzurum, Turkey. c Professor, Ataturk University, Faculty of Dentistry, Department of Prosthodontics, Erzurum, Turkey. a Corresponding author: Dt. Ozkan Miloglu Atatürk University, Faculty of Dentistry, Department of Oral Diagnosis and Oral Radiology, 25240, Erzurum, Turkey. Fax: +90 442 2360945 E-mail: European Journal of Dentistry 140 and comprises diverse phenotypic entities.1 AI has an estimated prevalence of approximately between 1:8000 and 1:700.2 As in hereditary disorder, clustering in certain geographic areas may occur, resulting in a wide range of reported prevalence. In general, both the deciduous and permanent dentitions are diffusely involved.3,4 Although AI is considered to primarily affect the enamel, further alterations could include unerupted teeth,1,4-8 congenitally missing teeth,4,8 taurodontism,1,4,6,7,9,10 pulpal calcifications,1,5,6,11 crown and root resorption,1,4-6,8 cementum deposition,5,6 truncated roots,6 dental and skeletal open bite,6,12 interradicular dentinal dysplasia,6,7 gingival hyperplasia5,8 and follicular hyperplasia.6 Miloglu, Karaalioglu, Caglayan, Yesil As mentioned above, additional dental pathologies such as eruption failure accompanying amelogenesis imperfecta and crown resorptions, may be in question. In literature reports, crown resorption in pre-eruptive teeth has been demonstrated in one or a few teeth at maximum. This article presents a male with generalized hypoplastic amelogenesis imperfecta, who has crown resorptions in multiple pre-eruptive teeth accompanying congenital tooth loss. CASE REPORT 20 years old male patient referred to the Department of Prosthodontic Dentistry in Ataturk University for aesthetic and tooth sensitivity complaints. His medical history and general physical condition were unremarkable. His hair, skin, and nails appeared normal. The pregnancy and the post-natal period had been uneventful. Patient’s parents were examined and showed unaffected permanent dentitions. No evidence of a similar condition could be elicited in the family Figure 1. Pretreatment view of teeth in occlusion. history. The patient lived in a non-fluoridated area and had never taken fluoride supplements. Clinically, the permanent teeth were yellowish in color with a rough enamel surface as a result of mild hypoplasia. The incisal edges were thin and the teeth were widely spaced (Figure 1). Both upper second molars and lower first and second molars were clinically not visible. A general enlargement of the gingival tissues was not diagnosed but periodontal pockets were detected on the adjacent fully erupted teeth. Because of poor oral hygiene, presence of plaque accumulation and related chronic marginal gingivitis was in question. The panoramic radiograph obtained at the referral showed permanent dentition which was affected by multiple intracoronal radiolucencies in both upper second molars and lower right second molar. Whereas, the defects of the both upper second molars were limited to enamel and dentin; in the lower right second molar the lesion involved the pulp chamber (Figure 2). The clinical examination of the patient revealed that the soft tissues overlying the unerupted affected teeth were intact. Lower left first and second molars were congenitally missing. Lower right first molar tooth was surgically removed and a residual mesial root was visible in radiography. Upper left third molar was congenitally missing, upper and lower third molars were present but unerupted. The contrast between enamel and dentin was normal and all teeth were affected from some degree of taurodontism but pulp stones were not visible in radiography. In recall evaluation after 1 year, no changes were determined in preeruptive resorption of second molars and in eruption of Figure 2. Panoramic radiograph of the patient at the first referral. April 2009 - Vol.3 European Journal of Dentistry 141 Pre-eruptive coronal resorption and congenitally missing teeth third molars (Figure 3). Upper right first molar and upper left canine were restored due to caries lesion and lower right second molar was extracted. All teeth had small clinical crowns and axial angle of the teeth were not prepared ideally of 3-6° angle because of the teeth morphology; fullarch fixed denture were planned considering the retention of the denture after cementation. Maxillary and mandibular anterior and posterior teeth were prepared for metal-ceramic restorations with narrow champher finish lines. Laboratory-processed provisional restorations were fabricated at an increased occlusal vertical dimension (3.0 mm), lined with methyl methacrylate acrylic resin (Major C&B-V Dentine, Major, Moncalieri, Italy) and cemented with zincoxide eugenol (Temp-Bond; Kerr Corp). The patient used the provisional restorations at the newly established occlusal vertical dimension for 6 Figure 3. Panoramic radiograph of patient after 1 year. Figure 4. Intraoral and extraoral views of the prostheses. European Journal of Dentistry 142 months without complications. Final impressions of the prepared maxillary and mandibular anterior teeth were obtained using vinyl polysiloxane impression material (Elite H-D; Zhermack). Working casts were generated from Type IV die stone (Bego Bremer Goldschlagerei Herbst GMBH Germany, 6124166) and mounted onto the articulator (Hager & Werken, Duisburg, Germany) using interocclusal records. Full arch metalceramic fixed denture (Ivoclar Vivadent) replacing teeth were fabricated, evaluated intraorally, adjusted to the (...truncated)