Case report: Idiopathic pre-eruptive coronal resorption of a maxillary permanent canine

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1 Case report: Idiopathic pre-eruptive coronal resorption of a maxillary permanent canine N.M. Manan*, S.K. Mallineni**, N.M. King*** *Paediatric Dentistry and Orthodontics Dept., Faculty of Dentistry, University of Malaya, Kuala Lumpur, Malaysia; **Paediatric Dentistry, Faculty of Dentistry, University of Hong Kong, Hong Kong SAR, China; ***School of Dentistry, University of Western Australia, Australia. Key words: Pre-eruptive coronal resorption, canine, internal resorption, external resorption Postal address: Prof. N.M. King, Paediatric Dentistry, School of Dentistry, Oral Health Centre of Western Australia, University of Western Australia, 17 Monash Avenue Nedlands, Perth, Western Australia, WA profnigelking@mac.com Abstract Background: Coronal resorption is a coronal degeneration of enamel and dentine in which ultimately the crown is replaced by vascular connective tissue through a defect in the enamel organ of an unerupted tooth. This is also known as pre-eruptive coronal resorption. However, the aetiology of resorption remains unclear. Case report: A 13 years 7 months old Caucasian boy who attended an orthodontic consultation for anterior crossbite correction presented with idiopathic pre-eruptive coronal resorption of an erupted right maxillary permanent canine (FDI 13). Clinically, the enamel remaining on the crown was extremely thin and had a shelllike appearance. There was erythematous soft tissue within the parameters of the crown that resembled pulp tissue. From the radiographs, the irregular radiolucency area within the crown portion extended widely into the enamel and dentine. Treatment: Following excisional biopsy, it was decided to retain the right maxillary canine and monitor its progress. FOLLOW-UP: He has been reviewed at frequent appointments over 18-months since the time there was radiographic evidence of resorption. CONCLUSION: It is prudent to make an early diagnosis of this condition and to formulate short and long-term treatment plans, which may involve keeping the affected tooth to retain the alveolar bone height and width to allow for the option of planning for an implant. Introduction Idiopathic coronal resorption of a tooth prior to eruption into the oral cavity resembles a carious lesion in appearance. Therefore, it is easily misdiagnosed. Pre-eruptive resorptive lesions often remain unobserved because they cannot be detected until the tooth has erupted [Brooks, 1988]. Hence, clinical identification of idiopathic coronal resorption has been difficult. Thus, radiographic records are useful to provide an accurate diagnosis of pre-eruptive lesions during a routine recall visit. However, preeruptive intra-coronal radiolucencies are characteristically noted as accidental findings on radiographs of unerupted teeth, where they frequently appear as distinct radiolucencies within the coronal dentine, immediately adjacent to the amelodentinal junction. Therefore, the radiological appearance of external resorption of a crown has to be distinguished from pre-eruptive radiolucent lesions such as caries, internal resorption and hypoplasia [Wood and Crozier, 1985; Rankow et al., 1986; Brooks, 1988]. The reported prevalence of affected subjects is between 1.56% and 27.3% and tooth prevalence is 0.5% to 2.1%. [Seow et al., 1999a and b; Nik and Rahman, 2003; Őzden and Acikgoz, 2009]. Usually, a single tooth is affected in an individual but two or more lesions have also been reported [Seow, 1998]. Most of these lesions have been found in the permanent dentition; however, the occurrences of these lesions in the primary dentition [Seow and Hackley, 1996] and an association with supernumerary teeth [Őzden and Acikgoz, 2009] have also been reported in the literature. In the permanent dentition the most commonly involved teeth have been the mandibular first permanent molars, followed by the maxillary first permanent molars, the mandibular second premolars, and then the mandibular second permanent molars. In one study it has been reported that 106 (46.9%) of 226 unerupted and impacted teeth with coronal resorption were maxillary permanent canines [Stafne and Slocumb, 1944]. These results were based on subjects of various ages. Coronal resorption is manifested histologically by degeneration of the enamel and dentine of the coronal portion of the tooth, which is ultimately replaced by vascular connective tissue it occurs because of a defect in the enamel organ of an unerupted tooth [Stafne and Austin, 1945; Blackwood, 1958; Owens et al., 1988]. The irregular resorption begins in localised areas of the enamel and subsequently involves the dentine of the unerupted tooth. These features were considered to describe an external resorptive process [Blackwood, 1958]. Intra-coronal or internal resorptive lesions appear radiographically as a well-circumscribed radiolucency in dentine [Luten, 1958; Seow, 1998; Nik and Rahman, 2003] and from histological findings they resemble globular fragments of dystrophic calcifications, remnants of necrotic dentine and aggregates of pre-collagenous fibres [Walton, 1980]. Seow and her co-workers [1999 a and b] speculated that resorptive cells from the surrounding bone get into the dentine through a breach in the external covering of the developing tooth. The majority of previous studies [Stafne and Austin, 1945; Browne, 1954; Blackwood, 1958; Luten, 1958; Grundy et al., 1984; Brooks, 1988; Owens et al., 1988] have supported the hypothesis that pre-eruptive lesions are resorptive in nature. However, the aetiology of this condition remains unclear; although several hypotheses [Skillen, 1941; Blackwood, 1958; Browne, 1954; Kronfeld, 1955; Muhler, 1957; Luten, 1958; Wooden and Kuftinee, 1974; Walton, 1980; Giunta and Kaplan, 1981; Grundy et al., 1984; Brooks, 1988; Ignelzi et al., 1990; Holan et al., 1994; Seddon et al., 1996; Seow et al., 1999 a and b] have been proposed, see Table 2. The purpose of this report is to describe an erupted right maxillary permanent canine with pre-eruptive coronal resorption with an emphasis on the initial clinical management. 98 European Archives of Paediatric Dentistry 13 (Issue 2). 2012

Pre-eruptive coronal resorption of maxillary canine Figure 1.

A follow-up periapical radiograph of the right maxillary permanent canine shows an apical radiolucent area and the dentine immediately adjacent to the pulp canal remains as a dentine lined tube.

A periapical radiograph of the right maxillary permanent canine shows an irregular radiolucent area, a thin layer of dentine protecting the pulp while most of the coronal dentine appears to have been

A review of the patient s previous medical history indicated that he was healthy except that he suffered from hay fever and asthma. Figure 3.

2 Pre-eruptive coronal resorption of maxillary canine Figure 1. Intra-oral photograph showing the right permanent maxillary canine with shell-like enamel fragments attached to erythematous soft tissue which resembles a pulp polyp. Figure 4. A follow-up periapical radiograph of the right maxillary permanent canine shows an apical radiolucent area and the dentine immediately adjacent to the pulp canal remains as a dentine lined tube. Figure 2. A periapical radiograph of the right maxillary permanent canine shows an irregular radiolucent area, a thin layer of dentine protecting the pulp while most of the coronal dentine appears to have been resorbed. Case report An orthodontic consultant referred a 13 years 7 months old Caucasian male because of gingival hyperplasia associated with his maxillary right canine tooth. A review of the patient s previous medical history indicated that he was healthy except that he suffered from hay fever and asthma. Figure 3. A panoramic radiograph, which was taken one year before exfoliation of the primary canine, shows evidence of pre-eruptive coronal resorption of the right permanent maxillary canine (arrow). Examination. Clinically, his oral hygiene was satisfactory and there was a soft tissue overgrowth between his maxillary right lateral incisor and first premolar. Enamel fragments were embedded in a pedunculated soft tissue overgrowth of normal to slightly erythematous colour (Fig. 1). It was apparent that the enamel and dentine of the canine had been almost totally resorbed (Fig. 1). A periapical radiograph (Fig. 2) showed no evidence of a retained primary canine. While a panoramic radiograph (Fig. 3), which had been taken about a year previously, revealed a coronal radiolucency resembling a carious lesion on the permanent canine with evidence of a retained primary canine. Treatment Following excisional biopsy under local analgesia, the histopathological examination of the soft tissue specimen from the crown was reported to be consistent with granulation tissue. A hard tissue fragment of dentine was colonised by a large aggregate of actinomyces like filamentous bacteria and neutrophilic infiltrates, intermixed with scanty inflamed granulation tissue and stratified squamous mucosa. There was evidence of polypoid squamous mucosa with a mildly hyperplastic epithelium. There was no evidence of dysplasia or malignancy. The overall features were said to be consistent European Archives of Paediatric Dentistry 99

3 N.M. Manan et al. Table 1. Hypotheses for the aetiology of pre-eruptive intra-coronal resorption reported in the literature. Author Blackwood, 1958; Brooks, 1988; Browne, 1954; Grundy et al., 1984 Giunta and Kaplan, 1981 Holan et al., 1994 Ignelzi et al., 1990; Luten, 1958 Kronfeld, 1955 Muhler, 1957 Seddon et al., 1996 Seow et al., 1999 a and b Skillen, 1941; Wooden and Kuftinee, 1974 Walton, 1980 with dental caries. The clinical and radiographic features plus the histopathological findings led to a diagnosis of idiopathic preeruptive coronal resorption. Treatment objectives were devised so as to improve the aesthetics, prevent further loss of tooth structure, maintain the space and improve the periodontal health. As part of the treatment plan, the alternatives were explained to the child and his parents. These included monitoring any further eruption; retain alveolar bone height and width for placement of an implant in the future, or space closure with an orthodontic fixed appliance if the root needed to be extracted. After considering all of the treatment options, a mutual decision was made to monitor the tooth for up to 12 months; the parents understood that this would temporarily compromise the aesthetics in the anterior region. Follow-up Aetiological factors Internal /external resorption Developmental abnormality in dentine Prolonged pre-eruptive period Developmental anomaly Break down of reduced enamel epithelium Apical inflammation of primary teeth Impacted teeth Ectopic position of particular tooth Caries Developmental abnormality in enamel During the follow-up period there has been no evidence of a sinus, or abscess clinically and no periapical pathology was found on any radiographs. However, after 18 months there was evidence of a sinus in relation to tooth 13, a periapical radiograph revealed an apical radiolucency (Fig. 4). As a consequence of the periapical infection, the prognosis of tooth 13 was considered to be poor even though the resorptive process had apparently ceased. Furthermore, the loss of bone and the chronic infection would be likely to compromise the success of any future implant. Therefore tooth 13 will only be retained whilst orthodontic treatment planning is being conducted, it should then be extracted as part of a long-term orthodontic treatment plan. This information was conveyed to the parents who favoured this approach. Discussion The coronal enamel and dentine resorption defect was evident radiographically before exfoliation of the primary canine (Fig. 3). In spite of the radiolucent area being identified preeruptively no active treatment could have knowingly been proposed. It has been reported that in unerupted teeth the resorptive process commences adjacent to the defective enamel epithelium [Blackwood, 1958], and progresses in an irregular manner from an isolated area within the enamel and subsequently continues to progress towards the dentine. Eventually, in order for complete crown resorption, the connective tissue has to be in direct contact with the enamel and maintain a vascular communication with the peripheral vessels. It has been reported that the tooth structure destroyed by this resorptive process is usually replaced by bone [Brooks, 1988]. The internal resorption begins at the junction of the pulp and dentine, and is at the expense of the dentine and so results in widening of the pulp chamber [Skillen, 1941; Browne, 1954; Muhler, 1957; Blackwood, 1958; Wooden and Kuftinee, 1974; Grundy et al., 1984; Brooks, 1988; Seddon et al., 1996; Seow et al., 1999 a and b; Nik and Rahman, 2003]. Lesions of intra-coronal resorption may appear radiographically to be similar to external resorption, which is commonly found in chronically unerupted or impacted teeth [Stafne and Austin, 1945]. The histopathological examination revealed that the soft tissue taken from this erupted tooth consisted of granulation tissue with bacterial invasion, while there could have been post-eruption contamination; such an appearance suggests that the histopathological appearance represents a carious lesion that occurred post-eruptively [Walton, 1980]. Brooks [1988] reported that long-term inflammation of the primary teeth might induce granulation tissue proliferation and an osteoclastic response. Furthermore, it is thought that this kind of resorptive process does not apply to the type of resorption found in permanent molars, because they do not have a primary predecessor similarly it does not apply to any permanent tooth in patients who have hypodontia where the primary predecessor is congenitally missing. This phenomenon may explain the different prevalence figures for the different tooth types. Nevertheless, Kronfeld [1955] gave an opinion that caries could not occur prior to eruption of a tooth. Several previous studies have reported that the pre-eruptive lesions are resorptive in nature [Browne, 1954; Luten, 1958; Blackwood, 1958; Wooden and Kuftinee, 1974; Walton, 1980; Grundy et al., 1984; Brooks, 1988; Owens et al., 1988; Ignelzi et al., 1990]. The clinical management depends on the extensiveness of the lesions. The greater the extent of a lesion the more rapid will be the rate of resorption. Progressive resorption leads to loss of the protective epithelium so that there is an invasion of connective tissue into the enamel and dentine [Blackwood, 1958]. 100 European Archives of Paediatric Dentistry 13 (Issue 2). 2012

4 Pre-eruptive coronal resorption of maxillary canine In the past the treatment recommended for an unerupted tooth was surgical exposure by removing the overlying soft tissue and curettage of the defects followed by lining with calcium hydroxide and restoration [Sullivan and Jolly, 1957; Wood and Crozier, 1985]. Multiple treatment options were considered and explained to the parents of the child reported herein, these included to monitor without any treatment and accept compromising the aesthetics, endodontic treatment followed by a post and core supported restoration; decoronisation of the tooth; and extraction followed by a resin bonded bridge as a temporary measure and to consider implants in the future. The parents were reluctant to have the tooth treated; they wanted it to be retained. Clinically it was considered impractical to retain the root because of the apparently progressive nature of the lesion; this further eliminated the option of extruding the remaining portion of the root prior to placement of a post-retained restoration. The basic principle behind the provision of treatment was that the prognosis of the tooth had to be good enough to retain the tooth for life and to avoid extraction of a sound tooth if space was required for orthodontic therapy. More recently it has been suggested that for the more progressive lesion, it is important to conserve the tooth by surgical exposure [Dowling et al., 1999]. Alternatively, if the lesion is thought to be slowly progressive, it should be allowed to erupt and then treatment can be commenced immediately upon eruption. Conclusion Teeth affected by idiopathic pre-eruptive coronal resorption can be retained if the problem is intercepted at an early stage and appropriate restorative procedures are performed. It is prudent to make an early diagnosis of the condition and to formulate short and long-term treatment plans, which may involve keeping the affected tooth to retain the alveolar bone height and width to allow for the option of planning an implant. References Blackwood HJJ. Resorption of enamel and dentine in the unerupted tooth. Oral Surg Oral Med Oral Pathol 1958; 11: Brooks JK. Detection of intracoronal resorption in an unerupted developing premolar: report of a case. J Am Dent Assoc 1988; 116: Browne WG. A histopathological study of resorption in some unerupted teeth. Dent Record 1954; 74: Dowling PA, Fleming P, Corcoran F. A case report of pre-eruptive coronal resorption in a mandibular canine. Dent Update 1999; 26: Giunta JL, Kaplan MA. "Caries-like" dentin radiolucency of unerupted permanent tooth from developmental defects. J Pedod 1981; 5: Grundy CE, Pyle RJ, Adkins KF. Intra-coronal resorption of unerupted molars. Aust Dent J 1984; 29: Holan G, Eidelman E, Mass S. Pre-eruptive coronal resorption of permanent teeth: report of three cases and their treatment. Pediatr Dent 1994; 16: Ignelzi MA Jr, Fields HW, White RP, Bengenholz G, Booth FA. Intracoronal radiolucencies within unerupted teeth: case report and review of the literature. Oral Surg Oral Med Oral Pathol 1990; 70: Kronfeld R. Histopathology of the teeth and their surrounding structures. 4th ed. Philadelphia: Lea & Febiger, 1955: Luten JR. Internal resorption or caries? A case report. J Dent Child 1958; 25: Muhler JC. The effect of apical inflammation of the primary teeth on dental caries in the permanent teeth. J Den Children 1957; 24: Nik NN, Rahman RA. Pre-eruptive intracoronal dentin defects of permanent teeth. J Clin Pediatr Dent 2003; 27: Owens PDA, Wangrangsimakul K, O Brien FV. Idiopathic external resorption of teeth. J Oral Pathol 1988; 17: Özden B, Acikgoz A. Prevalence and characteristics of intracoronal resorption in unerupted teeth in the permanent dentition: a retrospective study. Oral Radiol 2009; 25: Rankow H, Croll TP, Miller AS. Pre-eruptive idiopathic coronal resorption of permanent teeth in children. J Endodont 1986; 12: Seddon RP, Orth D, Smith PB. Early arrested development and coronal resorption of an impacted maxillary canine: report of case. J Dent Child 1996; 63: Seow WK, Hackley D. Pre-eruptive intracoronal radiolucent lesions in the permanent dentition: case report. Pediatr Dent 1996; 18: Seow WK, Lu PC, McAllan LH. Prevalence of pre-eruptive intracoronal dentin defects from panoramic radiographs. Pediatr Dent 1999a; 21: Seow WK, Wan A, McAllan LH. The prevalence of pre-eruptive dentin radiolucencies in the permanent dentition. Pediatr Dent 1999a; 21: Seow WK. Multiple pre-eruptive intracoronal radiolucent lesions in the permanent dentition: case report. Pediatr Dent 1998; 21: Skillen WG. So-called intra-follicular caries. Illinois. Dent J 1941; 10: Stafne EC, Slocumb CH. Idiopathic resorption of teeth. Am J Orthodont Oral Surg 1944; 30: Stafne EC, Austin LE. Resorption of embedded teeth. J Am Dent Assoc 1945; 32: Sullivan HR, Jolly M. Idiopathic resorption. Aus Dent J 1957; 2: Walton JL. Dentin radiolucencies in unerupted teeth: report of two cases. J Dent Child 1980; 47: Wood PF, Crozier DS. Radiolucent lesions resembling caries in the dentine of permanent teeth. A report of sixteen cases. Aust Dent J 1985; 30: Wooden EE, Kuftinec MM. Decay of unerupted premolar. Oral Surg Oral Med Oral Pathol 1974; 38: European Archives of Paediatric Dentistry 101

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W. Kim Seow, MDSc, DDSc, PhD, FRACDS P.C. Lu L. H. McAllan, BDS, MSc

Scientific Article Prevalence of pre-eruptive intracoronal dentin defects from panoramic radiographs W. Kim Seow, MDSc, DDSc, PhD, FRACDS P.C. Lu L. H. McAllan, BDS, MSc Dr. Seow is associate professor,