Management of an Extensive Dentigerous Cyst in a 12-Year-Old Boy

PEDIATRIC DENTAL JOURNAL 19(2): 234–239, 2009

Case Report Management of an extensive dentigerous cyst in a 12-year-old boy

Yoko Kamasaki, Yasunori Sasaki and Taku Fujiwara

Department of Pediatric Dentistry, Nagasaki University Graduate School of Biomedical Sciences 1-7-1 Sakamoto, Nagasaki 852-8588, JAPAN

Abstract Dentigerous cysts (DCs) are common cysts of the jaws and usually Key words easy to be treated when small. However, extensive cysts involving three or Dentigerous cysts, more teeth are difficult to be managed. The purpose of this case report was Marsupialization, to describe the management of an extensive DC in a child. The treatment Mixed dentition instituted was the marsupialization with the extraction of the involved deciduous teeth. This treatment allowed rapid healing of the lesion and eruption of the permanent teeth with minimum orthodontic therapy.

Introduction only a few reports regarding treatment of extensive DCs, as noted in Table 1. In this report, we presented A dentigerous cyst (DC) is the second most common a case of an extensive DC associated with mandibular cyst of the jaws accounting for 18.1% of all odon- left canine, and first and second premolar teeth in togenic cysts, and they are more frequent in males a 12-year-old boy and reviewed the literature for at a ratio of 1.861). DCs are diagnosed over a wide extensive cases. age range, with a peak incidence in the fifth1) or 2) sixth decades . The mandibular third molar region Case Report was by far the most common site of presentation accounting for 73.2%1). In pediatric populations aged A 12-year-old Japanese boy was referred to our 0–16 years, DCs account for approximately 30% of hospital from a private dental clinic due to unde- all odontogenic cysts3). If the cyst is small, removal tected mandibular left canine and first premolar or exfoliation of the primary tooth may lead to on the periapical radiographs. Approximately the ­resolution. Some small cysts can be treated by preceding 6 weeks ago, he had visited the dental enucleation and extraction of the involved tooth4,5), clinic with a swelling in the left mandibular region. or marsupialization and orthodontic traction of the Root canal therapy had been performed in the impacted tooth6–9). However, they are usually asymp- ­primary mandibular left first molar. tomatic, and occasionally reach an extensive size On the initial visit to our hospital, a slight unless they are discovered at an early stage on radio- ­swelling of the left mandible was noted. There graphic examination. The extensive DCs often block were no abnormal findings on general examination. eruption of teeth, displace these teeth, and destroy Intraorally, the primary mandibular left canine and the bone10). Motamedi et al. show that extensive molars were retained and were dull to percussion, DCs (involving three or more teeth) primarily occur but showed no signs of mobility, gingival swelling between the ages of 10–19 years10). Management of or redness (Fig. 1). Panoramic radiograph showed extensive DCs is difficult5,10–19). However, there are a broad range of radiolucent lesions in the area extending from the mandibular left incisors to the Received on March 18, 2009 primary second molar, impacted permanent canine Accepted on July 21, 2009 and first premolar. The highly dense structure, which

234 ExtensivE DENTIgerous CysT IN A growing Child 235

Table 1 Published cases of extensive cysts involving three or more teeth Age Location Treatment Ref. Year Sex (yrs.) (involved teeth) (extracted tooth) Follow up Recurrence Mx. Right GA, Enucleation 18 2006 M 12 N/A N/A (12,13,14,15) (53,54,55,12,13,14,15) Md. Left GA, Enucleation 19 2006 M 14 N/A N/A (36,37,38) (36,37,38) Md. Right GA, Curettage 17 2004 M 6 1 year No (85,46,47) (85,46,47) Md. Left GA, Marsupialization 30 months 14 2003 F 9 No (31,32,33,34,35) (73,74,75) All teeth erupted Md. Left GA, Marsupialization 13 2003 M 6 6 months No (75,36,37) (75) Md. Left GA, Enucleation 12 2001 F 7 6 months No (35,36,37) (74,75,35,36,37) Md. Left LA, Marsupialization 3 years M 9 No (33,34,35) (73,74,75) All teeth erupted 11 1999 Md. Left GA, Marsupialization F 8 1 year No (33,34,35,36,37) (74) Md. Right GA, Fenestration 16 1997 M 8 10 months No (43,44,45) (84,85) Md. Right 18 months 15 1997 F 12 LA, Marsupialization No (43,44,45) All teeth erupted Ref., reference; M, male; F, female; Md., mandibular; Mx., maxillary; GA, general anesthesia; LA, local anesthesia; N/A, not available

was thought to be root canal filling material, extended could be observed through the defect (Fig. 5-a). A into the cystic lesion (Fig. 2-a). According to the lingual arch appliance was then placed instead of referral form, calcium hydroxide and iodoform paste the removable space maintainer, and traction of (Vitapex, Neo Dental Chemical Products Co., Tokyo, the second premolar was initiated (Fig. 5-b). Four Japan) had been used to fill the root canal. CT months later, the second premolar erupted into the imaging was performed to three-dimensionally clarify expected position (Fig. 5-c). The first premolar and the spatial expansion of the cyst (Fig. 3). CT images canine spontaneously emerged into the oral cavity 9 suggested that this large lesion was a dentigerous and 10 months after marsupialization, respectively. cyst associated with the crowns of the mandibular A multibracket system was applied for alignment of canine and first premolar. the lower teeth, 18 months after marsupialization, Extraction of primary mandibular left canine the canine was arranged almost completely in the and first and second molars and marsupialization expected position (Fig. 6). were performed under general anesthesia by a dental No recurrence of the cyst was noted 4.5 years surgeon. The foreign material was removed and after marsupialization (Fig. 2-b). It was showed confirmed to be Vitapex. A biopsy of the cystic lesion that mandibular left canine was rotated, which was was taken, and gauze medicated with achromycin thought to be due to regression. was inserted into the cyst cavity to prevent early wound closure and to keep it open. The microscopic Discussion diagnosis was DC (Fig. 4). A removable space maintainer was placed in the DC arises from an extraordinary expansion of the mandibular arch to cover over the fenestrated cyst, dental follicle of an unerupted tooth and is seen in which medicated gauze was packed. Three months attached to its cervix20). DC can be suspected if later, the buccal surface of the second premolar the follicular space on the radiograph is more than 236 Kamasaki, Y., Sasaki, Y. and Fujiwara, T.

Fig. 1 Intraoral photographs taken at initial visit

Fig. 3 Axial section views of the mandibular dentascan at three different levels (a) View from the depth of cervical area of the mandibular right second premolar. Follicular space of the left second premolar is enlarged (arrow) and connected with the cystic lesion, which displaces the roots of mandibular incisors (broad arrow). (b) View from the depth of the middle of the right canine root. Two cystic lesions connected to each other are observed. One cystic lesion in the middle of the mandible encloses the left canine, and the other encloses the left first premolar and some high-density structures (arrow). (c) View from the depth of the periapical area of the right canine root. The two cystic lesions are independent. The crown of the left canine points to the labial side (arrow).

Fig. 2 Panoramic radiographs taken at initial visit (a) and 4.5 years (b) after marsupialization ExtensivE DENTIgerous CysT IN A growing Child 237

Fig. 4 The specimen shows the cyst wall composed of the fibrous connective tissue lined by squamous epithelium Round cell infiltration is found below the epithelium. (Hematoxylin-eosin stain, original magnification2100)

5 mm21). On radiographic images, DC appears as unilocular radiolucent lesion of varying sizes, asso- ciated with the crown of an unerupted tooth19,22). In extensive cyst cases, CT imaging is useful since radiolucent lesions occasionally show a multilocular pattern10,17). The CT images of our case provided important information as demonstrated in Fig. 3. The cystic lesion was seemed to be derived from two independent cysts and connected to each other. The CT clearly showed that one cystic lesion was attached to the cervix of the labially-inclined canine, and the other was attached to the cervix of the ­mesially inclined first premolar and enclosed some high-density structures, and those two lesions and the enlarged follicular space of the second premolar were merged in the upper part of the mandible. CT Fig. 5 Photographs of the affected region taken at 3 months images also revealed expansion of the buccal cortex (a, b) and 4 months (c) after marsupialization which is intact, and helped to rule out solid and fibro-osseous lesions22). Although involvement of the tooth and cortical expansion are characteristic of DCs, there is a possibility of a more aggressive lesion. small cysts in growing children4,5,23,24). Marsupializa- Other lesions such as unicystic ameloblastoma and tion reduces the size of the cyst by relieving the keratocysts should not be excluded only by radio- hydraulic pressure, which may be responsible for cyst graphic examination. Histopathological analysis of enlargement or displacing the involved tooth, and the lesion is essential for a definitive diagnosis. preserves the involved permanent tooth6,7,11,13–17,23). The treatment of DCs is generally dictated by Although marsupialization has been applied suc- cyst sizes and sites, patients’ age, the dentition cessfully to treat DCs in growing children, there involved, and involvement of vital structures10,23). have been few reports of cases with extensive cyst Enucleation and extraction of the tooth are effective involving three or more teeth, in which long-term in the cases involving a single impaction such as a follow-up to assess treatment results and recurrence third molar in adult10), and are also performed for was performed (Table 1). Motamedi et al.10) report 238 Kamasaki, Y., Sasaki, Y. and Fujiwara, T.

Fig. 6 Intraoral photographs taken at 18 months after marsupialization

26 cases of extensive DCs in children aged 0–19 against infection in the oral cavity. In our case, years, and cyst enucleation and extraction of the the removable space maintainer was applied for 3 impacted tooth or teeth are performed in 20 cases. months after the marsupialization, since it enabled In an extensive cyst, enucleation and extraction lead to protect the operative wound by covering over cyst to a loss of several teeth4,5,12,17–19,23,24), which results cavity, in which medicated gauze was packed, as in functional, cosmetic and psychological demerits well as to maintain space for the unerupted teeth. for children. In six cases11,13–16) of the previous papers Then, minimal orthodontic intervention was initiated. (Table 1) and our case, marsupialization was suc- The second premolar emerged by traction for 1 cessful in saving the involved permanent teeth. Two month, the canine and the first premolar erupted children aged below 10 years show normal eruption spontaneously. Our case indicated that this proce- of the involved teeth without orthodontic interven- dure was a successful treatment for an extensive tion11,14). These case reports suggest that teeth of DC involving three teeth. Based on the previous growing children with immature roots have great papers7,9–11,13–16,20,25,26) and our case, marsupialization eruptive potential, and children have a great capacity is thought to be useful in facilitating the eruption of to regenerate the bony structure. In consideration of impacted teeth associated with DCs in the growing these characteristics of children, extensive DCs in child. If there were adequate space for eruption and children should be treated differently from those in favorable cooperation of the patient, it would be adults. Miyawaki et al.25) report that cyst shrinkage better to treat the pediatric patient with an extensive can promote tooth eruption during the first 3 months DC by marsupialization and preservation of the after marsupialization. The cyst cavity must be kept impacted teeth. It was thought to be less demanding opened in order to shrink by relief of intracystic method for children. pressure, and at the same time must be protected ExtensivE DENTIgerous CysT IN A growing Child 239

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