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

Received on March 18, 2009

Accepted on July 21, 2009

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 Sciences1-7-1 Sakamoto, Nagasaki 852-8588, JAPAN

Abstract Dentigerous cysts (DCs) are common cysts of the jaws and usually easy to be treated when small. However, extensive cysts involving three or more teeth are difficult to be managed. The purpose of this case report was to describe the management of an extensive DC in a child. The treatment 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.

only a few reports regarding treatment of extensive DCs, as noted in Table 1. In this report, we presented a case of an extensive DC associated with mandibular left canine, and first and second premolar teeth in a 12-year-old boy and reviewed the literature for extensive cases.

Case Report

A 12-year-old Japanese boy was referred to our hospital from a private dental clinic due to unde-tected mandibular left canine and first premolar on the periapical radiographs. Approximately the preceding 6 weeks ago, he had visited the dental clinic with a swelling in the left mandibular region. Root canal therapy had been performed in the primary mandibular left first molar.

On the initial visit to our hospital, a slight swelling of the left mandible was noted. There were no abnormal findings on general examination. Intraorally, the primary mandibular left canine and molars were retained and were dull to percussion, but showed no signs of mobility, gingival swelling or redness (Fig. 1). Panoramic radiograph showed a broad range of radiolucent lesions in the area extending from the mandibular left incisors to the primary second molar, impacted permanent canine and first premolar. The highly dense structure, which

Introduction

A dentigerous cyst (DC) is the second most common cyst of the jaws accounting for 18.1% of all odon-togenic cysts, and they are more frequent in males at a ratio of 1.861). DCs are diagnosed over a wide age range, with a peak incidence in the fifth1) or sixth decades2). The mandibular third molar region was by far the most common site of presentation accounting for 73.2%1). In pediatric populations aged 0–16 years, DCs account for approximately 30% of all odontogenic cysts3). If the cyst is small, removal or exfoliation of the primary tooth may lead to resolution. Some small cysts can be treated by enucleation and extraction of the involved tooth4,5), or marsupialization and orthodontic traction of the impacted tooth6–9). However, they are usually asymp-tomatic, and occasionally reach an extensive size unless they are discovered at an early stage on radio-graphic examination. The extensive DCs often block eruption of teeth, displace these teeth, and destroy the bone10). Motamedi et al. show that extensive DCs (involving three or more teeth) primarily occur between the ages of 10–19 years10). Management of extensive DCs is difficult5,10–19). However, there are

Key wordsDentigerous cysts,Marsupialization,Mixed dentition

Case Report

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was thought to be root canal filling material, extended into the cystic lesion (Fig. 2-a). According to the referral form, calcium hydroxide and iodoform paste (Vitapex, Neo Dental Chemical Products Co., Tokyo, Japan) had been used to fill the root canal. CT imaging was performed to three-dimensionally clarify the spatial expansion of the cyst (Fig. 3). CT images suggested that this large lesion was a dentigerous cyst associated with the crowns of the mandibular canine and first premolar.

Extraction of primary mandibular left canine and first and second molars and marsupialization were performed under general anesthesia by a dental surgeon. The foreign material was removed and confirmed to be Vitapex. A biopsy of the cystic lesion was taken, and gauze medicated with achromycin was inserted into the cyst cavity to prevent early wound closure and to keep it open. The microscopic diagnosis was DC (Fig. 4).

A removable space maintainer was placed in the mandibular arch to cover over the fenestrated cyst, in which medicated gauze was packed. Three months later, the buccal surface of the second premolar

could be observed through the defect (Fig. 5-a). A lingual arch appliance was then placed instead of the removable space maintainer, and traction of the second premolar was initiated (Fig. 5-b). Four months later, the second premolar erupted into the expected position (Fig. 5-c). The first premolar and canine spontaneously emerged into the oral cavity 9 and 10 months after marsupialization, respectively. A multibracket system was applied for alignment of the lower teeth, 18 months after marsupialization, the canine was arranged almost completely in the expected position (Fig. 6).

No recurrence of the cyst was noted 4.5 years after marsupialization (Fig. 2-b). It was showed that mandibular left canine was rotated, which was thought to be due to regression.

Discussion

DC arises from an extraordinary expansion of the dental follicle of an unerupted tooth and is seen attached to its cervix20). DC can be suspected if the follicular space on the radiograph is more than

Table 1 Published cases of extensive cysts involving three or more teeth

Ref. Year Sex

Age Location Treatment Follow up Recurrence (yrs.) (involved teeth) (extracted tooth)

18 2006 M 12 Mx. Right GA, Enucleation N/A N/A (12,13,14,15) (53,54,55,12,13,14,15)

19 2006 M 14 Md. Left GA, Enucleation N/A N/A (36,37,38) (36,37,38)

17 2004 M 6 Md. Right GA, Curettage 1 year No (85,46,47) (85,46,47)

14 2003 F 9 Md. Left GA, Marsupialization 30 months No (31,32,33,34,35) (73,74,75) All teeth erupted

13 2003 M 6 Md. Left GA, Marsupialization 6 months No (75,36,37) (75)

12 2001 F 7 Md. Left GA, Enucleation 6 months No (35,36,37) (74,75,35,36,37)

M 9 Md. Left LA, Marsupialization 3 years No 11 1999

(33,34,35) (73,74,75) All teeth erupted

F 8 Md. Left GA, Marsupialization 1 year No (33,34,35,36,37) (74)

16 1997 M 8 Md. Right GA, Fenestration 10 months No (43,44,45) (84,85)

15 1997 F 12 Md. Right LA, Marsupialization 18 months 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

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Fig. 1 Intraoral photographs taken at initial visit

Fig. 2 Panoramic radiographs taken at initial visit (a) and 4.5 years (b) after marsupialization

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).

Kamasaki, Y., Sasaki, Y. and Fujiwara, T.

237EXTENSIVE DENTIGEROUS CYST IN A GROWING CHILD

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 images also revealed expansion of the buccal cortex 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. Other lesions such as unicystic ameloblastoma and keratocysts should not be excluded only by radio-graphic examination. Histopathological analysis of the lesion is essential for a definitive diagnosis.

The treatment of DCs is generally dictated by cyst sizes and sites, patients’ age, the dentition involved, and involvement of vital structures10,23). Enucleation and extraction of the tooth are effective in the cases involving a single impaction such as a third molar in adult10), and are also performed for

small cysts in growing children4,5,23,24). Marsupializa-tion reduces the size of the cyst by relieving the hydraulic pressure, which may be responsible for cyst enlargement or displacing the involved tooth, and preserves the involved permanent tooth6,7,11,13–17,23). Although marsupialization has been applied suc-cessfully to treat DCs in growing children, there have been few reports of cases with extensive cyst involving three or more teeth, in which long-term follow-up to assess treatment results and recurrence was performed (Table 1). Motamedi et al.10) report

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 magnification 100)

Fig. 5 Photographs of the affected region taken at 3 months (a, b) and 4 months (c) after marsupialization

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26 cases of extensive DCs in children aged 0–19 years, and cyst enucleation and extraction of the impacted tooth or teeth are performed in 20 cases. In an extensive cyst, enucleation and extraction lead to a loss of several teeth4,5,12,17–19,23,24), which results in functional, cosmetic and psychological demerits for children. In six cases11,13–16) of the previous papers (Table 1) and our case, marsupialization was suc-cessful in saving the involved permanent teeth. Two children aged below 10 years show normal eruption of the involved teeth without orthodontic interven-tion11,14). These case reports suggest that teeth of growing children with immature roots have great eruptive potential, and children have a great capacity to regenerate the bony structure. In consideration of these characteristics of children, extensive DCs in children should be treated differently from those in adults. Miyawaki et al.25) report that cyst shrinkage can promote tooth eruption during the first 3 months after marsupialization. The cyst cavity must be kept opened in order to shrink by relief of intracystic pressure, and at the same time must be protected

against infection in the oral cavity. In our case, the removable space maintainer was applied for 3 months after the marsupialization, since it enabled to protect the operative wound by covering over cyst cavity, in which medicated gauze was packed, as well as to maintain space for the unerupted teeth. Then, minimal orthodontic intervention was initiated. The second premolar emerged by traction for 1 month, the canine and the first premolar erupted spontaneously. Our case indicated that this proce-dure was a successful treatment for an extensive DC involving three teeth. Based on the previous papers7,9–11,13–16,20,25,26) and our case, marsupialization is thought to be useful in facilitating the eruption of impacted teeth associated with DCs in the growing child. If there were adequate space for eruption and favorable cooperation of the patient, it would be better to treat the pediatric patient with an extensive DC by marsupialization and preservation of the impacted teeth. It was thought to be less demanding method for children.

Fig. 6 Intraoral photographs taken at 18 months after marsupialization

Kamasaki, Y., Sasaki, Y. and Fujiwara, T.

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References

1) Jones, A.V., Craig, G.T. and Franklin, C.D.: Range and demographics of odontogenic cysts diagnosed in a UK population over a 30-year period. J Oral Pathol Med 35: 500–507, 2006.

2) Dunsche, A., Babendererde, O., Luttges, J. and Springer, I.N.: Dentigerous cyst versus unicystic ameloblastoma — differential diagnosis in routine histology. J Oral Pathol Med 32: 486–491, 2003.

3) Jones, A.V. and Franklin, C.D.: An analysis of oral and maxillofacial pathology found in children over a 30-year period. Int J Paediatr Dent 16: 19–30, 2006.

4) Asian-Gonzalez, E., Pereira-Maestre, M., Conde-Fernandez, D., Vilchez, I., Segura-Egea, J.J. and Gutierrez-Perez, J.L.: Dentigerous cyst associated with a formocresol pulpotomized deciduous molar. J Endod 33: 488–492, 2007.

5) Kalaskar, R.R., Tiku, A. and Damle, S.G.: Dentiger-ous cysts of anterior maxilla in a young child: A case report. J Indian Soc Pedod Prev Dent 25: 187–190, 2007.

6) Delbem, A.C., Cunha, R.F., Afonso, R.L., Bianco, K.G. and Idem, A.P.: Dentigerous cysts in primary dentition: report of 2 cases. Pediatr Dent 28: 269–272, 2006.

7) Martinez-Perez, D. and Varela-Morales, M.: Conser-vative treatment of dentigerous cysts in children: a report of 4 cases. J Oral Maxillofac Surg 59: 331–333, 2001.

8) Rubin, D.M., Vedrenne, D. and Portnof, J.E.: Ortho-dontically guided eruption of mandibular second premolar following enucleation of an inflammatory cyst: case report. J Clin Pediatr Dent 27: 19–23, 2002.

9) Takagi, S. and Koyama, S.: Guided eruption of an impacted second premolar associated with a denti-gerous cyst in the maxillary sinus of a 6-year-old child. J Oral Maxillofac Surg 56: 237–239, 1998.

10) Motamedi, M.H. and Talesh, K.T.: Management of extensive dentigerous cysts. Br Dent J 198: 203–206, 2005.

11) Kozelj, V. and Sotosek, B.: Inflammatory dentigerous cysts of children treated by tooth extraction and decompression — report of four cases. Br Dent J 187: 587–590, 1999.

12) Counts, A.L., Kochis, L.A., Buschman, J. and Savant, T.D.: An aggressive dentigerous cyst in a seven-year-old child. ASDC J Dent Child 68: 268–271, 2001.

13) Jones, T.A., Perry, R.J. and Wake, M.J.: Marsupial-ization of a large unilateral mandibular dentigerous cyst in a 6-year-old boy — a case report. Dent Update 30: 557–561, 2003.

14) Ertas, U. and Yavuz, M.S.: Interesting eruption of 4 teeth associated with a large dentigerous cyst in mandible by only marsupialization. J Oral Maxillofac Surg 61: 728–730, 2003.

15) Fortin, T., Coudert, J.L., Francois, B., Huet, A., Niogret, F., Jourlin, M. and Gremillet, P.: Marsupi-alization of dentigerous cyst associated with foreign body using 3D CT images: a case report. J Clin Pediatr Dent 22: 29–33, 1997.

16) Ziccardi, V.B., Eggleston, T.I. and Schneider, R.E.: Using fenestration technique to treat a large dentig-erous cyst. J Am Dent Assoc 128: 201–205, 1997.

17) Kawamura, J.Y., de Magalhaes, R.P., Sousa, S.C. and Magalhaes, M.H.: Management of a large dentigerous cyst occurring in a six-year-old boy. J Clin Pediatr Dent 28: 355–357, 2004.

18) Shashikiran, N.D., Kumar, N.C. and Reddy, V.V.: Unusual presentation of inverted impacted premolars as a result of dentigerous cyst: a case report. J Indian Soc Pedod Prev Dent 24: 97–99, 2006.

19) Freitas, D.Q., Tempest, L.M., Sicoli, E. and Lopes-Neto, F.C.: Bilateral dentigerous cysts: review of the literature and report of an unusual case. Dentomaxil-lofac Radiol 35: 464–468, 2006.

20) Benn, A. and Altini, M.: Dentigerous cysts of inflam-matory origin. A clinicopathologic study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 81: 203–209, 1996.

21) Ko, K.S., Dover, D.G. and Jordan, R.C.: Bilateral dentigerous cysts — report of an unusual case and review of the literature. J Can Dent Assoc 65: 49–51, 1999.

22) Ustuner, E., Fitoz, S., Atasoy, C., Erden, I. and Akyar, S.: Bilateral maxillary dentigerous cysts: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 95: 632–635, 2003.

23) Sands, T. and Tocchio, C.: Multiple dentigerous cysts in a child. Oral Health 88: 27–29, 1998.

24) Lustig, J.P., Schwartz-Arad, D. and Shapira, A.: Odontogenic cysts related to pulpotomized decidu-ous molars: clinical features and treatment outcome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 87: 499–503, 1999.

25) Miyawaki, S., Hyomoto, M., Tsubouchi, J., Kirita, T. and Sugimura, M.: Eruption speed and rate of angulation change of a cyst-associated mandibular second premolar after marsupialization of a dentig-erous cyst. Am J Orthod Dentofacial Orthop 116: 578–584, 1999.

26) Hyomoto, M., Kawakami, M., Inoue, M. and Kirita, T.: Clinical conditions for eruption of maxillary canines and mandibular premolars associated with dentigerous cysts. Am J Orthod Dentofacial Orthop 124: 515–520, 2003.

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