o o o

Vol. 97 No. 1 January 2004

ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY ORAL AND MAXILLOFACIAL PATHOLOGY
Editor: Alan R. Gould

Benign cementoblastoma involving multiple maxillary teeth: Report of a case with a review of the literature
Kousuke Ohki, DDS,a Hiroyuki Kumamoto, DDS, PhD,b Yasutaka Nitta, DDS, PhD,c Hiroshi Nagasaka, DDS, PhD,c Hiroshi Kawamura, DDS, PhD,d and Kiyoshi Ooya, DDS, PhD,e Sendai, Japan
TOHOKU UNIVERSITY

A rare case of benign cementoblastoma involving multiple deciduous and permanent teeth is presented with a review of the literature. A 12-year-old boy was admitted for a swelling in the right maxillary premolar-molar region. A radiologic examination revealed a well-dened, round, radiopaque mass extending from the right maxillary rst premolar to the second permanent molar. The tumor was removed with all associated teeth. A histologic examination of the surgical specimen revealed a well-circumscribed tumor composed of cementum-like tissue surrounded by a brous capsule. The tumor was attached to the roots of the second deciduous molar, rst premolar, and the rst and second permanent molars and embedded in the crown and root of the right maxillary second premolar, suggesting that the lesion had arisen from the second deciduous molar. There has been no recurrence of the lesion more than 18 months after the surgical procedure. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;97:53-8)

Benign cementoblastoma, rst described by Dewey in 1927,1 is a relatively rare tumor of odontogenic ectomesenchyme origin characterized by proliferating cementum-like tissue occurring in juxtaposition to tooth roots.2-4 This tumor accounts for 0.8% to 2.6% of all odontogenic tumors.5-7 Nearly all benign cementoblastomas are closely related to and partly surround a root or roots of a single erupted permanent tooth.2,8 We present an unusual case of benign cementoblastoma involving multiple maxillary deciduous and permanent teeth.

Graduate Student, Division of Maxillofacial Surgery, Department of Oral Medicine and Surgery, Graduate School of Dentistry. b Assistant Professor, Division of Oral Pathology, Department of Oral Medicine and Surgery, Graduate School of Dentistry. c Assistant Professor, Division of Maxillofacial Surgery, Department of Oral Medicine and Surgery, Graduate School of Dentistry. d Professor, Division of Maxillofacial Surgery, Department of Oral Medicine and Surgery, Graduate School of Dentistry. e Professor, Division of Oral Pathology, Department of Oral Medicine and Surgery, Graduate School of Dentistry. Received for publication May 21, 2003; returned for revision Jul 9, 2003; accepted for publication Aug 1, 2003. 1079-2104/$ - see front matter 2004, Elsevier Inc. All rights reserved. doi:10.1016/j.tripleo.2003.08.012

Fig 1. Intraoral appearance showing marked expansion of the right posterior portion of the maxilla.

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ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY January 2004

Fig 2. The radiologic features of our patient. A, A panoramic radiograph reveals a radiopaque mass of the apical region extending from the right maxillary rst premolar to the second permanent molar. The lesion contains the unerupted second premolar. B, An axial computed tomograph depicts a well-dened, high-density mass in the right maxillary premolar-molar region. The lesion, which involves the crown of the second premolar, is attached to the roots of the rst premolar, the second deciduous molar, and the rst permanent molar.

CASE REPORT
A 12-year-old boy was admitted to the Department of Maxillofacial Surgery of Tohoku University Dental Hospital (Sendai, Japan) for a swelling in the right maxillary region. The patient was in good general health, but he had a history of atopic dermatitis in infancy. The maxillary swelling had been noticed 2 months before admission. An intraoral examination revealed marked enlargement of the posterior portion of the maxilla (Fig 1). The swelling was bony and tender. The overlying mucosa was normal, and all associated teeth were immobile and symptom-free. A vitality test with an electrical pulp tester yielded a negative reaction for the right maxillary rst premolar and a weakly positive reaction for the right maxillary second deciduous molar and rst permanent molar. A panoramic radiographic examination revealed a round, radiopaque mass measuring 3.7 3.0 cm and extending from the right maxillary rst premolar to the second permanent molar (Fig 2, A). Axial computed tomographs revealed a well-dened, high-density mass in the right maxillary premolar-molar region (Fig 2, B). This lesion was attached to the roots of the right maxillary second deciduous molar, rst premolar, and the rst and second permanent molars. Both the crown and the root of the unerupted second premolar were embedded in the lesion. On the basis of the clinical and radiologic features of the lesion, a maxillary tumor was suspected. An incisional biopsy was performed with the patient under local anesthesia, and the histopathologic diagnosis was benign cementoblastoma. While the patient was under general anesthesia, a trapezoid palatal and buccal mucoperiosteal ap was raised in the right maxillary premolar-molar region. The right side of the maxillary sinus oor had been lifted by the tumor in the alveolar region, but the sinus mucosa was intact. A round tumor was removed with the associated right maxillary second deciduous molar, the rst and second premolars,

Fig 3. On the buccal aspect of the surgical specimen, a well-circumscribed round tumorous mass involving the rst premolar, the second deciduous molar, and the rst and second permanent molars is observed.

and the rst and second permanent molars. The wound was irrigated and closed. Soft tissue healing was complete by 3 weeks. There has been no recurrence of the lesion as of the time of this writing. The extirpated mass contained tumor tissue and 5 involved

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Fig 4. Gross appearance of buccopalatal slices of the decalcied specimen. A, A round tumor located in the apical portion of the right maxillary second deciduous molar. The second premolar is embedded in this lesion. B, A tumor attached to the right maxillary rst permanent molar, with resorbed roots.

teeth: the rst and second premolars, the second deciduous molar, and the rst and second permanent molars (Fig 3). The calcied tumor mass measured 3.5 3.0 3.1 cm. The entire bony surface was irregular and covered with a thin capsule of soft tissue. Serial buccopalatal slices at intervals of 0.5 cm disclosed a round whitish tumor located in the apical portion of the right maxillary second deciduous molar (Fig 4, A). The tumor embedded the crown and root of the right maxillary second premolar and was attached to the rst premolar and the rst and second permanent molars (Fig 4, B). A histologic examination of the decalcied surgical specimen revealed a well-circumscribed tumor involving the adjacent teeth. The tumor was composed of cementum-like tumor tissue with irregular lacunae, entrapped cells, and numerous reversal lines and was fused to the cementum on the root surface (Fig 5, A). At the periphery of the mass, tumor tissue was arranged in radiating trabeculae; the tumor was surrounded by a brous capsule (Fig 5, B). The roots of the right maxillary second deciduous molar, second premolar, and rst permanent molar had been resorbed, and the tumor had apparently invaded the palatal root canals of the second deciduous molar and rst permanent molar (Fig 5, C). The pulpal tissues were vital, and no inammatory change was found.

DISCUSSION Benign cementoblastoma is a neoplasm of the jaws2-4 most commonly found in the second and third decades of life.8 Some studies have reported that this tumors arises slightly more frequently in males2,9,10 or in females,8 whereas others have found no difference between the sexes.11,12 Virtually all benign cementoblastomas occur in the premolar-molar region, more commonly in the mandible than the maxilla.2-4,8,11 In this study, the tumor occurred in the right maxillary

premolar-molar region of a 12-year-old boy. Benign cementoblastoma is most often associated with a single erupted permanent tooth.2-4,8 In our patient, the tumor was located in the apical portion of the right maxillary second deciduous molar and involved the rst and second premolars, in addition to the rst and second permanent molars, suggesting that the lesion had originated in the second deciduous molar. Most reported cases of benign cementoblastoma of deciduous tooth origin were associated with mandibular second deciduous molars (Table I).11,13-18 The mandible is the more common site of benign cementoblastoma, yet benign cementoblastomas involving multiple teeth more commonly occur in the maxilla (Table II).8,11,13,19-25 Such cementoblastomas have been reported to reect the high growth potential of these tumors; however, no recurrence has been described. Benign cementoblastoma is histopathologically characterized by the formation of sheets of cementum-like tissue containing many reversal lines, irregular lacunae, and cellular brovascular stromata. The periphery of the mass or the more-active growth areas are often unmineralized.2-4 This tumor may sometimes resemble osteoblastoma, osteoid osteoma, or atypical osteosarcoma, which are not distinctively related to tooth roots, and may be difcult to distinguish from these tumors.2,21,26-28 Our patient had characteristic hard tissue formation close to the root surface cementum and was diagnosed as having benign cementoblastoma. Despite histologic evidence of vital, noninamed pulp tissue, the associated tooth may be unresponsive to vitality tests with an electrical pulp tester.3,30 This paradox has

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ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY January 2004

Fig 5. Histopathologic ndings. A, Cementum-like tumor tissue with irregular lacunae, entrapped cells, and numerous reversal lines fused to the tooth root (hematoxylin-eosin, original magnication 50). B, Tumor tissue is arranged in radiating trabeculae at the periphery and surrounded by a brous capsule (hematoxylin-eosin, original magnication 30). C, Tumor tissue invades the palatal root canal of the rst permanent molar. The pulpal tissue is vital and noninamed (hematoxylin-eosin, original magnication 10).

Table I. Reported cases of cementoblastoma associated with deciduous teeth

Author Chaput and Marc Vilasco et al14 Zachariades et al11
13

Year 1965 1969 1985

Age (y)/sex 10/F 8/F 7/F

Location Right mandibular rst premolar and second deciduous molar Right mandibular second deciduous molar Right mandibular rst and second deciduous molars, and rst permanent molar Left mandibular rst and second deciduous molars Mandibular deciduous central incisors Right mandibular second deciduous molar Right mandibular second deciduous molar Right maxillary second deciduous molar, rst and second premolars, and rst and second permanent molars

Size (cm) 2.1 ND 3.0

Pain

Recurrence ND ND

Herzog15 Papageorge et al16 Cannell17 Schafer et al18 Present case

1987 1987 1991 2001 2003

7/F 6/M 8/F 8/F 12/M

1.4 4.5 ND 1.5 3.3

ND

ND

ND, No data.

been proposed as suggestive of the disruption of normal neural impulse transmission, which occurs because the tumor encompasses the root apex.10,20 In our patient, the associated right maxillary second deciduous molar

was weakly responsive to vitality testing, but its pulp tissue was histopathologically vital and noninamed. Because benign cementoblastoma has unlimited growth potential, the usual treatment is complete sur-

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Table II. Reported cases of cementoblastoma involving multiple teeth

Author Wertheimer et al
19

Year 1961 1965

Age (y)/sex 14/M 10/F

Location Right maxillary rst and second permanent molars Right mandibular rst premolar and second deciduous molar Right maxillary rst and second permanent molars Left maxillary second premolar, and rst, second, and third permanent molars Left maxillary rst and second permanent molars Right mandibular rst and second deciduous molars, and rst permanent molar Left mandibular rst and second deciduous molars Right maxillary rst, second and impacted third permanent molars Right maxillary rst and second permanent molars Right mandibular central and lateral incisors, canine, rst and second premolars, and second permanent molar Right mandibular rst premolar, and rst and second permanent molars Right maxillary second deciduous molar, rst and second premolars, and rst and second permanent molars

Size (cm) 2.5 2.1

Pain

Recurrence ND ND

Chaput and Marc13

Abrams et al20 Corio et al21

1974 1976

17/M 19/M

ND 4.5 ND

Brocheriou et al22 Zachariades et al11

1979 1985

18/F 7/F

3.0 3.0

ND

Herzog15 Garlick et al23

1987 1990

7/F 19/M

1.4 3.5

ND

Slootweg24 Jelic et al25

1992 1993

22/M 18/M

ND 5.0

ND

ND ND

Ulmansky et al8

1994

20/F

3.5

ND

Present case

2003

12/M

3.3

ND, No data.

gical excision with extraction of the associated teeth, even though the pulp may be vital.10,20,21,30-33 Recurrence does not occur if the mass is totally removed; however, incomplete excision is usually followed by recurrence.3,20,34-36 Because the present tumor involved multiple maxillary teeth, it was extirpated with the right maxillary second deciduous molar, the rst and second premolars, and the rst and second permanent molars. There has been no recurrence more than 18 months after the surgical procedure.
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