Vol. 97 No. 1 January 2004

ORAL AND MAXILLOFACIAL PATHOLOGY Editor: Alan R. Gould

Benign cementoblastoma involving multiple maxillary teeth:Report of a case with a review of the literatureKousuke Ohki, DDS,a Hiroyuki Kumamoto, DDS, PhD,b Yasutaka Nitta, DDS, PhD,c

Hiroshi Nagasaka, DDS, PhD,c Hiroshi Kawamura, DDS, PhD,d andKiyoshi Ooya, DDS, PhD,e Sendai, JapanTOHOKU UNIVERSITY

A rare case of benign cementoblastoma involving multiple deciduous and permanent teeth is presented witha 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-defined, round, radiopaque mass extending from the right maxillary firstpremolar to the second permanent molar. The tumor was removed with all associated teeth. A histologic examinationof the surgical specimen revealed a well-circumscribed tumor composed of cementum-like tissue surrounded by afibrous capsule. The tumor was attached to the roots of the second deciduous molar, first premolar, and the first andsecond permanent molars and embedded in the crown and root of the right maxillary second premolar, suggesting thatthe lesion had arisen from the second deciduous molar. There has been no recurrence of the lesion more than 18months after the surgical procedure. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;97:53-8)

Benign cementoblastoma, first described by Dewey in1927,1 is a relatively rare tumor of odontogenic ecto-mesenchyme origin characterized by proliferating ce-mentum-like tissue occurring in juxtaposition to toothroots.2-4 This tumor accounts for 0.8% to 2.6% of allodontogenic tumors.5-7 Nearly all benign cementoblas-tomas are closely related to and partly surround a rootor roots of a single erupted permanent tooth.2,8 Wepresent an unusual case of benign cementoblastomainvolving multiple maxillary deciduous and permanentteeth.

aGraduate Student, Division of Maxillofacial Surgery, Department ofOral Medicine and Surgery, Graduate School of Dentistry.bAssistant Professor, Division of Oral Pathology, Department of OralMedicine and Surgery, Graduate School of Dentistry.cAssistant Professor, Division of Maxillofacial Surgery, Departmentof Oral Medicine and Surgery, Graduate School of Dentistry.dProfessor, Division of Maxillofacial Surgery, Department of OralMedicine and Surgery, Graduate School of Dentistry.eProfessor, Division of Oral Pathology, Department of Oral Medicineand 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 theright posterior portion of the maxilla.

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CASE REPORTA 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 historyof atopic dermatitis in infancy. The maxillary swelling hadbeen noticed 2 months before admission. An intraoral exam-ination revealed marked enlargement of the posterior portionof the maxilla (Fig 1). The swelling was bony and tender. Theoverlying mucosa was normal, and all associated teeth wereimmobile and symptom-free. A vitality test with an electricalpulp tester yielded a negative reaction for the right maxillaryfirst premolar and a weakly positive reaction for the rightmaxillary second deciduous molar and first permanent molar.A panoramic radiographic examination revealed a round,radiopaque mass measuring 3.7 � 3.0 cm and extending fromthe right maxillary first premolar to the second permanentmolar (Fig 2, A). Axial computed tomographs revealed awell-defined, high-density mass in the right maxillary premo-lar-molar region (Fig 2, B). This lesion was attached to theroots of the right maxillary second deciduous molar, firstpremolar, and the first and second permanent molars. Both thecrown and the root of the unerupted second premolar wereembedded in the lesion. On the basis of the clinical andradiologic features of the lesion, a maxillary tumor was sus-pected. An incisional biopsy was performed with the patientunder local anesthesia, and the histopathologic diagnosis wasbenign cementoblastoma. While the patient was under generalanesthesia, a trapezoid palatal and buccal mucoperiosteal flapwas raised in the right maxillary premolar-molar region. Theright side of the maxillary sinus floor had been lifted by thetumor in the alveolar region, but the sinus mucosa was intact.A round tumor was removed with the associated right max-illary second deciduous molar, the first and second premolars,

and the first and second permanent molars. The wound wasirrigated and closed. Soft tissue healing was complete by 3weeks. There has been no recurrence of the lesion as of thetime of this writing.

The extirpated mass contained tumor tissue and 5 involved

Fig 2. The radiologic features of our patient. A, A panoramic radiograph reveals a radiopaque mass of the apical region extendingfrom the right maxillary first premolar to the second permanent molar. The lesion contains the unerupted second premolar. B, Anaxial computed tomograph depicts a well-defined, 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 first premolar, the second deciduous molar, andthe first permanent molar.

Fig 3. On the buccal aspect of the surgical specimen, awell-circumscribed round tumorous mass involving the firstpremolar, the second deciduous molar, and the first andsecond permanent molars is observed.

54 Ohki et al ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGYJanuary 2004

teeth: the first and second premolars, the second deciduousmolar, and the first and second permanent molars (Fig 3). Thecalcified tumor mass measured 3.5 � 3.0 � 3.1 cm. Theentire bony surface was irregular and covered with a thincapsule of soft tissue. Serial buccopalatal slices at intervals of0.5 cm disclosed a round whitish tumor located in the apicalportion of the right maxillary second deciduous molar (Fig 4,A). The tumor embedded the crown and root of the rightmaxillary second premolar and was attached to the first pre-molar and the first and second permanent molars (Fig 4, B). Ahistologic examination of the decalcified surgical specimenrevealed a well-circumscribed tumor involving the adjacentteeth. The tumor was composed of cementum-like tumortissue with irregular lacunae, entrapped cells, and numerousreversal lines and was fused to the cementum on the rootsurface (Fig 5, A). At the periphery of the mass, tumor tissuewas arranged in radiating trabeculae; the tumor was sur-rounded by a fibrous capsule (Fig 5, B). The roots of the rightmaxillary second deciduous molar, second premolar, and firstpermanent molar had been resorbed, and the tumor had ap-parently invaded the palatal root canals of the second decid-uous molar and first permanent molar (Fig 5, C). The pulpaltissues were vital, and no inflammatory change was found.

DISCUSSIONBenign cementoblastoma is a neoplasm of the

jaws2-4 most commonly found in the second and thirddecades of life.8 Some studies have reported that thistumors arises slightly more frequently in males2,9,10 orin females,8 whereas others have found no differencebetween the sexes.11,12 Virtually all benign cemento-blastomas occur in the premolar-molar region, morecommonly in the mandible than the maxilla.2-4,8,11 Inthis study, the tumor occurred in the right maxillary

premolar-molar region of a 12-year-old boy. Benigncementoblastoma is most often associated with a singleerupted permanent tooth.2-4,8 In our patient, the tumorwas located in the apical portion of the right maxillarysecond deciduous molar and involved the first andsecond premolars, in addition to the first and secondpermanent molars, suggesting that the lesion had orig-inated in the second deciduous molar. Most reportedcases of benign cementoblastoma of deciduous toothorigin were associated with mandibular second decid-uous molars (Table I).11,13-18 The mandible is the morecommon site of benign cementoblastoma, yet benigncementoblastomas involving multiple teeth more com-monly occur in the maxilla (Table II).8,11,13,19-25 Suchcementoblastomas have been reported to reflect thehigh growth potential of these tumors; however, norecurrence has been described.

Benign cementoblastoma is histopathologically char-acterized by the formation of sheets of cementum-liketissue containing many reversal lines, irregular lacunae,and cellular fibrovascular stromata. The periphery ofthe mass or the more-active growth areas are oftenunmineralized.2-4 This tumor may sometimes resembleosteoblastoma, osteoid osteoma, or atypical osteosar-coma, which are not distinctively related to tooth roots,and may be difficult to distinguish from these tu-mors.2,21,26-28 Our patient had characteristic hard tissueformation close to the root surface cementum and wasdiagnosed as having benign cementoblastoma. Despitehistologic evidence of vital, noninflamed pulp tissue,the associated tooth may be unresponsive to vitalitytests with an electrical pulp tester.3,30 This paradox has

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

Ohki et al 55ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGYVolume 97, Number 1

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

was weakly responsive to vitality testing, but its pulptissue was histopathologically vital and noninflamed.

Because benign cementoblastoma has unlimitedgrowth potential, the usual treatment is complete sur-

Fig 5. Histopathologic findings. A, Cementum-like tumor tissue with irregular lacunae, entrapped cells, and numerous reversallines fused to the tooth root (hematoxylin-eosin, original magnification �50). B, Tumor tissue is arranged in radiating trabeculaeat the periphery and surrounded by a fibrous capsule (hematoxylin-eosin, original magnification �30). C, Tumor tissue invadesthe palatal root canal of the first permanent molar. The pulpal tissue is vital and noninflamed (hematoxylin-eosin, originalmagnification �10).

Table I. Reported cases of cementoblastoma associated with deciduous teeth

Author Year Age (y)/sex Location Size (cm) Pain Recurrence

Chaput and Marc13 1965 10/F Right mandibular first premolarand second deciduous molar

2.1 � ND

Vilasco et al14 1969 8/F Right mandibular seconddeciduous molar

ND � ND

Zachariades et al11 1985 7/F Right mandibular first and seconddeciduous molars, and firstpermanent molar

3.0 � �

Herzog15 1987 7/F Left mandibular first and seconddeciduous molars

1.4 � ND

Papageorge et al16 1987 6/M Mandibular deciduous centralincisors

4.5 � �

Cannell17 1991 8/F Right mandibular seconddeciduous molar

ND � ND

Schafer et al18 2001 8/F Right mandibular seconddeciduous molar

1.5 � �

Present case 2003 12/M Right maxillary second deciduousmolar, first and secondpremolars, and first and secondpermanent molars

3.3 � �

ND, No data.

56 Ohki et al ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGYJanuary 2004

gical excision with extraction of the associated teeth,even though the pulp may be vital.10,20,21,30-33 Recur-rence does not occur if the mass is totally removed;however, incomplete excision is usually followed byrecurrence.3,20,34-36 Because the present tumor involvedmultiple maxillary teeth, it was extirpated with the rightmaxillary second deciduous molar, the first and secondpremolars, and the first and second permanent molars.There has been no recurrence more than 18 monthsafter the surgical procedure.

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togenic tumours. 2nd ed. Berlin: Springer-Verlag; 1992. p. 23-4.3. Verbin RS, Appel BN. Odontogenic Tumors. In: Barnes L,

editor. Surgical pathology of the head and neck. 2nd ed. NewYork: Marcel Dekker, Inc.; 2001. p. 1603-6.

4. Sciubba JJ, Fantasia JE, Kahn LB. Atlas of tumor pathology. 3rd

series; fasc. 29. Tumors and cysts of the jaws. Washington (DC):Armed Forces Institute of Pathology; 2001. p. 109-10.

5. Mosqueda-Taylor A, Ledesma-Montes C, Caballero-Sandoval S,Portilla-Robertson J, Ruiz-Godoy Rivera LM, Meneses-GarciaA. Odontogenic tumors in Mexico: a collaborative retrospectivestudy of 349 cases. Oral Surg Oral Med Oral Pathol Oral RadiolEndod 1997;84:672-5.

6. Lu Y, Xuan M, Takata T, Wang C, He Z, Zhou Z, et al.Odontogenic tumors. A demographic study of 759 cases in aChinese population. Oral Surg Oral Med Oral Pathol Oral RadiolEndod 1998;86:707-14.

7. Ochsenius G, Ortega A, Godoy L, Penafiel C, Escobar E. Odon-togenic tumors in Chile: a study of 362 cases. J Oral Pathol Med2002;31:415-20.

8. Ulmansky M, Hjørting-Hansen E, Praetorius F, Haque MF. Be-nign cementoblastoma. A review and five new cases. Oral SurgOral Med Oral Pathol 1994;77:48-55.

9. Farman AG, Kohler WW, Nortje CJ, Van Wyk CW. Cemento-blastoma: report of case. J Oral Surg 1979;37:198-203.

10. Vindenes H, Nilsen R, Gilhuus-Moe O. Benign cementoblas-toma. Int J Oral Surg 1979;8:318-24.

11. Zachariades N, Skordalaki A, Papanicolaou S, Androulakakis E,

Table II. Reported cases of cementoblastoma involving multiple teeth

Author Year Age (y)/sex Location Size (cm) Pain Recurrence

Wertheimer et al19 1961 14/M Right maxillary first andsecond permanent molars

2.5 � ND

Chaput and Marc13 1965 10/F Right mandibular firstpremolar and seconddeciduous molar

2.1 � ND

Abrams et al20 1974 17/M Right maxillary first andsecond permanent molars

ND � �

Corio et al21 1976 19/M Left maxillary secondpremolar, and first,second, and thirdpermanent molars

4.5 � ND

Brocheriou et al22 1979 18/F Left maxillary first andsecond permanent molars

3.0 � ND

Zachariades et al11 1985 7/F Right mandibular first andsecond deciduous molars,and first permanent molar

3.0 � �

Herzog15 1987 7/F Left mandibular first andsecond deciduous molars

1.4 � ND

Garlick et al23 1990 19/M Right maxillary first, secondand impacted thirdpermanent molars

3.5 � �

Slootweg24 1992 22/M Right maxillary first andsecond permanent molars

ND ND ND

Jelic et al25 1993 18/M Right mandibular central andlateral incisors, canine,first and second premolars,and second permanentmolar

5.0 � ND

Ulmansky et al8 1994 20/F Right mandibular firstpremolar, and first andsecond permanent molars

3.5 � ND

Present case 2003 12/M Right maxillary seconddeciduous molar, first andsecond premolars, and firstand second permanentmolars

3.3 � �

ND, No data.

Ohki et al 57ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGYVolume 97, Number 1

Bournias M. Cementoblastoma: review of the literature andreport of a case in a 7-year-old girl. Br J Oral Maxillofac Surg1985;23:456-61.

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Reprint requests:

Kousuke Ohki, DDSDivision of Maxillofacial SurgeryDepartment of Oral Medicine and SurgeryTohoku University Graduate School of Dentistry4-1 Seiryo-machi, Aoba-ku, Sendai 980-8575, Japank-ohki@mail.tains.tohoku.ac.jp

58 Ohki et al ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGYJanuary 2004