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J. max.-fac. Surg. 10 (1982) 182-186 © 1982 Georg Thieme Verlag Stuttgart . New York
Benign Cementoblastoma Case Report and Literature Review
Summary A new case of benign cementoblastoma with a three year observation period is added to those cases previously described in the literature. Clinical, radiographic and histological characteristics, summarized from the relevant literature, are discussed.
Miro Makek, Glenn Lello
Key-Words Institute of Pathology (Directors: Prof. Ch. Hedinger, M.D. and Prof. J. R. Rfitther, M.D.), and the Clinic of Maxillo-Facial Surgery (Director: Prof. H. Obwegeser, M.D., D.M.D.) University Hospital Zurich, Switzerland
Introduction The benign cementoblastoma is a rare odontogenic tumour. Initially described by Norberg (1930) approximately 32 adequately described and verifiable cases have subsequently been added to the literature. We wish to report on another case, which, during the period 1955-1981 proved to be the only one diagnosed in our clinic. This case is presented in detail, having been followed radiographically for a period of 3 years prior to surgery. An attempt is made to define typical characteristics pertaining to this tumour based on our case and those previously published. Case Description In December 1968 a 19-year-old male was referred to the Policlinic of the Dental Institute, Zurich University; complaining of an occlusal interference in the region of the first permanent right mandibular molar, and a dull ache in this area. Radiography revealed a well-defined, sharply demarcated, mottled area extending between the second molar distally and first premolar mesially. Superiorly the lesion reached the alveolar crest between the first molar and
Fig. 1 a SharpLy demarcated lesion involving the mandibular right first molar (18/12/1968).
Benign cementoblastoma
second premolar, and encroached inferiorly upon the lower border of the mandible, displacing the mandibular canal (Fig. l a ) . The roots of the second premolar and second molar were displaced, and both mesial and distal first molar roots were resorbed by approximately one half of their original length. The turnout had expanded predominantly the lingual cortical plate, and had thinned the buccal cortical plate (Fig. I b). The periapical film shows the mottled appearance, and obvious resorption of mesial and distal first molar roots. In January 1969 a biopsy of the lesion, taken after a buccal decortication, revealed reddish-blue soft tissue with hard inclusions, which clinically had had no clear demarcation from the surrounding bone. The histopathological diagnosis was recorded as an osteoblastoma. In May of the same year the first molar was extracted owing to increasing pain. Regular follow-up over the next 21/2 years revealed no untoward signs or symptoms initially, with no apparent alteration in tumour size; until in May 1970, one year after extraction of the first molar, a gradual but obvious increase in tumour size was observed (Fig. 2 a and 2b). At this stage the patient was referred to the Maxillo-Facial Department of the Zurich University Hospi-
Fig. 1 b Occlusal plane radiograph demonstrating the thinned buccal cortical plate and predominantly lingual expansion of the tumour (18/12/1968).
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Fig. 2 a A gradual increase in turnout size is noted one year after extraction of second molar (13/5/70).
Fig. 2 b
Increased tumour expansion bucco-lingually (13/5/70).
Fig. 3 a Involvement of the mesial root of the second molar, the root of the second premolar and the mental foramen. Greatly thinned lower border of mandible (20/10/71).
Fig. 3 b
Occlusal view. Showing increased tumour size (20/10/71).
tal Surgery Department, with a diagnosis of sclerosing osteoblastoma. A mild facial asymmetry, intact facial sensibility, non-vital second molar and second premolar teeth (which were both previously vital in December 1968) and an expanded buccal and lingual cortical plate with pressure sensitivity in the region of the extracted first molar was noted. Although in the initial radiographs the mesial root of the second molar, the root of the second premolar and the mental foramen appeared to be practically unaffected,
radiographs taken on the 20 ta October, 1971 (Fig. 3 a and 3 b) indicate at this stage a definite involvement, with an apparent thinning of the lower border of the mandible having occurred as well. In November 1971 under nasal intubation anaesthesia, a buccal and lingual free gingival margin incision from the right third molar and extending anteriorly to the right first incisor served to expose the involved area adequately. Both mandibular right premolars and second molar were extracted, and the tumour was enucleated. The involved
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Fig. 4 One year after autologous lilac crest bone transplant reconstruction of the defect. Perfect healing is evident (12/2/1979).
M. Makek, G. Lello
Fig. 5
Tumour tissue eroding dentine (HE x 120).
portion of the inferior alveolar nerve was removed together with the turnout, as were the soft tissues in contact with the tumour through the buccal and lingual cortical plate perforations. The resultant cavity was packed with Vioform gauze and the intraoral wound closed. Intermaxillary fixation was applied using interdental wire loop splints. The splinting was removed 6 weeks post-operatively, and in 1978, 61/2 years later, an autologous iliac crest bone transplant was used to reconstruct the deficient right mandible (Fig. 4). An absence of sensibility in the inferior alveolar and mental nerve area remains, otherwise the patient is symptomless.
Histology
Fig. 6 Collections of cementoblasts and acellular tumour foci (HE x 275)
The tumour is sharply demarcated from the adjacent bone, although an obvious capsule is not present. The tumour tissue consists of a tangled, thick meshwork of trabeculae with a varying degree of mineralization. Centrally, the tissue is homogeneous, and an acellular, amorphous connection to the root is evident. A mosaic pattern of abundant basophilic reversal lines is apparent. Erosion of dentine is present (Fig. 5). The periphery of the tumour is composed of a loose vascular fibrous tissue, rich in cells. Active cementoblasts; large oval to round cells containing much cytoplasm and a round nucleus, lie in aggregates, producing a pale-staining osteoid-like substance, which is separated from the "older" tumour (Fig. 6). The mature zones contain densely calcified sheets of cemen-
Benign Cementoblastoma Table 1
J. max.-fac. Surg. 10 (1982)
Benign cementoblastoma. Cases reviewed
Case
Author
Year
Sex
Age
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 !8 19 20 21 22 23 24 25 26 27 26 29 30 31 32 33 34
Norberg Tt~oma Sonesson Agazzi and Belloni Wertheimer et al. Klineetal. Chaputand Marc Chaput et al. Vialasco et al. Curran and Collins Eversole et al. Eversole et al. Cherricketal. Cherrick et al. Astacio and Mendez Astacio and Mendez Abrams et al. Abrams et al. Abrams et al. Abrams et al. Abrams et al. Abrams et aL Abrams et al. Wiggins and Karian Anneroth et al. Langdon Corio et al. Larsson et al. Takagi Towns et al. Towns et al, Vindenes et al. Farman et al. Present Case
1930 1937 1949 1953 1961 1961 1965 1969 1969 1973 1973 1973 1974 1974 1974 1974 1974 1974 1974 1974 1974 1974 1974 1975 1975 1976 1976 1978 1978 1979 1979 1979 1979
F F M M M M F F F F F M M F F M M M M F M F F M M F M F F F F M M M
30 40 21 35 14 12 10 12 8 30 17 14 15 26 16 36 15 16 17 18 20 21 22 26 23 17 19 15 11 14 14 19 19 19
turn-like substance with haematoxyphilic reversal lines reminiscent of Paget's bone disease. Diagnosis: Benign Cementoblastoma. Discussion
Cementomas were classified by Pindborg and Kramer in 1971 for the W.H.O. into 4 distinct subgroups; cementifying fibroma, periapical cemental dysplasia, gigantiform cementoma, and the benign cementoblastoma which is characterized histologically by "the formation of sheets of cementum-like tissue, which may contain a very large number of reversal lines and be unmineralized at the periphery of the mass, or in the more active growth areas". Based on the W.H.O. criteria, cases described by Thoma (1937) Pindborg (1951), Gorlin et al. (1961) and Freidel and Gonnon (1962) are not considered to be true examples of benign cementoblastoma. Thoma's case and the 63-year-old female case of Pindborg are considered to be examples of periapical cemental dysplasia, while Pindborg's second case is most likely a cementifying fibroma. Following personal communication with Gorlin in 1980, the certainty of the diagnosis of his published case is considered in the light of present knowledge, to be doubtful. In Friedel and Gonnon's case radiological and histological
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evidence is absent, and the age, localization, symptomatology and the nature of the lesion is not typical. Reviewing the relevant literature and considering the above described case, the following features and characteristics pertaining to the benign cementoblastoma would appear to be pertinent to reaching a correct diagnosis. 1. Equal sex distribution: Contrary to other reports (Chefrick et al., 1974; Corio et al., 1976) which specified a definite predilection for males, the literature contains 17 male and 17 female cases. 2. Age distribution: Most cases are diagnosed in patients younger than 20 years, helping to differentiate the lesion from periapical cemental dysplasia which occurs more commonly in females in the 4 th and 5 th decades. 3. Symptoms: In contradistinction to the other 3 cementoma subgroups, the benign cementoma is almost always accompanied by either swelling (10 cases), low grade intermittent pain (2 cases), or both (18 cases). Only 4 of the 34 cases were totally symptomless. 4. Localization: Typically in the molar region. 85 % of tumours were located within the mandible (15 % in maxilia) and 60 % were associated with the first mandibular molar. 5. Radiography: The specific radiographic features are excellent diagnostic aids. 32 described cases were typical in that they tended towards being round, and exhibited a mottled to dense central radio-opacity outlined by a narrow, well delineated peripheral radiolucency. A sharply defined border between tumour and surrounding bone was evident. A direct connection between the lesion and the involved tooth or teeth, which remained vital in the absence of concomitant pathology, was usually clearly seen (28 cases) and in 16 cases resorption of the related roots was described. Adjacent teeth may be displaced. Loss of buccal, labial or inferior alveolar canal cortical plates frequently occurred, and expansion of buccal and lingual cortical plates was common although facial asymmetry was infrequently noted. Sizes varied between 1 cm. and 8 cm. in diameter, and a growth rate of approximately 0.5 cm. in diam. per year is claimed (Vindenes et al., 1979) thus confirming the benign nature of the lesion, despite Langdon's (1976) reference to the possible aggressive nature of the tumour. The benign cementoblastoma is said by Cherrick (1974) to have an unlimited growth potential. Histological diagnosis can be difficult depending upon the extent and location of the biopsy. Fibrous capsules were mentioned in 17 cases, while 6 authors reported on the absence of a capsule. The differential diagnosis would include periapical cemental dysplasia, cementifying fibroma, gigantiform cementoma; and the histology of the active areas of the lesion needs to be differentiated from benign osteoblastoma, osteoid osteoma and osteosarcoma. No reports of malignant alteration exist in connection with the benign cementoblastoma. Therapy comprises extraction of the involved tooth or teeth and enucleation of the tumour. The prognosis of benign cementoblastomas, treated as recommended, is excellent, with no recurrences having been reported. References
Abrams, A. M., J. W. Kirby, R. J. Melrose:Cementoblastoma, a clinical-pathologic study of seven new cases. Oral Surg. 38 (1974) 394-404
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Agazzi, C., L. Belloni: Gli odontomi duri dei mascalleni; contributo clinico-roentgenologico e anatomomicroscopo con particulare riguardo alle forme ad ampia estensione e alia comparsa familiare. Arch. Ital. Otol. (Suppl. 16) 64 (1953) 3-102 Anneroth, G., G. Isacsson, A. Sigurdsson: Benign Cementoblastoma (true cementoma). Oral Surg. 40 (1) (1975) 141-146 Astacio, J. N., J. E. Mendez: Benign Cementoblastoma (true cementoma). Oral Surg. 38 (1974) 95-99 Chaput, A., A. Marc: Un cas de c4mentome localis6 sur une molaire temporaire. Schweiz. Monatsschr. Zahnheilk. 75 (1965) 48-52 Chaput, A., R. Bataille, M. Minc: Deux cas de c6mentome. Rev. Stomatol. 70 (1969) 325-328 Cherrick, H. M., O. H. King, M. Lucatoro, D. M. Suggs: Benign Cementoblastoma; A clinicopathologic evaluation. Oral. Surg. 34 (1974) 54-63 Corio, R.L., B. E. Crawford, S.J. Schaberg: Benign Cementoblastoma. Oral Surg. 41 (1976) 524-530 Curran, J. B., A. P. Collins: Benign (true) cementoblastoma of the mandible. Oral Surg. 35 (1973) 168-172 Eversole, L. R., W. R. Sabes, V. G. Dauchess: Benign Cementoblastoma. Oral Surg. 36 (1973) 824-830 Farman, A. G., W. W. Koehler, C. J. Nortje, C. W. van Wyk: Cementoblastoma: Report of case. J. O. Surg. Vol. 37 (1979) 198 Freidel, Ch., F. Gonnon: Un cas de c6mentome. Ann. odontostomatol. 19 (1962) 205-209 Gorlin, R. J., A. P. Chaudhry', J. J. Pindborg: Odontogenic Tumors. Cancer 14 (1961) 73-101 Kline, S. N., S. S. Spatz, H. J. Zubrow, M. Fader: Large cementoma of the mandible; Report of a case. Oral Surg. 14 (1961) 1421-1426 Langdon, J. D.: Ten benign cementoblastoma - Just how benign? Brit. J. Oral Surg. 13 (1976) 239-249 Larsson, A., O. Fossberg, S. Sjoegren: Benign cementoblastoma cementum analogue of benign osteoblastoma? J. Oral Surg. 36 (1978) 299-303
M. Makek, G. Lello: Benign Cementoblastoma No1~berg, 0.: Zur Kenntnis der dysontogenetischen Geschwiilste der Kieferknochen. Vjschr. Zahnheilk. 46 (1930) 321-355 Pindborg, J. J.: Om Cementoblastomer (Cementomer). Odont. Tidskr. 59 (1951) 80-87 Pindborg, J. J.: C4mentoblastomes - C6mentomes. Rev. Stomatol. 52 (1951) 745-753 Pindborg, J. J., I. R. H. Kramer: Histological Typing of Odontogenic Tumors, Jaw Cysts and Allied Lesions. WHO Geneva, p. 31, 1971 Sonesson, A.I.: Ett bidrag till cementoblastomets diagnostik och r6ntgenologiska differentialdiagnostik. Tidskr. 57 (1949) 409-414 Thoma, K. H.: Cementoblastoma. Internat. J. Orthodont. 23 (1937) 1127-1137 Takagi, M.: Zementoblastom. Dtsch. Zahn~irztl. Z. 33 (1978) 712 Towns, T. M., R. B. Marks, R. F. Carr: Benign (true) cementoblastoma: Report of cases. J. Oral Surg. 37 (1979) 342-345 Vilasco, J., J. Mazere, J. C. Douesnard, R. Loubiere: Un cas de c6mentoblastome. Rev. Stomatol. 70 (1969) 329-332 Vindenes, H., R. Nilsen, O. Gilhuus-Moe: Benign Cementoblastoma. Int. J. Oral Surg. 8 (1979) 318-324 Wertheimer, F.W., E.J. Driscoll, H . R . Stanley: True (attached) cementoma with root canal involvement. Oral Surg. 14 (1961) 630-634 Wiggins, H.E., B.K. Karian: Cementoblastoma of the maxilla: Report of case. J. Oral Surg. 33 (1975) 302-303
M. Makek, M.D. Institut of Pathology Zurich University Schmelzbergstr. 12 CH-8091 Zurich~Switzerland G. E. Lello, B.D.S., F.D.S., R.C.S., L.R.C.P., M.R.C.S., M.B., B.Ch. MaxilIo-Facial Surgery ClinicZurich University Plattenstrafle 11 CH-8028 Zurich~Switzerland