CLINICAL REVIEW

Mark K. Wax, Section Editor

OSTEOBLASTOMA OF THE MANDIBLE:

CLINICOPATHOLOGIC STUDY OF FOUR CASES
AND LITERATURE REVIEW
Saverio Capodiferro, DDS,1 Eugenio Maiorano, MD, MS,2 Carmela Giardina, MD,2
Maria Grazia Lacaita, MD, DMD,1 Lorenzo Lo Muzio, MD, DMD, PhD,3 Gianfranco Favia, MD, DMD1
1
Department of Dental Sciences and Surgery, University of Bari, Bari, Italy
2
Department of Pathologic Anatomy and Genetics, University of Bari, Piazza Giulio Cesare,
11, 70124 Bari, Italy. E-mail: [email protected]
3
Institute of Dental Sciences, Università Politecnica delle Marche, Ancona, Italy

Accepted 10 January 2005

Published online 2 May 2005 in Wiley InterScience (www.interscience.wiley.com). DOI: 10.1002/hed.20192

logic features and usually manifest an indolent clinical course.

Abstract: Background. Osteoblastoma is a benign bone tu-
A 2005 Wiley Periodicals, Inc. Head Neck 27: 616 – 621, 2005
mor accounting for 1% of all bone tumors; it commonly involves
the spine and the sacrum of young individuals, with less than 5% Keywords: osteoblastoma; bone tumors; osteoid osteoma; head
being localized to the posterior mandible. In view of its rarity in and neck; mandible
the maxilla and mandible, osteoblastoma is rarely diagnosed as
such in the absence of interdisciplinary cooperation.
Methods. A retrospective study of four benign osteoblasto-
mas was performed based on a review of the clinical, radio- Osteoblastoma is a rare benign bone tumor first
graphic, and histopathologic features of all cases. described by Lichtestein1 and Jaffe2 as a distinct
Results. The tumors involved the posterior mandible of neoplasm in 1956. This disease accounts for less
young patients (age range, 10 – 21 years; two male and two
than 1% of all bone tumors and most commonly
female patients) and appeared as painful bone expansions.
Radiologically, they were poorly defined, radiolucent/radi- involves the spine and sacrum of young individ-
opaque lesions containing calcifications and not showing uals.3 – 7 Less than 10% of osteoblastomas are
sclerotic borders or periosteal reactions. Histologically, they localized to the skull, and nearly half of these
were composed of osteoid and woven bone surrounded by cases affect the mandible, especially the posterior
plump osteoblast-like cells with interposed fibroblasts, inflam-
segments.8 The first well-documented case of
matory cells, and red blood cells. All patients were disease free
after prolonged follow-up. osteoblastoma of the jaw bones was described by
Conclusions. Osteoblastomas may be distinguished from Borello and Sedano in 1967,9 and 69 additional
other bone tumors, fibro-osseous lesions, and odontogenic neo- cases have since been recorded.6 Clinically,
plasms on the basis of integrated clinical, radiologic, and histo- osteoblastomas present mainly with pain, swell-
ing, and expansion of the bone cortex1,4 – 6 Radio-
graphic features are variable, usually showing a
Correspondence to: E. Maiorano combination of radiolucent and radiopaque pat-
B 2005 Wiley Periodicals, Inc. terns, depending on the degree of calcification,

616 Osteoblastoma of the Mandible HEAD & NECK July 2005

but without perilesional sclerotic border or per- buffered formalin and then decalcified in Osteo-
pendicular periosteal reactions. Histologically, dec (Bio Optica, Milan, Italy).
osteoblastoma is a bone-forming tumor charac-
terized by osteoid and woven bone deposition and RESULTS
abundant osteoblasts that are frequently in close The salient clinicopathologic features of the cases
association with newly formed bone. Occasionally, are summarized in Table 1.
osteoblastomas may appear richly cellular, con- Overall, all cases occurred in adolescents or
tain an abundant osteoclast-like component, and young adults, with a 1:1 male/female ratio. Three
show plump osteoblasts that may evoke a diag- cases occurred in the left hemimandible, and one
nosis of osteosarcoma, thus leading to unneces- case occurred in the right hemimandible, with the
sary overtreatment. same proportion of cases being located distally to
In view of the purported benign nature of the first molar or in the premolar-molar area.
this tumor, conservative surgical excision is the Most patients complained initially of pain and
treatment of choice, because recurrence is a rare swelling of variable duration (2 weeks– 2 years),
event (13.6%) and usually attributable to incom- with one case occurring simultaneously with the
plete excision.6 eruption of the third molar.
The current retrospective study was aimed at At clinical examination, in all four cases a firm
reporting the salient clinicopathologic features of mandibular mass was detected that was not
four cases of osteoblastoma involving the man- associated with signs of local inflammation,
dible, three in the retromolar area (treated by pericoronitis, paresthesia, or mucosal ulceration
surgical excision) and one occurring in the man- (Figure 1). Two patients underwent extraction of
dibular body (treated by en-bloc resection and teeth by a general dentist after a diagnosis of
reconstruction by free tissue transfer technique), pericoronitis or odontogenic abscess.
all displaying a benign clinical course without Radiographically, all lesions were poorly de-
recurrence at 3 to 25 years’ follow-up. fined and displayed mixed radiolucent/radiopaque
features (Figure 2) with focal to well-evident in-
tralesional calcifications, which were particularly
MATERIALS AND METHODS evident on CT scans (Figure 3). No periosteal
All the clinical charts of the patients admitted to reactions, sclerotic borders, or peripheral spec-
the Department of Dental Sciences and Surgery of ulation, and no root resorption of adjacent teeth
the University of Bari during the period 1970 to were evident.
2002 who underwent surgical procedures for After the provisional clinical differential diag-
osseous lesions were retrieved and reviewed after nosis of odontogenic neoplasms (eg, odontogenic
approval of this study by the Internal Ethical myxoma, fibro-osseous bone lesion, or central ce-
Committee. These cases with histologic diagnosis mento-ossifying fibroma), three patients under-
of benign or malignant bone tumors were sub- went local excision or curettage under general
jected to histopathologic review of the original anesthesia. The remaining patient underwent an
preparations performed by two pathologists (EM incisional biopsy, which was sent for intraoper-
and CG), which subsequently resulted in the ative examination and resulted in a provisional
identification of the four cases of osteoblastoma diagnosis of low-grade osteosarcoma. This diag-
described herein. nosis was probably based on the presence of
The original paraffin blocks of each case were hyperchromatic osteoblast-like cells with large
cut and stained with hematoxylin-eosin. The nuclei interpreted as malignant cells on cryostatic
tissues had been previously fixed in 10% neutral sections. Consequently, an en-bloc resection of the

Table 1. Clinicopathological features of the study population (including age at first clinical presentation, site referred to adjacent tooth/
teeth, duration of symptoms before medical assessment, and size as radiographically assessed).

Case no. Age, y Sex Disease site Duration of symptoms Clinical diagnosis Size, cm
1 16 F 47 2 mo Odontogenic neoplasm 3
2 10 M 37 2 wk Odontogenic abscess 2.5
3 21 F 38 2 y Pericoronitis 2
4 20 M 36 – 37 3 mo Odontogenic myxoma 3

Osteoblastoma of the Mandible HEAD & NECK July 2005 617

FIGURE 1. Clinical image illustrates a smooth expansion of the
posterior mandibular area covered by intact epithelium.

affected segment of the mandible was performed,

followed by free tissue transfer reconstruction. FIGURE 3. CT scan of a mandibular osteoblastoma causing
deformity of the bone architecture and containing large amounts
Macroscopically, the tumors were reddish of calcified material. The lesion is not associated with sclerotic
to brown in color and of friable consistency. borders or periosteal alteration.
Those tumors that were enucleated intact were
irregularly round to oval and appeared rather
well demarcated.
Histologically, all tumors contained abundant tory cells, red blood cells, and an abundant
mineralized material both as osteoid and as capillary component (Figure 6). Cells displaying
woven bone trabeculae (Figure 4) that were overt dysplastic features or abnormal mitotic
distinctly separated from the adjacent trabecular activity were not detected, and there was no car-
bone. The newly formed trabeculae were of tilaginous differentiation.
different size and sometimes closely packed upon
each other. At higher-power resolution, the tu- DISCUSSION
mors were composed of a double cell population:
plump osteoblast-like cells, with eosinophilic Osteoblastoma is a rare bone-forming tumor that
cytoplasm and large nuclei, that were frequently very rarely involves the maxilla and mandible,
juxtaposed to the newly formed bone trabec- particularly the posterior mandible. This diag-
ulae, and less abundantly intermingled spindle nostic challenge may cause relevant problems
cells with poorly visible cytoplasms and rounded with the differential diagnosis in view of the
to oval nuclei (Figure 5). Frequently, the osteo- tumor’s rarity, ambiguous clinicoradiologic pre-
blast-like cells were arranged concentrically to sentation, and histopathologic features, which
osteoid accumulations (osteoblastic rimming), sometimes resemble osteosarcoma.
whereas the fibroblast-like component was The first osteoblastoma of the jawbones was
loosely packed and intermingled with inflamma- described relatively recently.9 To date, with single
case reports and other large series, including our
cases, a total of 69 cases of osteoblastomas occur-
ring in the mandible have been reported.5,6,10 – 15
Overall, the age range in the reported cases
was 5 to 69 years, with a slight predominance in
male patients (58%) and a definite prevalence in
the mandible (74%) versus the maxilla. Sponta-
neous pain was identified in 54 cases (78%),
whereas bone swelling was observed in 62 cases
(90%). The roots of the adjacent teeth were in-
volved in 20 cases (29%), but root resorption was
evident in only four. Cytologic atypia (including
hyperchromasia and pleomorphism) were ob-
FIGURE 2. Panoramic radiograph shows a radiolucent lesion
of the left mandibular molar area (arrows), in close association served in four cases (7%).6
with an unerupted tooth, with regular contours and containing Conceptually, osteoblastoma shares similar
fine calcifications. clinicopathologic features with cementoblastoma,

618 Osteoblastoma of the Mandible HEAD & NECK July 2005

FIGURE 4. Photomicrograph of histopathologic section of os- FIGURE 6. Photomicrograph demonstrates that the neoplastic
teoblastoma. The tumor is composed of large and irregular osteoblast-like cells frequently show bizarre and hyperchromatic
clusters of calcified material with interposed loose collagenous nuclei without atypical features, nucleoli, or abnormal mitoses. In
fascicles (hematoxylin-eosin stain, original magnification 50). addition, inflammatory cells and red blood cells are frequently
dispersed in the stroma, which also contains several blood
vessels (hematoxylin-eosin stain, original magnification 400).

with the exception of strict relationships of the

latter with roots of a tooth. Nevertheless, because rarity and nonspecific presentation. At clinical
of the difficulty of distinguishing true cementum inspection, a bone expansion of hard consistency
from bone in heavily mineralized lesions, and is detectable that manifests benign features
because of the overlapping clinical management because of slow and progressive growth and
of both lesions, we prefer to consider these as absence of mucosal ulceration, paresthesia, or
variants of a single entity. abnormal tooth mobility.
Diagnosing osteoblastoma at first clinical Radiologic assessment is commonly performed
presentation is usually difficult because of its and highlights mixed radiolucent/radiopaque le-
sions with more or less defined borders, and these
usually lack sclerotic borders, periosteal reac-
tions, or perpendicular bone speculations. In the
presence of such findings, osteosarcoma should be
considered in the differential diagnosis.
On the basis of the preceding characteris-
tics, the differential diagnosis may include the fol-
lowing: (1) fibro-osseous lesions (such as ossifying
fibroma, fibrous dysplasia, periapical cemento-
osseous dysplasia, and central giant cell granulo-
ma), (2) bone tumors (including osteoid osteoma),
and (3) odontogenic tumors (such as ameloblasto-
ma and calcifying epithelial odontogenic tumor).4
Ossifying fibroma and fibrous dysplasia of
bone may share many similarities with osteoblas-
toma but usually are less mineralized lesions,
revealing fine calcifications rather than large
FIGURE 5. Photomicrograph at higher magnification shows the clusters of mineralized material. In addition, fi-
tumor is composed of large osteoblast-like cells, showing eosino- brous dysplasia is less circumscribed radiologi-
philic cytoplasm and plump nuclei, frequently surrounding clus- cally than osteoblastoma and may be multifocal, a
ters of osteoid substance. The intervening collagenous stroma
feature that is exceedingly rare for osteoblastoma.
contains osteoclast-like multinucleated cells and loosely aggre-
gated fibroblast-like cells. There is no evidence of entrapped or Periapical cemento-osseous dysplasia is a
infiltrated residual bone trabeculae or of cartilaginous differ- painless fibro-osseous lesion that involves the
entiation (hematoxylin-eosin stain, original magnification 200). apical root region of a tooth and may share simi-

Osteoblastoma of the Mandible HEAD & NECK July 2005 619

lar radiographic and histopathologic features and red blood cells. More important, the absence of
with osteoblastoma. Nevertheless, the former is lace-like osteoid and cartilaginous differentiation
a self-limited process of small size (<1 cm), with is classic in osteoblastoma.6,8
the exception of the so-called florid periapical Aside from osteosarcoma, the histologic differ-
cemento-osseous dysplasia. This is a rare condi- ential diagnosis of osteoblastoma compared with
tion that involves several dental elements, a fea- fibro-osseous lesions and odontogenic tumors
ture that is exceptionally rare for osteoblastoma usually does not represent a problem. Although
of the jaws and has never been reported in the osteoid osteoma shares the same histologic char-
English language literature. Central giant cell acteristics as osteoblastoma, it is a lesion limited
granuloma at its early stage of development can in size to 2 cm or less. The diagnosis is mainly
exhibit the same clinicoradiologic features that based on the characteristic architectural distri-
osteoblastoma exhibits. Central giant cell gran- bution of the mineralized and nonmineralized
uloma has a propensity for crossing the midline, tissues in osteoid osteoma, the abundant fibro-
especially in the maxilla, with divergence of tooth blastic stroma and peculiar morphologic features
roots and resorption of lamina dura and roots, and of the bony spicule in ossifying fibroma and fibrous
usually appears multilocular on radiographs, with dysplasia, the rich giant cell component in giant
wispy internal septa at advanced stages.16,17 cell granuloma, and the definite presence of an
Osteoid osteoma is extremely rare in the jaws, epithelial component in ameloblastoma and calci-
commonly shows a well-defined sclerotic host bone fying epithelial odontogenic tumor.
reaction with a radiolucent center (nidus) on The preceding distinctions are clinically rele-
radiographs, and, conventionally, should be small- vant in consideration of the attenuated biologic
er than 2 cm in larger dimension.13 Ameloblasto- behavior of osteoblastoma as demonstrated by the
mas, especially the multicystic and solid types, current short series of cases; in all of these cases,
usually show scalloped borders with tiny, irregular the patients are disease free even after prolonged
intralesional calcifications, whereas the calcifying clinical observation. Nevertheless, osteoblastoma
epithelial odontogenic tumor may be radiologically may show a low tendency to recur (13.6%)6 and,
similar to osteoblastoma but much more fre- very uncommonly, aggressive behavior or sarcoma-
quently arises in association with impacted teeth. tous changes.18 – 20
Histologic examination is paramount in con- It should be emphasized that some osteo-
firming a definitive diagnosis and excluding blastoma variants, namely the so-called toxic
osteosarcoma. The examination of small tissue osteoblastoma and the aggressive osteoblastoma,
fragments, as for intraoperative examination, can may display unusual features, such as fever, an-
be ominous occasionally and lead to an incorrect orexia, hypergammaglobulinemia, and cachexia
diagnosis, as occurred in one of the cases in this in the former and epithelioid osteoblasts, atypi-
series. Furthermore, frozen section analysis is cal mitoses, and lace-like osteoid in the latter.
limited to soft tissues, because bone fragments, Both of these tumor variants may possibly show
needing decalcification, cannot be cut during this an aggressive clinical course.8 The debate is still
procedure. Nevertheless, on permanent sections, ongoing as to whether these variants repre-
and even after decalcification, the diagnosis of sent true osteoblastomas or poorly character-
osteoblastoma versus osteosarcoma may still be ized osteosarcomas, and it may be that some
difficult to accomplish. This is mainly due to the previously reported cases of osteoblastoma af-
presence of plump or even bizarre-appearing fecting the jawbones and manifesting sarcoma-
osteoblast-like cells in osteoblastoma, often bor- tous changes, multiple recurrences, or distant
dering osteoid substance, mimicking the atypical metastases could have been categorized into
cells that characterize osteosarcoma. In such these variants.
instances, the diagnosis is greatly facilitated by
the accurate interpretation of the histologic fea-
tures in view of the radiologic findings that do not
REFERENCES
disclose the aggressive characteristics of osteosar-
1. Lichtenstein L. Benign osteoblastoma: a category of
coma. In addition, osteoblastoma never shows osteoid and bone-forming tumors other than classical
peripheral permeation of the neoplastic tissue into osteoid osteoma, which may be mistaken for giant-cell
the adjacent lamellar bone and has a more loosely tumor or osteogenic sarcoma. Cancer 1956;9:1044 – 1052.
2. Jaffe HL. Benign osteoblastoma. Bull Hosp Joint Dis
arranged supportive stroma rich in capillary blood 1956;17:141 – 151.
vessels, which also contains inflammatory cells 3. Lichtestein L, Sawyer WR. Benign osteoblastoma: further

620 Osteoblastoma of the Mandible HEAD & NECK July 2005

 observations and report of twenty additional cases. J Bone 13. Ribera MJ. Osteoblastoma in the anterior maxilla mim-
Joint Surg Am 1964;46:755 – 765. icking periapical pathosis of odontogenic origin. J Endo-
4. Regezi JA, Sciubba JJ. Oral pathology: clinical-pathologic dont 1996;22:142 – 146.
correlations. 2nd ed. Philadelphia: WB Saunders; 1993. 14. Matsuzaka K, Shimono M, Uchiyama T, Noma H. Le-
p 404 – 406. sions related to the formation of bone, cartilage or ce-
5. Ahmed MS, Nwoku AL. Benign osteoblastoma of the mentum arising in the oral area: a statistical study and
mandibular ramus: review of the literature and report of a review of the literature. Bull Tokyo Dent Coll 2002;43:
case. J Oral Maxillofac Surg 2000;58:1310 – 1317. 173 – 180.
6. Gordon SC, MacIntosh RB, Wesley RKO. A review of 15. Ozturk M, Ozec I, Aker H, Muslehiddinoglu A. Osteoblas-
osteoblastoma and case report of metachronous osteoblas- toma of the mandible with root resorption: a case report.
toma and unicystic ameloblastoma. Oral Surg Oral Med Quintessence Int 2003;34:135 – 138.
Oral Pathol Oral Radiol Endod 2001;91:570 – 575. 16. Goaz PW, White SC. Oral radiology: principles and inter-
7. Haug RH, Huaer C, De Camillo AJ, Araneta M. Benign pretation. 3rd ed. St Louis, MO: Mosby – Year Book; 1994.
osteoblastoma of the mandible: report of a case. J Oral p 398 – 676.
Maxillofac Surg 1990;48:743 – 748. 17. Cohen MA, Hertzanu Y. Radiologic features, including
8. Mirra JM, Picci P, Gold RH. Bone tumors. Clinical, those seen with computed tomography, of central giant cell
radiologic and pathologic correlations. Philadelphia: Lea granuloma of the jaws. Oral Surg Oral Med Oral Pathol
& Febiger; 1989. p 389 – 430. 1988;65:255 – 261.
9. Borello ED, Sedano HO. Giant osteoid osteoma of the 18. Ohkubo T, Hernandez JC, Ooya K, Krutchkoff DJ.
maxilla. Oral Surg Oral Med Oral Pathol 1967;23: Aggressive osteoblastoma of the maxilla. Oral Surg Oral
563 – 566. Med Oral Pathol 1989;68:69 – 73.
10. El-Mofty S, Refai H. Benign osteoblastoma of the maxilla. 19. Mitchell ML, Ackerman LV. Metastatic and pseudomalig-
J Oral Maxillofac Surg 1989;47:60 – 64. nant osteoblastoma: report of two unusual cases. Skeletal
11. Strand-Pettinen I, Lukinmaa PL, Holmtrom T, Lindqvist Radiol 1986;15:213 – 218.
C, Hietanen J. Benign osteoblastoma of the mandible. Br 20. Vigneswaran N, Fernandes R, Rodu B, Baughman RA,
J Oral Maxillofac Surg 1990;28:311 – 316. Siegal GP. Aggressive osteoblastoma of the mandible
12. Asada Y, Suzuki I, Suzuki M, Fukushima M. Atypical closely simulating calcifying epithelial odontogenic tumor:
multiple benign osteoblastomas accompanied by simple report of two cases with unusual histopathologic findings.
bone cysts. J Craniomaxillofac Surg 1991;19:166 – 171. Pathol Res Pract 2001;197:569 – 576.

Osteoblastoma of the Mandible HEAD & NECK July 2005 621