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Oral Radiology

https://doi.org/10.1007/s11282-019-00370-9

CASE REPORT

Benign osteoblastoma of the palate: a rare clinical presentation


Dhanya Mary Sam1 · Sreeja P. Kumar1 · Beena Varma1 · Anju P. David1 · Rakesh Suresh1

Received: 6 August 2016 / Accepted: 6 December 2018


© Japanese Society for Oral and Maxillofacial Radiology and Springer Nature Singapore Pte Ltd. 2019

Abstract
Osteoblastoma is a rare, benign type of osteoblastic tumor. It constitutes approximately 1% of all primary bone tumors.
Osteoblastoma most commonly affects the long bones; it very rarely affects the jaw bones. Because of its clinical and histo-
logical similarity with other bony tumors, such as osteoid osteoma and fibro-osseous lesions, osteoblastoma is a diagnostic
challenge. Very few cases of osteoblastoma involving the maxillofacial region have been reported to date. We herein describe
a 15-year-old female patient with osteoblastoma that presented as a palatal swelling of 6 months’ duration.

Keywords  Benign · Lesion · Osteoblastoma · Pain · Radiolucent · Radiopaque · Swelling

Introduction was sudden in onset and had not changed in size since its
appearance. 1 week after the appearance of the swelling,
Osteoblastoma is a rare benign osteoblastic tumor of bone. the patient developed pain in the upper left front teeth. The
It accounts for 1% of all bone tumors, and approximately pain was sharp, pricking, localized, and intermittent and
11% of bone osteoblastomas occur in the skull [1]. “This became aggravated during biting. She consulted a dentist
lesion was first reported by Jaffe and Mayer in 1932 as an in her locality, and endodontic treatment was begun for the
osteoblastic osteoid tissue-forming tumor. This tumor has involved teeth. After completion of the treatment, however,
also been called ‘giant osteogenic fibroma’ and ‘giant oste- the swelling did not decrease in size and the patient was
oid osteoma” [2]. Jaffe and Lichenstein termed this lesion as referred to our department. The patient’s medical history
“benign osteoblastoma” in 1956, which was later adopted by was not contributory.
the World Health Organization [2]. Osteoblastoma is most Extraoral examination revealed no significant findings.
prevalent in the second decade of life. Intraoral examination revealed a solitary ovoid swelling of
Most bony lesions pose a diagnostic challenge to the cli- approximately 1.5 × 2.0 cm with well-demarcated borders on
nician because they exhibit very close clinical, radiological, the left side of the anterior region of the hard palate (Fig. 1).
and histological interrelations. We herein report one such The swelling was present on the anterior third of the hard
case of osteoblastoma with a rare clinical presentation and palate, 1 cm away from the region of the upper left cen-
provide a brief review of the literature. tral and lateral incisors and 5.0–5.5 cm posteriorly, in front
of the hard–soft palate junction. The overlying mucosa and
the surrounding areas had a normal appearance. The surface
Case report of the swelling was smooth with no signs of discharge or
ulcerations. The swelling was tender on palpation and firm
A 15-year-old female patient presented to the Department to hard in consistency. Tenderness on percussion was noted
of Oral Medicine and Radiology with a 6-month history of in the associated teeth (central and lateral incisors with slight
a swelling on the front region of the palate. The swelling extrusion of the lateral incisor (Fig. 2). There was no mobil-
ity of the teeth. As a part of the chairside investigation, elec-
tric pulp testing was performed in relation to the upper left
* Dhanya Mary Sam central and lateral incisors by keeping the upper right central
dhanya16888@gmail.com and lateral incisor as the control teeth. Testing revealed that
1
Department of Oral Medicine and Radiology, Amrita School
the upper left central incisor was nonvital.
of Dentistry, AIMS Campus, Ponekkara, Kochi 682041,
India

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Oral Radiology

Fig. 1  Intraoral photograph with mirroring. A well-circumscribed


solitary ovoid swelling (arrows) of approximately 1.5 × 2.0 cm is seen
on the left anterior hard palate with normal overlying mucosa

Fig. 3  Intraoral periapical radiograph. An oval-shaped periapical


mixed radiolucent lesion (arrows) is shown in the middle third region
of the radicular region between the upper left central and lateral inci-
sors with a radiolucent rim. Loss of the lamina dura is evident in the
periapical region of these two teeth

Fig. 2  Intraoral photograph from the anterior view. Extrusion of the


upper left central incisor is demonstrated (arrow)

Considering the patient’s history, clinical examination


findings, and chairside investigation findings, a provisional
diagnosis of a radicular cyst of the upper left central inci-
sor and differential diagnoses of an impacted supernumer-
ary tooth and odontoma were given. Further investigations
included intraoral periapical radiographs, maxillary true
occlusal radiographs, and cone beam computed tomogra- Fig. 4  Occlusal radiograph of the maxilla. Significant root divergence
of the upper left central and lateral incisors have been caused by the
phy (CBCT). lesion
Intraoral periapical radiographs of the upper left cen-
tral and lateral incisors revealed an oval-shaped periapi-
cal mixed radiopaque/radiolucent lesion of approximately a mixed hyperdense/hypodense lesion of 1 × 1 cm in the
1 × 1 cm in the middle third region of the radicular por- inter-radicular region of these two teeth (Fig. 5). The sag-
tion of these two teeth (Fig. 3). The lesion was well cir- ittal section revealed that the lesion was 1 mm away from
cumscribed by a radiolucent rim, and loss of the lamina the apices of the teeth and was causing displacement of the
dura in the periapical regions of these two teeth was also teeth (Fig. 6). A well-defined hypodense rim was clearly
appreciated. A maxillary occlusal radiograph showed that visible on the coronal sections of CBCT (Fig. 7). Based
the lesion had caused divergence of the roots of the upper on the radiographic appearance and location of the lesion,
left central and lateral incisors (Fig. 4). CBCT revealed a radiographic provisional diagnosis of odontoma and a

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Oral Radiology

Fig. 5  Axial cone beam computed tomography image. A lesion of


mixed density and size of approximately 1 × 1 cm (arrows) is shown
Fig. 7  Coronal cone beam computed tomography image. A defined
on the palatal side of the upper left  central and lateral incisors. The
hypodense rim (arrow) is present around the lesion
lesion extends between both of the roots with an unclear border and is
displacing the teeth

Fig. 8  Histopathological (×10) magnified findings. Abundant irregu-


lar trabeculae of lamellar and woven bone are present in a highly vas-
cular fibrous stroma

trabeculae was composed of spindle- and stellate-shaped


fibroblasts with numerous small and medium-sized capil-
laries. Focal collections of multinucleated giant cells were
Fig. 6  Sagittal cone beam computed tomography image. Clear
demarcation of the lesion is present from the apex of the upper left also seen in the stroma, close to the bony trabeculae (Fig. 8).
central incisor Based on this histopathological report, a diagnosis of
benign osteoblastoma was made. Healing was uneventful
without any swellings or other complications.
differential diagnosis of other fibro-osseous lesions of the
maxilla were given.
Because the lesion was not very vascular and was well Discussion
circumscribed, it was excised in toto and submitted for histo-
pathological examination. Complete hemostasis and primary Benign osteoblastoma rarely occurs in the jaws. Trauma,
closure were performed. Histopathological examination of inflammation, an abnormal local response of the tissues to
the specimen revealed irregular trabeculae of bone along injury, and local alterations in bone physiology are some of
with areas of osteoid and woven bone in a cellular fibrous the reported etiologies of this tumor [3]. There are two main
connective tissue stroma. Large osteocytes were seen within clinicopathological entities of osteoblastoma: the benign and
the trabeculae. Numerous osteoclasts (multinucleated giant aggressive forms. The benign form, which grows slowly
cells) were seen in lacunae in the resorptive areas of the over many years and has a well-defined sclerotic margin, is
bony trabeculae. The fibrous stroma interspersing the bone fairly well vascularized with a mild inflammatory response

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[4]. The aggressive form of osteoblastoma exhibits locally curettage or conservative surgical excision. Wozniak et al.
aggressive behavior with a propensity to recur and has atypi- [4] in their case report involving malignant transformation
cal histopathological features, often making differentiation of an osteoblastoma of the mandible, suggested complete
from low-grade osteosarcoma difficult [5]. Osteoblastomas resection with the margins located in the normal tissues as
can also be classified as cortical, medullary, or periosteal the treatment of choice and recommend additional radio-
depending on which component of the bone is involved [6]. therapy and/or chemotherapy in more aggressive cases. The
Those involving the jaws are either medullary or periosteal, reported recurrence rate of benign osteoblastoma is 13.6%,
with the cortical variant commonly seen in extragnathic which makes surgical excision of the entire tumor the main
sites [7]. Barlow et al. [8] suggested that pain is an early treatment of choice because there is high chance of recur-
symptom in patients with the cortical type of osteoblastoma rence when curettage is performed [3].
because of restriction of expansion by cortical bone and
close proximity to the periosteum, which has a rich nerve
supply. Conversely, when the tumor arises from medullary Conclusion
bone, impingement on the periosteum occurs only when the
tumor becomes large in size. Pain is also an early symptom We have herein reported an interesting case of osteoblastoma
because of prostaglandin 2 production; prostaglandin 2 is not with distinct clinical, histological, and radiographic patterns.
present in giant cell tumors, for which pain is present only To the best of our knowledge, this is the first reported case of
in advanced lesions [8]. benign osteoblastoma in the anterior maxillary region pre-
Generally, this lesion affects patients in the first three dec- senting in a female patient. Although benign osteoblastoma
ades of life, with a male:female ratio of 2:1. The patient’s is a rare tumor, our case report suggests that it can be consid-
age in the present case was within the reported age range of ered as a differential diagnosis for a swelling in the anterior
conventional osteoblastomas; i.e., 5–24 years, the second region of the maxilla mimicking a periapical pathosis.
decade being the most prevalent age [9]. The patient was
female, whereas the literature suggests a male predilection.
Osteoblastoma occurs more frequently in the spinal column Compliance with ethical standards 
and long bones. Other less common sites are the bones of
the skull cap, extremities, and face [9]. Of the reported cases Conflict of interest  All authors declare that they have no conflict of
of benign osteoblastoma, only 10–15% involved the jaws; of interest.
these, the mandibular posterior region was affected in most Research involving human participants  All procedures followed were
cases [9]. Only two cases of benign osteoblastoma have been in accordance with the ethical standards of the responsible committee
reported in the palate (posterior aspect) [10, 11]. Our patient (institutional and national) and with the Helsinki Declaration of 1964
was a female with osteoblastoma of the maxillary anterior and later versions.
region. Benign osteoblastoma clinically presents with slight Informed consent  Informed consent was obtained from the patient.
pain, swelling, and expansion of the bony cortex. It has a
limited growth potential and typically does not exceed 4 cm Research involving animal rights  This article does not contain any
in diameter [3]. This is consistent with our case except that studies with animal subjects performed by any of the authors.
our patient exhibited no cortical expansion.
The radiographic findings of osteoblastoma are not very
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