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Journal Pre-Proof: Oral Surg Oral Med Oral Pathol Oral Radiol
Journal Pre-Proof: Oral Surg Oral Med Oral Pathol Oral Radiol
PII: S2212-4403(21)00608-8
DOI: https://doi.org/10.1016/j.oooo.2021.09.001
Reference: OOOO 4717
To appear in: Oral Surg Oral Med Oral Pathol Oral Radiol
Please cite this article as: Scarlet Charmelo-Silva DDS , Allison Buchanan DMD, MS ,
Sajitha Kalathingal BDS, MS , Rafik Abdelsayed DDS, MS , Osteosarcoma of the Jaws: Report
of 3 Cases with Emphasis on the Early Clinical and Radiographic Signs, Oral Surg Oral Med Oral
Pathol Oral Radiol (2021), doi: https://doi.org/10.1016/j.oooo.2021.09.001
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Radiographic Signs
1. Assistant Professor of Oral Medicine Department of Oral Biology & Diagnostic Sciences,
The Dental College of Georgia at Augusta University
2. Professor of Oral Radiology Department of Oral Biology & Diagnostic Sciences, The
Dental College of Georgia at Augusta University
3. Professor of Oral Radiology Department of Oral Biology & Diagnostic Sciences, The
Dental College of Georgia at Augusta University
4. Professor of Oral Pathology Department of Oral Biology & Diagnostic Sciences, The
Dental College of Georgia at Augusta University
Email: albuchanan1@augusta.edu
Phone: 706-721-2264
Fax: 706-721-4937
This article reviews the early radiographic signs of osteosarcoma occurring in the interradicular
region. Early diagnosis of osteosarcoma of the jaws is paramount as it significantly improves the
Osteosarcoma is the most common primary malignancy of the jaws and is treated by radical
surgical resection. Early detection is crucial since removing the lesion with clean margins
interradicular region of the mandible, two of which were thought to represent non-malignant
processes upon initial presentation. All three demonstrated early but significant clinical and
radiographic features indicative of the malignant disease process. Radiographically, all of these
cases had in common unilateral periodontal ligament (PDL) space widening, displacement of
teeth, and growth of the crestal bone. Clinically, all three cases presented with a painless
intraoral swelling. Case 1: A 20 year old African American male presented with a swelling and
high grade was made. Case 2: A 75 year old Caucasian male presented with a 2 x 2 cm
of chondroblastic osteosarcoma was made. Case 3: A 63 year old Caucasian female presented
of chondroblastic osteosarcoma was made. All three cases were treated with wide surgical
resection.
Introduction
accounts for approximately 20% of all bone cancers and has a predilection for the distal femoral
metaphysis, proximal tibia and humeral metaphysis. 2,4,5 Osteosarcoma occurs most frequently
during the second decade of life but there is a smaller peak of incidence after the age of 50.4-6
According to the World Health Organization, there are several variants of osteosarcoma
conventional osteosarcoma, which arises centrally within bone, is the most frequent type.
matrix production. Of these subtypes osteoblastic, chondroblastic, and fibroblastic variants are
the most common.4 Secondary osteosarcoma arises from pre-existing conditions and has been
osteosarcomas.2,4,5,8 However, when osteosarcoma occurs in the craniofacial bones, the mandible
and maxilla are the most common sites of occurrence. In the jaws, osteosarcoma accounts for 8%
of all malignancies and is the most common primary malignancy of the jaws.3,9 Osteosarcoma of
osteosarcoma of the jaws has a clinical behavior and natural history distinct from osteosarcoma
of the trunk and long bones. 3,10,11 Jaw osteosarcomas usually affect patients 10–20 years older,
are less likely to give rise to distant metastases, and are associated with a better 5-year survival
rate.2,4,6,8,10-15 The mandibular body is most frequently involved, followed by the angle,
symphysis and ascending ramus.16 Maxillary tumors have been reported to show a predilection
for the posterior regions of the alveolar process and the maxillary sinus.5,16,17
lesions, or mixed lytic and sclerotic lesions.3 An important radiographic feature is Garrington’s
sign, which is a symmetric widening of the PDL due to infiltration of tumor cells and is
mandible which demonstrated early but significant clinical and radiographic features and 2)
Case Series
Case 1
Clinical presentation
A 20 year old African American male presented with a 1.5 month duration of an intraoral
swelling in the anterior mandible with loosening of his teeth (Figure 1). No pain or paresthesia
were present. The patient’s physician prescribed antibiotics, but the lesion failed to resolve.
Intraoral exam revealed a swelling extending from the mandibular left lateral incisor across the
midline to the mandibular right first premolar (Figure 1). The lesion was indurated, hyperemic
and involved both the buccal and lingual aspects (Figure 1). The mandibular right lateral incisor
was displaced lingually and the mandibular incisors had class I mobility (Figure 1). The
Panoramic and Cone beam computed tomography (CBCT) imaging exams were ordered.
The panoramic radiograph revealed displacement of the mandibular right lateral incisor with
unilateral widening of the periodontal ligament (PDL) space. Unilateral widening of the PDL
space was present on the mandibular right canine as well. The interradicular bone between these
teeth had a patchy sclerotic appearance and growth of the crestal bone was evident. The
periapical bone was radiolucent. Figure 1 demonstrates the panoramic imaging findings. The
CBCT exam revealed a patchy sclerotic bone overgrowth between the mandibular right lateral
incisor and canine (Figure 2). Interestingly, despite some interruption, the adjacent facial plate
appeared relatively normal in contour (Figure 2). A surgical biopsy was performed. The surgical
specimen was decalcified before tissue processing. Formalin-fixed, hematoxylin and stained
neoplastic osteoid production (Figure 2A and B). The cellular proliferation is characterized by
pleomorphic polygonal and occasionally spindled cells with round and ovoid hyperchromatic
nuclei, some of which exhibited atypical mitotic figures (Figure 2B). The cellular elements
were intermixed with prominent lacy and diffuse eosinophilic osteoid matrix exhibiting variable
degrees of mineralization. A diagnosis of osteoblastic osteosarcoma, high grade was made. The
patient underwent resection of his mandible from the mandibular right first molar to the
Case 2
Clinical presentation
A 75 year old Caucasian male presented with a 2 x 2 cm asymptomatic, fixed and expansile
lesion in the left anterior mandible. The patient reported that he noticed a painless mass on his
gum two months prior. The patient sought out care by his dentist. The medical history was
positive for coronary artery disease, hypertension, skin cancer (3 lesions of unknown type), a 57
year history of smoking ½ pack/day, and the use of smokeless tobacco for 30 years.
imaging were acquired. The panoramic radiograph revealed very similar findings as that of case
1. The lesion presented with hyperdense, osteosclerotic crestal bone overgrowth between the
mandibular left canine and lateral incisor. There was unilateral widening of the PDL space on
these teeth and the left lateral incisor was displaced (Figure 3). In addition to the radiographic
findings from the panoramic radiograph, the CBCT exam revealed that the patchy sclerotic bone
pattern extended beyond the facial plate and that the lesion extended from the mandibular left
canine to the midline (Figure 4). Interestingly, despite the spread of the tumor beyond the facial
plate, in general, the alveolar bone maintained an overall normal contour (Figure 3). In cross
sections, the patchy sclerotic bone pattern displayed a “sunburst” pattern (Figure 3). The MDCT
exam consisted of multidetector helical scanning in the axial projection at 2.5 mm slice thickness
and pitch of 0.52 mm acquired post administration of contrast (Omnipaque 350). Bone and soft
tissue density windows were available. The MDCT imaging confirmed the less aggressive nature
of the histopathological diagnosis (see below) through the absence of lymphadenopathy, the
absence of any notable spread into the mandibular body, and the absence of an adjacent soft
tissue component (Figure 4). The patient’s dentist referred him to ENT for a biopsy. The
surgical specimen was decalcified before tissue processing. Microscopic examination of the
decalcified hematoxylin and eosin stained sections revealed a lobular growth with predominantly
matrix supporting numerous mononuclear and multinucleated cellular lacunae, which contained
cells with hyperchromatic and pleomorphic nuclei and atypical mitotic figures. These areas
tissue with prominent osteoblasts exhibiting hyperchromatic nuclei (Figure 4). A diagnosis of
chondroblastic osteosarcoma was made. The patient was treated with resection from the left first
Case 3
Clinical presentation
A 63 year old Caucasian female presented with a 5-6 mm asymptomatic pink, red and raised
ulcerated firm lesion in the interproximal gingiva between the mandibular left premolars for at
least 1 month. The patient's medical history was essentially unremarkable except for
hyperlipidemia for which she was on medications. She denied smoking or any other use of
substances. Clinically, the lesion was thought to be benign and a differential diagnosis of
An intraoral and panoramic radiograph as well as MDCT were acquired. On the conventional 2-
dimensional radiographs, there was unilateral PDL space widening along the mandibular left first
and second premolars (Figure 5). Spacing of the mandibular premolars was present and there
was growth of the crestal bone (Figure 5). Changes in the interradicular bone pattern were
minimal and difficult to appreciate on 2-dimensional imaging. The MDCT exam consisted of
multidetector helical scanning in the axial projection at 2.5 mm slice thickness and pitch of 0.4
mm. Bone and soft tissue density algorithms were available. Contrast was not administered. A
thin bone series was available as well with a slice thickness of 0.7 mm. The MDCT imaging
confirmed that little bone growth was present and only minimal changes in the bony contour at
the level of the ridge crest existed (Figures 5 and 6). Since only minimal bony changes were
visible, the radiographic findings that were characteristic of osteosarcoma were better visualized
on the axial CT images and the custom cross sections created from the thin CT bone series
(Figures 5 and 6). The patient was referred to a local oral surgeon who surgically removed the
gingival nodule and submitted the specimen for histopathological evaluation. Histologic
gingival nodule covered by ulcerated squamous epithelium and supported a cellular myxoid
mesenchymal tissue proliferation with chondroid and osteoid matrix production. The cellular
haphazardly arranged spindled mesenchymal cells with elongated and ovoid hyperchromatic and
pleomorphic nuclei, some of which exhibited mitotic figures. These are embedded in abundant
ground substance, which imparted a myxoid appearance, focally interspersed by chondroid and
osteoid matrix (Figure 6). A diagnosis of chondroblastic osteosarcoma was made. The patient
was treated with segmental resection from the midline to the left molar region and is currently in
Discussion
The reported three cases shared early radiographic and clinical features of osteosarcoma
outcome. While chemotherapy prior to surgery is the treatment of choice for osteosarcomas of
the long bones, this treatment option is controversial for osteosarcomas of the jaws.2,3,11,14,15 This
is perhaps due to their differences in behavior. Osteosarcomas of the jaws, unlike osteosarcomas
of the long bones, have decreased incidence of distant metastasis.8,11,12,14,15 In a study of 214
patients.14 Therefore, osteosarcomas of the jaws, relative to osteosarcomas of the long bones,
have a better prognosis.12 In a study by ElKordy and colleagues only 1 out of 21 cases of
osteosarcomas of the jaws had distant metastases at presentation.12 Consequently, the mainstay
of therapy for osteosarcomas of the jaws is radical resection.2,3,10-12,14 Bertin and colleagues
report an overall survival rate of 77% (at 5 years) after complete resection of local disease.2 The
ability to remove the lesion with clean margins has the biggest effect on outcome for
between clean margins and improved survival with a 70% increase in survival for every 1 cm
radiographic and clinical changes that can aid in the detection of osteosarcoma at an earlier stage.
Radiographically, all of these cases had in common unilateral PDL space widening,
displacement of teeth, and growth of the crestal bone. Symmetric widening of the PDL space has
been reported as an early radiographic sign associated with osteosarcoma of the jaws.6,8
Garrington et al. reported that in some cases osteosarcomas can show symmetric widening of the
PDL space before displaying any other prominent radiographic findings.10 The widening of the
PDL space is the result of tumor infiltration.6 In the case of osteosarcoma occurring in an
interradicular location, PDL infiltration is the route of least resistance and therefore it is a
common early radiographic finding. In fact, in a case series reported by Garrington et al, for the
few patients with osteosarcoma of the mandible that did not present with swelling, the most
common presenting symptom was loose teeth and; therefore, they considered this to be an
Two of our three cases demonstrated a change in the interradicular bone pattern that
presented as sclerotic in nature. Changes in the interradicular bone pattern were not easily
identifiable on two dimensional imaging for case #3. Additionally, although osteophytic bone
production was demonstrated on CT for all three cases, only one of them (case #2) produced the
classic “sunburst” pattern. These findings suggest that tooth displacement and PDL space
widening may precede obvious radiographic changes in bone pattern and emphasizes the
Despite prominent tumor growth in cases 1 and 2 the overall contour of the alveolar bone
was relatively normal, particularly the buccal and lingual portions. This radiographic finding is
associated with malignancy. A similar finding of malignant tumor infiltration without affecting
the morphology of the alveolar process was reported by Buchanan et al 2015. In this case the
malignancy was non-Hodgkin lymphoma, none the less, this radiographic finding demonstrates
This is consistent with the reported preponderance of the chondroblastic type in craniofacial
osteosarcomas, particularly the mandible and maxilla when contrasted with the long bones.
On clinical presentation, all three of our cases presented with swelling but no pain. This
is in contrast to osteosarcoma of other bones that commonly presents with pain.10 Indeed
however, the most common clinical sign associated with osteosarcoma of the jaws is
swelling was 100% in 14 cases of osteosarcoma of the jaws.6 In contrast, only 2 of their 14 cases
presented with pain.6 In another series, Garrington and colleagues reported swelling in 50 out of
54 cases of jaw osteosarcomas while pain was reported for only 23.10 Similarly, Granowski-
LeCornu and colleagues reported swelling in 74.5% and pain in 31.9% of 47 cases of
osteosarcomas of the jaws.15 Therefore, swelling without pain is a common clinical presentation
The growth rate of osteosarcoma is usually rapid.10 Two of our cases reported a 1.5-2
month duration of intraoral swelling and the third case reported that the swelling was present for
at least 1 month. In a case series of 17 jaw osteosarcomas, Ogunlewe et al. reported an average
duration of symptoms of 6.1 months.5 Although the duration of symptoms can vary from 2 to 18
months, a history of intraoral swelling with rapid growth and the absence of pain can be
There was a wide age distribution at initial disease presentation in the currently reported
three cases. One of the patients was young (age 20) while the other two patients were ages 75
and 63. The mean age of occurrence for osteosarcomas of the jaws has been reported to range
from 30-43 years.2,3,10-12,14 Nevertheless, and similar to our findings, Garrington et. al reported a
wide age range for osteosarcomas occurring in the mandible (age range 4-64 years) and found
that occurrence was just as likely in the elderly population as in the younger population.10
Paparella and colleagues also found that the occurrence rate of mandibular osteosarcomas was
similar throughout life.3 The fact that osteosarcomas of the mandible can occur over a wide age
range only further emphasizes the importance of the radiographic and clinical findings that aid in
early detection.
within the mandible, each of which presented with important early radiographic and clinical
indicators of this disease process. The radiographic findings included unilateral PDL space
widening, displacement of teeth, and growth of the crestal bone. The clinical findings included a
painless intraoral swelling with a history of rapid growth. Since the ability to remove the lesion
with clean margins has the biggest effect on outcome for osteosarcomas of the jaws, early
detection is key for these patients.3,11,14,15,18 The importance of close radiographic inspection
along with clinical correlation cannot be stressed enough in their importance in the detection of
None
No financial support was received from any external source or vendor for this project.
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