OSTEOSARCOMA OF MAXILLA: - A CASE OF MISSED INITIAL

DIAGNOSIS

*Antony George, **Varghese Mani , * Sunil S, *Sreenivasan BS, * Devi Gopakumar.

Abstract
Primary neoplasms of human skeleton are rare, accounting for 0.2% of overall human
tumor burden. Osteosarcoma accounts for 15-35% of all primary bone tumors. While
osteosarcoma of jaw bones are even rarer, representing 4-8% of all osteosarcomas. Peak
incidence for jaw osteosarcoma is 3rd-4th decade, while osteosarcomas of long bones show a
bimodal age distribution. Here we report an unusual case history of a 54yrs old male with
swelling in anterior gingiva of maxilla with mobility of teeth, which on x-ray was defined as non-
contributory, and on incisional biopsy was diagnosed as giant cell granuloma. However on
histopathological examination after wide surgical excision, it showed large areas of lobular
malignant cartilage with few areas of disorganized tumor osteoid and atypical spindle cells, and
was reported as chondroblastic osteosarcoma.

Keywords: bone tumors, osteosarcoma, osteogenic sarcoma, chondroblastic, jaw, maxilla.

Introduction (EOS) arise in soft tissues, commonly in

Osteosarcoma are malignant bone thigh, upper extremity and retroperitoneum,
tumors characterized by formation of with no case reports of any in the oral
disorganized immature woven bone or cavity.8
osteoid tissue from mesenchymal tumor Men develop osteosarcomas more
cells.1-5 Osteosarcoma accounts for 15-35% frequently than women.4,5,9 Among
of all primary bone tumors followed by osteosarcomas of jaws 60% were males.10
chondrosarcoma and Ewing’s sarcoma, and The age-specific frequencies and incidence
is the second most common malignant bone rates of conventional osteosarcoma is
1,2
tumor after multiple myeloma. bimodal, whereas the peak incidence for jaw
Osteosarcoma of jaw bones are uncommon, osteosarcomas were in the 3rd-4th decade
representing 4-8% of all osteosarcomas.2,4-7 (33yrs).1,5,9 Osteosarcoma of jaws show a
More rarely extraskeletal osteosarcoma predilection for mandible, however some
Oral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
studies found that it affects the mandible normal limits. He gave no history of any
4,5,9-11
and maxilla almost equally,. Mandibular habits – smoking, chewing or alcohol. A
tumors arise more frequently in posterior clinical diagnosis of inflammatory gingival
horizontal ramus, while maxillary lesions are swelling and chronic periodontitis was made
commonly discovered in alveolar ridge, at local health center, and extraction of 14
sinus floor and palate.5,11 was done under local anesthesia and
This article presents an unusual case antibiotic coverage. Excisional biopsy of the
report of 54yrs old male with well-defined gingival swelling was also performed during
swelling in anterior gingiva of maxilla with extraction, which was histopathologically
mobility of associated teeth and pain. On x- reported as giant cell granuloma. Recurrence
ray investigation at a local health care center of the lesion occurred at the same site, and
in another country a non-contributory was slowly increasing in size. Patient was
finding was given. Clinically it was advised re-excision of the gingival swelling.
diagnosed as an inflammatory lesion of After 1 month he reported to our hospital
gingiva and on incisional biopsy at the same on medical leave for surgical excision of the
center it was diagnosed as giant cell said gingival swelling. Clinical examination
granuloma. Later on histopathological revealed a 5 x 6 cm well-defined firm sessile
examination after wide surgical excision at asymptomatic gingival swelling in 13-15
our hospital it was reported as regions, overhanging the 14 extraction
chondroblastic osteosarcoma. Post socket and covering the cervical third of 13
operative x-ray’s & CT scan’s showed no & 15. Surface was smooth and shiny having
classical imaging features of osteosarcoma, grayish white to pale pink mucosa
or any other bony or soft tissue lesion. interspersed with ulcerated areas.
Case History Keeping the non-contributory OPG
A 54yrs old male of malayalee origin and histopathological report of giant cell
(Kerala, S.India) working as businessman in granuloma in mind, wide surgical excision
Dubai (UAE) since 12yrs reported to a local with extraction of 13 & 15, along with
health center there with mobility of upper buccal cortical plate and interdental alveolar
side teeth and pain on chewing, since 2 bone septal osteotomy was performed under
weeks. On examination he was reported to general anesthesia. The gingival lesion could
have a localized gingival swelling with grade be excised cleanly from the underlying bone
st
II mobility of 14 (right maxillary 1 during surgical procedure. No perforation to
premolar). Ortho pantograph (OPG) maxillary sinus or deeper palatal bone
examination was reported as being within erosion was noted. Primary closure of

Oral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
surgical site with buccal flap using vycryl 3.0 Central (Medullary)
sutures was performed. Excised lesion was a. Conventional osteosarcoma.
submitted for histopathological i. Chondroblastic.
examination. ii. Fibroblastic.
Histopathology iii. Osteoblastic.
Given soft tissue section showed b. Telangiectatic osteosarcoma.
lobular areas of chondroblastic c. Intraosseous well-differentiated
differentiation with atypical chondrocytes osteosarcoma.
and chondroblasts, and actively proliferating d. Small cell osteosarcoma.
fibroblast showing nuclear and cellular b. Low-grade central osteosarcoma.
pleomorphism. Peripheral areas of few of c. High-grade central
these neoplastic chondroid islands showed osteosarcoma.
formation of disorganized woven immature e. Secondary osteosarcoma.
bone. Focal areas of atypical haphazardly
arranged spindle shaped cells with Surface (Peripheral)
myxomatous areas were also seen. Stroma a. Parosteal (juxtacortical) well-
showed few diffuse bizarre multinucleated differentiated osteosarcoma.
giant cells. Deeper areas showed chronic b. Periosteal osteosarcoma.
inflammatory cell infiltrate of lymphocytes, c. High-grade surface
plasma cells and eosinophils, along with osteosarcoma.
areas of numerous vascular channels, and
Modified from WHO 2002 classification
extravasated RBC’s. Based on these varied
of bone tumors and European and
histopathological findings a diagnosis of
American Osteosarcoma Study Group
chondroblastic osteosarcoma was given.
(EURAMOS) classification.
Table1: Osteosarcoma Variants.

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 Fig3: Post Operative Maxillary Occlusal
Radiograph.
Fig1: Clinical Appearance. Well-defined firm
sessile asymptomatic gingival swelling in 13-
15 regions, extending from sulcus to palatal
surface, overhanging 14 extracted tooth
socket and covering 13 & 15 tooth surface.

Fig4: Post Operative Maxillary Computed

Tomography (CT scan).

Fig2: Pre Operative Ortho Pantograph

(OPG).

Fig5: Photomicrograph 10x. Lobular areas

of neoplastic cartilage.

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 metallic implants, joint prostheses, and in
some genetic syndromes – Li-Fraumeni
syndrome, hereditary retinoblastoma and
Rothmund-Thomson syndrome.1,6,9,10 Our
case appears to have developed de novo.
The peak incidence for jaw
rd th
osteosarcoma is 3 -4 decade (33yrs), which
is about 10-15yrs later than the mean age of
long bone osteosarcomas.4,5,9-11 But this
Fig6: Photomicrograph 10x. Areas of tumor
patient was a 54yrs old male. Swelling, pain,
osteoid.
and general discomfort are the usual non-
specific clinical findings.2,5,9,11 Present case
had well-defined firm sessile asymptomatic
gingival swelling with pain associated with
mobility of teeth (Fig1). Radiographic
findings vary from sclerotic to mixed
sclerotic (moth-eaten or cumulus cloud) to
radiolucent lesion.1,5,9 “Classic” sunray or
sunburst appearance due to osteophytic
bone production on surface is noted in 50%

Fig7: Photomicrograph10x. Chondromyxoid of jaw osteosarcoma, best demonstrated in

areas with atypical spindle cells. occlusal radiographs and CT scan.2,5,6,9,11

Panoramic radiograph may show

Discussion Garrington’s sign – widening of the

Osteosarcoma is the most common periodontal ligament space around affected
non haemopoietic primary malignant tumor teeth with tapered resorption of tooth roots
of bone.1,4,5,9,11 Majority of primary bone due to tumor infiltration.2,5,6 The x-ray

malignancies arise do novo, but some evaluation of present case was initially
apparently develop in association with reported with no abnormal findings. On re-

recognizable precursors (precancerous evaluation of the pre-operative pantograph,

conditions) such as Paget disease, fibrous after receiving a histopathological report of
dysplasia, bone infarcts, chronic osteosarcoma, it showed a irregular
osteomyelitis, trauma, and exposure to multilocular radioluceny in 13 region with

radiation.1,6,9 It also has been associated with suggestive root resorption (Fig2). One of

Oral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
the reasons why this could have been and radiographically if it presents itself as a
missed initially is that the canine regions gingival swelling which may clinically appear
frequently show radiolucent radiographic as inflammatory lesion, and with no
artifacts, due to curvature of the jaws. The contributory imaging investigatory findings.
post operative xrays and CT scans showed In an incisonal biopsy too the diagnosis can
no contributory changes associated with be missed if adequate and proper site has
osteosarcoma (Fig3,4). not been chosen. After diagnosing the lesion
Histopathologically all as chondroblastic osteosarcoma, the present
osteosarcomas have sarcomatous stroma case was referred to higher oncology center
that directly produces tumor osteoid.1,4,5,9,11 for further evaluation and treatment.
Variable histological patterns have been Chemotherapy was initiated and he is under
described, and have been designated as long term periodical follow-up.
variants based on clinical, imaging and
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* Dept. of Oral & Maxillofacial Pathology, Mar Baselios Dental College,

Kothamangalam.Kerala, India
** Dept of Oral & Maxillofacial Surgery, Mar Baselios Dental College, Kothamangalam.
Kerala, India

Oral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225
Oral & Maxillofacial Pathology Journal [ OMPJ ] Vol 1 No 1 Jan- Jun 2010 ISSN 0976-1225