|Year : 2018 | Volume
| Issue : 1 | Page : 18-21
Parosteal osteosarcoma of the maxilla: A case report and review of the literature
Osawe Felix Omoregie1, Anthony Osaguona1, Ehimwenma Ogbeide2
1 Department of Oral Surgery and Pathology, University of Benin, Benin City, Nigeria
2 Department of Oral Diagnosis and Radiology, University of Benin, Benin City, Nigeria
|Date of Submission||22-Oct-2013|
|Date of Acceptance||26-Jul-2018|
|Date of Web Publication||20-Jan-2020|
Dr. Osawe Felix Omoregie
Department of Oral Surgery and Pathology, University of Benin, Benin City
Source of Support: None, Conflict of Interest: None
This study reports a rare case of parosteal osteosarcoma, a low-grade malignant lesion diagnosed in a 67-year-old female who presented with a slow growing oval-shaped, well circumscribed, bony hard, left maxillary swelling of 4 months' duration; extending from the region of the left upper lateral incisor tooth (22) to the left upper first molar tooth (26) and fixed to the overlying gingival. Oblique lateral radiograph showed the left maxillary alveolar ridge with lobulated ossified mass. The clinical differential diagnoses of the lesion were osteoma, and fibrous dysplasia histopathological diagnosis of parosteal osteosarcoma was made after incisional biopsy of the lesion. Surgical resection (with a wide margin) of the affected maxillary segment and the associated teeth was performed, and postsurgical biopsy confirmed the diagnosis of parosteal osteosarcoma. Parosteal osteosarcoma shares similar clinicopathological characteristics with some periosteal lesions. However, special imaging techniques and histopathological evaluation remain the most reliable tools for definitive diagnosis of these lesions.
Keywords: Diagnostic challenge, maxilla, parosteal osteosarcoma, prognosis, treatment
|How to cite this article:|
Omoregie OF, Osaguona A, Ogbeide E. Parosteal osteosarcoma of the maxilla: A case report and review of the literature. Niger J Surg Sci 2018;28:18-21
|How to cite this URL:|
Omoregie OF, Osaguona A, Ogbeide E. Parosteal osteosarcoma of the maxilla: A case report and review of the literature. Niger J Surg Sci [serial online] 2018 [cited 2020 Feb 17];28:18-21. Available from: http://www.njssjournal.org/text.asp?2018/28/1/18/276111
| Introduction|| |
Parosteal osteosarcoma is a low-grade malignant lesion accounting for 1.6%–2% of all malignant bone tumors. The lesion is found commonly in the distal dorsal femur, with a wide age range of 2nd–8th decades of life and female predilection., In contrast, a recent report by Samardzisk et al., shows a slight male predilection for parosteal osteosarcoma, which presented mostly as painless swelling with an average duration on presentation of 12.5 months and radiologically the lesions showed a densely ossified and lobulated mass. The histology of most of the lesions showed regular trabeculae interspersed with a scarcely cellular, spindle cell, and collagenous stroma, with low-to-moderate cellular atypia and low mitotic activity.
However, parosteal osteosarcoma is rare in the oral cavity and most cases reported were found in the maxilla, occurring in both genders.,, Hewitt et al. reported  a case of maxillary parosteal osteosarcoma of 1-year duration in a 32-year-old male that presented as a firm, painless maxillary swelling with palatal extension. The patient had a partial maxillectomy without adjunct therapy, and no evidence of local recurrence was observed after 3 years. Bianchi et al. reported a maxillary parosteal osteosarcoma with a sclerotic radiological pattern. Furthermore, radiographically, the typical appearance is that of an exophytic mass which has a lobulated configuration and is adjacent to bone with central dense ossification. A fine radiolucent cleft may be found between the tumor and adjacent outer table in cranial parosteal osteosarcoma  and the string sign has been described as representing the cleavage plane separating the tumor and adjacent normal cortex. There is often cortical thickening and lack of aggressive periosteal reaction. This lesion radiologically mimics myositis ossificans  and half of the cases of myositis ossificans adheres to the periosteum and are referred to as parosteal myositis ossificans., Other parosteal lesions that are commonly misdiagnosed as parosteal osteosarcoma, because they share similar clinicopathological characteristics with parosteal osteosarcoma are osteochondroma,, osteoma, juxtacortical chondrosarcoma, high-grade surface osteosarcoma, and periosteal chondroma.,,,
This study reports a rare case of parosteal osteosarcoma, a low-grade malignant lesion diagnosed in our center, to highlight the diagnostic challenges, treatment considerations, and prognosis of the lesion, compared with other reactive, benign, and malignant jaw lesions.
| Case Report|| |
A 67-year-old female presented with a slow growing left maxillary swelling of 4 months' duration, in the Oral and Maxillofacial Surgery Department, University of Benin Teaching Hospital, Benin City, Nigeria. The lesion was oval shaped, well circumscribed, bony hard, measuring 5 cm by 2 cm, extending from the region of the left upper lateral incisor tooth (22) to the left upper first molar tooth (26) and fixed to the overlying gingival [Figure 1]. Plain radiographs including posterior-anterior skull view, oblique lateral and occlusal views of the maxilla were taken. Only the oblique lateral view clearly demonstrated the lesion in the left maxillary alveolar ridge with lobulated ossified mass. The surrounding alveolar bone and adjacent dentition appeared normal [Figure 2]. The clinical differential diagnoses of the lesion were osteoma and fibrous dysplasia.
|Figure 1: Clinical photograph showing parosteal osteosarcoma presenting as a left maxillary swelling|
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|Figure 2: Oblique lateral radiograph showing the left maxillary alveolar ridge with lobulated calcific mass|
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Histopathological diagnosis of parosteal osteosarcoma was made after incisional biopsy of the lesion. The lesion showed thick islands and streaming parallel trabeculae of woven bone with intervening sparsely cellular fibrous connective tissue stroma, with outer thick periosteum (fibrovascular capsule) [Figure 3]. There are foci showing low-cellular atypia consisting of bone cells with pale, pleomorphic nuclei, and no obvious mitosis [Figure 4].
|Figure 3: Parosteal osteosarcoma showing streaming parallel trabeculae of woven bone with intervening sparsely cellular fibrous connective tissue stroma and outer periosteum (H and E, ×10)|
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|Figure 4: Parosteal osteosarcoma showing low cellular atypia consisting of bone cells with pale, pleomorphic nuclei, and no obvious mitosis (H and E, ×40)|
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Surgical resection (with a wide margin) of the affected maxillary segment and the associated teeth was carried out, and postsurgical biopsy confirmed the diagnosis of parosteal osteosarcoma. No adjunct therapy was included in the patient's treatment. The patient attended two postoperative follow-up visits in 1 month, with the operation site showing a defect in the left maxilla [Figure 5], and there was no evidence of postoperative complication or recurrence. The patient was referred to the prosthodontist for review and rehabilitation with upper partial denture to replace the missing teeth. Further follow-up visits in the Oral and Maxillofacial Surgery Unit were scheduled for 3-month, 6-month, and 1-year periods.
|Figure 5: Clinical photograph showing postsurgical left maxillary defect|
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| Discussion|| |
Parosteal osteosarcoma is rare in the oral cavity and most cases reported were in the maxillary region.,, Similarly, this study reports a case of parosteal osteosarcoma in an elderly female patient, with a long-standing history of maxillary swelling, which was clinically misdiagnosed as osteoma or fibrous dysplasia. This is consistent with previous reports of misdiagnosis of parosteal osteosarcoma for other bone forming tumors including osteoma and fibrous dysplasia., Cross-sectional imaging with computed tomography and magnetic resonance imaging, although not done in the case presented, gives a better anatomic delineation of such lesions and would have assisted in definitive diagnosis of this lesion. The recognition of the distinct radiological and histopathological characteristics of these lesions is useful to ascertain the definitive diagnoses of these lesions. The diagnostic challenges and treatment considerations for parosteal osteosarcoma emphasizes the need for close cooperation between oral and maxillofacial surgeons, pathologists, and radiologists in the management of this lesion.
A comparison of the treatment and prognosis of low-grade parosteal osteosarcoma with those of the suspected reactive lesion (fibrous dysplasia) and benign lesion (osteoma) shows that parosteal osteosarcoma requires wide margin of resection to prevent recurrence,,, while fibrous dysplasia is usually a self-limiting lesion, which may require chemotherapy (with nonsteroidal anti-inflammatory drug [NSAIDs] and bisphosphonate) or surgical paring down and it has potential for malignant change. Similarly, osteoid osteoma has recently been reported to have potential to regress, so conservative medical treatment with NSAIDs is recommended, while surgery is reserved for rare cases with persistent symptoms.
Previous reports , on the pathomorphology of surface osteosarcoma identified the histological variants of this lesion as low-grade parosteal osteosarcoma (Broders Grade 1–2), periosteal osteosarcoma, an intermediate lesion (Broders Grade 2–3), high-grade surface osteosarcoma (Broders Grade 3–4), and dedifferentiated parosteal osteosarcoma, a high-grade lesion (Broders Grade 3–4). Bertoni et al., also observed that surgical resection with wide margin, without adjunct therapy, was adequate for low-grade parosteal osteosarcoma, and this gives a good prognosis; whereas, the dedifferentiated parosteal osteosarcoma required surgical resection with wide margin, together with adjunct chemotherapy, to avoid metastasis of the lesion and a high rate of mortality. In agreement with the prescribed treatment options for the histological diagnosis of a low-grade parosteal osteosarcoma, the patient had only surgical resection (with a wide margin) without adjunct therapy. Furthermore, rehabilitation of the postoperative maxillary defect with upper partial denture was initiated within the 1-month follow-up visits.
| Conclusion|| |
Parosteal osteosarcoma shares similar clinicopathological characteristics with other periosteal lesions. However, special imaging techniques and histopathological evaluation remain the most reliable tools for definitive diagnosis of these lesions. This study recommends close cooperation between oral and maxillofacial surgeons, pathologists, and radiologists in the management of parosteal osteosarcoma.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]