Periosteal Osteosarcoma of the Mandible: A Case Report Roman G. Meyliker, DMD,* and Robert A. Strauss, DDS, MDy This report describes a rare case of a periosteal osteosarcoma of the mandible in a 50-year-old AfricanAmerican woman who showed no underlying bony changes at panoramic radiography or computed tomography. This report describes the diagnostic workup used to obtain the definitive diagnosis and the surgical treatment and recommended method for subsequent tumor surveillance. Emphasis is placed on distinguishing periosteal osteosarcomas as a separate entity from conventional intraosseous osteosarcomas in the head and neck region. Ó 2015 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 73:787.e1-787.e4, 2015 This report describes a rare case of a periosteal osteosarcoma of the mandible in a 50-year-old woman who showed no underlying bony changes at panoramic radiography or computed tomography (CT). This report describes the diagnostic workup used to obtain the definitive diagnosis and the surgical treatment and recommended method for subsequent tumor surveillance. Emphasis is placed on distinguishing periosteal osteosarcomas as a separate entity from conventional intraosseous osteosarcomas in the head and neck region.
A 50-year-old woman presented to the oral and maxillofacial surgery (OMS) clinic with the chief complaint of ‘ having a growth on my gums.’’ She reported noting something growing in the midportion of the gingiva of the right mandible and perceiving a small lump for approximately 6 months. During the 2 weeks before presentation, she noticed that the lump had rapidly increased in size. She presented to the emergency department at the Virginia Commonwealth University Medical Center (Richmond, VA), where they noted a 1-cm nodular growth on the gingiva between the right mandibular first molar and second bicuspid. Based on the innocuous appearance of the lesion and the lack of any major symptoms, she was given an appointment in the OMS clinic for 2 weeks later.
On presentation to the OMS clinic, her medical history was noted to be important for type 2 diabetes mellitus, arthritis, gastroesophageal reflux disease, and depression. Her surgical history included tonsillectomy, left knee surgery, bilateral carpal tunnel repair, left wrist ganglion cyst removal, bunionectomy, and tubal ligation. She had an allergy to trazodone. She had no noteworthy social history other than a 10 pack-year history of smoking. Her family history was noteworthy for cancer, hypertension, diabetes, and asthma. She had a negative review of systems. The extraoral examination was unremarkable. Cranial nerves II to XII were grossly intact, with no evidence of paresthesia or neurosensory deficit in the distribution of the trigeminal nerve. Intraoral examination showed a 1.5- 1-cm, soft, nonulcerated, reddish, exophytic lesion on the interdental papilla between the right mandibular first molar and second bicuspid (Fig 1). The lesion was not sensitive to palpation and there was no expansion of the mandible. Radiographically, there was evidence of moderate horizontal interdental bone loss. However, there was no evidence of any radiolucent or radiopaque changes within the bone, and there was no evidence of root resorption or tooth displacement (Figs 2, 3). Based on the clinical presentation, the differential diagnosis included pyogenic granuloma, peripheral ossifying fibroma, and peripheral giant cell lesion.
Received from the Department of Oral and Maxillofacial Surgery,
North 11th Street, Box 980566, Richmond, VA 23298-0566;
Virginia Commonwealth University Medical Center, Richmond, VA.
e-mail: [email protected]
REPORT OF CASE
*Chief Resident.
Received August 14 2014
yProfessor and Residency Program Director.
Accepted November 10 2014
Address correspondence and reprint requests to Dr Meyliker:
Ó 2015 American Association of Oral and Maxillofacial Surgeons
Department of Oral and Maxillofacial Surgery, Virginia Common-
0278-2391/14/01717-0
wealth University Medical Center, Wood Memorial Building, 521
http://dx.doi.org/10.1016/j.joms.2014.11.010
787.e1
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PERIOSTEAL OSTEOSARCOMA OF THE MANDIBLE
lesion, the underlying bone was examined and deemed unremarkable. The specimen was sent for histologic examination. The biopsy examination disclosed a well-differentiated chondroblastic osteosarcoma, a diagnosis normally reserved for intraosseous osteosarcomas. After obtaining a negative head and neck CT scan and chest radiograph, this case was reviewed at the tumor board and the diagnosis was changed to a surface osteosarcoma. In this case, excision of the lesion and overlying hard and soft tissues with clinical margins of 1.5 cm was used, which ultimately resulted in a segmental mandibulectomy. The final specimen confirmed the diagnosis of surface osteosarcoma consistent with a periosteal osteosarcoma (Figs 4, 5). The patient was followed immediately after surgery with serial clinical examinations every 2 weeks for the first 2 months and subsequently with serial panoramic radiographs, chest radiograph, and clinical examination every 3 months. There was no recurrence or metastasis after 24 months.
Discussion
FIGURE 1. Initial presentation of soft tissue mass between the right mandibular first molar and second bicuspid. Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
Owing to the benign nature of the differential diagnosis, the decision was made to perform an excisional biopsy under local anesthesia. After excision of the
Juxtacortical osteosarcomas (also known as surface osteosarcomas) were first described in the orthopedic literature by Geschickter and Copeland1 in 1951. They are regarded to have a better prognosis and lower metastatic rate than intraosseous osteosarcomas. These lesions can be divided into parosteal, periosteal, and high-grade surface osteosarcomas.2-4 Parosteal osteosarcomas are considered to have a low rate of malignancy.5 Histologically, they consist of streams of bone trabeculae that are irregularly shaped and usually are covered with a single layer of
FIGURE 2. Panoramic radiograph at initial presentation. Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
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FIGURE 3. Computed tomogram (axial view, bone window) showing a cortical plate without any erosion, no osteolytic changes, and no extracortical bone formation (white arrow). Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
osteoblasts or spindle cells.6 In addition, the intertrabecular spaces of parosteal osteosarcomas contain cellular fibrous tissue.7 In contrast, periosteal osteosarcomas are classically an intermediate grade between that of the usual low-grade parosteal osteosarcoma and that of the conventional higher-grade osteosarcoma.7 Histologically, it contains radiating spicules of
FIGURE 4. Low-power excisional biopsy. This tumor arises from the surface of the mandible with superficial invasion into the cortical bone (hematoxylin and eosin stain; magnification, 100). Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
bone perpendicular to the long axis of the bone with malignant-appearing osteoid in the central portions.2 High-grade surface osteosarcomas should be differentiated from parosteal and periosteal osteosarcomas because they are associated with a poorer prognosis and are more likely to metastasize. Although they arise from the surface, the histologic findings are more typical of conventional osteosarcomas.2 Previous case reports of head and neck surface osteosarcomas have shown radiographic osseous changes on CT scans or panoramic radiographs.8,9 However, the present case was unusual in that it did not have any radiographic evidence of bony abnormality, although it histologically showed bony invasion (Fig 5). The ideal treatment of surface osteosarcomas is unclear owing to the limited number of cases reported to date. The current treatment has essentially been extrapolated from evidence from orthopedic surface osteosarcomas or intraosseous head and neck osteosarcomas. Because these 2 entities are considered more aggressive than head and neck surface osteosarcomas, more conservative resection margins might be appropriate. Unlike squamous cell carcinomas, head and neck osteosarcomas do not require lymph node dissection because they typically metastasize by tumor emboli into the blood stream, most commonly into the lungs. It is important to rule out lung metastasis at the time of diagnosis and provide continuous follow-up with serial chest radiographs. The European Medical Society of
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FIGURE 5. Tumor morphology is similar to that of a chondromyxoid fibroma-like osteosarcoma with malignant cells (arrow) producing osteoid (hematoxylin and eosin stain; magnification, 200). Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
Oncology has recommended follow-up every 6 weeks to 3 months in years 1 and 2 after diagnosis, every 2 to 4 months in years 3 and 4, every 6 months in years 5 to 10, and every 6 to 12 months thereafter. Each visit should include a history, a physical examination, and a chest radiograph. Additional radiographs of the primary tumor site are recommended every 4 months until the end of year 4. Late metastases can occur beyond 10 years after diagnosis and there is no universally accepted stopping point for tumor surveillance.10 Although the differential diagnosis consisted of benign lesions, which would require simple excision, this case stresses the importance of a histologic diagnosis. Furthermore, the clinician should be able to differentiate between conventional intraosseous osteosarcomas and surface osteosarcomas. Treatment and follow-up should be similar to current management of intraosseous osteosarcomas; however, the clinician should be aware that literature on head and neck surface osteosarcoma is limited and more research is needed.
References 1. Geschickter CF, Copeland MM: Parosteal osteoma of bone: A new entity. Ann Surg 133:790, 1951 2. Okada K, Unni KK, Swee RG, et al: High grade surface osteosarcoma: A clinicopathologic study of 46 cases. Cancer 85:1044, 1999 3. Wold LE, Unni KK, Beabout JW, et al: High-grade surface osteosarcomas. Am J Surg Pathol 8:181, 1984 4. Minic AJ: Periosteal osteosarcoma of the mandible. Int J Oral Maxillofac Surg 24:226, 1995 5. Dwinnell LA, Dahlin DC, Ghormley RK: Parosteal (juxtacortical) osteogenic sarcoma. J Bone Joint Surg Am 36:732, 1954 6. Unni KK, Dahlin DC, Beabout JW: Parosteal osteogenic sarcoma. Cancer 37:2466, 1976 7. Klein MJ, Siegal GP: Osteosarcoma anatomic and histologic variants. Am J Clin Pathol 125:555, 2006 8. Yoon JH, Yook JI, Kim HJ, et al: Periosteal osteosarcoma of the mandible. J Oral Maxillofac Surg 63:699, 2005 9. Bertoni F, Dallera P, Bacchini P, et al: The Istituto Rizzoli-Beretta experience with osteosarcoma of the jaw. Cancer 68:1999, 1991 10. Bielack S, Carrle D, Casali PG: Osteosarcoma. ESMO clinical recommendations for diagnosis, treatment and follow-up. Ann Oncol 20:137, 2009
A 50-year-old woman presented to the oral and maxillofacial surgery (OMS) clinic with the chief complaint of ‘ having a growth on my gums.’’ She reported noting something growing in the midportion of the gingiva of the right mandible and perceiving a small lump for approximately 6 months. During the 2 weeks before presentation, she noticed that the lump had rapidly increased in size. She presented to the emergency department at the Virginia Commonwealth University Medical Center (Richmond, VA), where they noted a 1-cm nodular growth on the gingiva between the right mandibular first molar and second bicuspid. Based on the innocuous appearance of the lesion and the lack of any major symptoms, she was given an appointment in the OMS clinic for 2 weeks later.
On presentation to the OMS clinic, her medical history was noted to be important for type 2 diabetes mellitus, arthritis, gastroesophageal reflux disease, and depression. Her surgical history included tonsillectomy, left knee surgery, bilateral carpal tunnel repair, left wrist ganglion cyst removal, bunionectomy, and tubal ligation. She had an allergy to trazodone. She had no noteworthy social history other than a 10 pack-year history of smoking. Her family history was noteworthy for cancer, hypertension, diabetes, and asthma. She had a negative review of systems. The extraoral examination was unremarkable. Cranial nerves II to XII were grossly intact, with no evidence of paresthesia or neurosensory deficit in the distribution of the trigeminal nerve. Intraoral examination showed a 1.5- 1-cm, soft, nonulcerated, reddish, exophytic lesion on the interdental papilla between the right mandibular first molar and second bicuspid (Fig 1). The lesion was not sensitive to palpation and there was no expansion of the mandible. Radiographically, there was evidence of moderate horizontal interdental bone loss. However, there was no evidence of any radiolucent or radiopaque changes within the bone, and there was no evidence of root resorption or tooth displacement (Figs 2, 3). Based on the clinical presentation, the differential diagnosis included pyogenic granuloma, peripheral ossifying fibroma, and peripheral giant cell lesion.
Received from the Department of Oral and Maxillofacial Surgery,
North 11th Street, Box 980566, Richmond, VA 23298-0566;
Virginia Commonwealth University Medical Center, Richmond, VA.
e-mail: [email protected]
REPORT OF CASE
*Chief Resident.
Received August 14 2014
yProfessor and Residency Program Director.
Accepted November 10 2014
Address correspondence and reprint requests to Dr Meyliker:
Ó 2015 American Association of Oral and Maxillofacial Surgeons
Department of Oral and Maxillofacial Surgery, Virginia Common-
0278-2391/14/01717-0
wealth University Medical Center, Wood Memorial Building, 521
http://dx.doi.org/10.1016/j.joms.2014.11.010
787.e1
787.e2
PERIOSTEAL OSTEOSARCOMA OF THE MANDIBLE
lesion, the underlying bone was examined and deemed unremarkable. The specimen was sent for histologic examination. The biopsy examination disclosed a well-differentiated chondroblastic osteosarcoma, a diagnosis normally reserved for intraosseous osteosarcomas. After obtaining a negative head and neck CT scan and chest radiograph, this case was reviewed at the tumor board and the diagnosis was changed to a surface osteosarcoma. In this case, excision of the lesion and overlying hard and soft tissues with clinical margins of 1.5 cm was used, which ultimately resulted in a segmental mandibulectomy. The final specimen confirmed the diagnosis of surface osteosarcoma consistent with a periosteal osteosarcoma (Figs 4, 5). The patient was followed immediately after surgery with serial clinical examinations every 2 weeks for the first 2 months and subsequently with serial panoramic radiographs, chest radiograph, and clinical examination every 3 months. There was no recurrence or metastasis after 24 months.
Discussion
FIGURE 1. Initial presentation of soft tissue mass between the right mandibular first molar and second bicuspid. Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
Owing to the benign nature of the differential diagnosis, the decision was made to perform an excisional biopsy under local anesthesia. After excision of the
Juxtacortical osteosarcomas (also known as surface osteosarcomas) were first described in the orthopedic literature by Geschickter and Copeland1 in 1951. They are regarded to have a better prognosis and lower metastatic rate than intraosseous osteosarcomas. These lesions can be divided into parosteal, periosteal, and high-grade surface osteosarcomas.2-4 Parosteal osteosarcomas are considered to have a low rate of malignancy.5 Histologically, they consist of streams of bone trabeculae that are irregularly shaped and usually are covered with a single layer of
FIGURE 2. Panoramic radiograph at initial presentation. Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
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MEYLIKER AND STRAUSS
FIGURE 3. Computed tomogram (axial view, bone window) showing a cortical plate without any erosion, no osteolytic changes, and no extracortical bone formation (white arrow). Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
osteoblasts or spindle cells.6 In addition, the intertrabecular spaces of parosteal osteosarcomas contain cellular fibrous tissue.7 In contrast, periosteal osteosarcomas are classically an intermediate grade between that of the usual low-grade parosteal osteosarcoma and that of the conventional higher-grade osteosarcoma.7 Histologically, it contains radiating spicules of
FIGURE 4. Low-power excisional biopsy. This tumor arises from the surface of the mandible with superficial invasion into the cortical bone (hematoxylin and eosin stain; magnification, 100). Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
bone perpendicular to the long axis of the bone with malignant-appearing osteoid in the central portions.2 High-grade surface osteosarcomas should be differentiated from parosteal and periosteal osteosarcomas because they are associated with a poorer prognosis and are more likely to metastasize. Although they arise from the surface, the histologic findings are more typical of conventional osteosarcomas.2 Previous case reports of head and neck surface osteosarcomas have shown radiographic osseous changes on CT scans or panoramic radiographs.8,9 However, the present case was unusual in that it did not have any radiographic evidence of bony abnormality, although it histologically showed bony invasion (Fig 5). The ideal treatment of surface osteosarcomas is unclear owing to the limited number of cases reported to date. The current treatment has essentially been extrapolated from evidence from orthopedic surface osteosarcomas or intraosseous head and neck osteosarcomas. Because these 2 entities are considered more aggressive than head and neck surface osteosarcomas, more conservative resection margins might be appropriate. Unlike squamous cell carcinomas, head and neck osteosarcomas do not require lymph node dissection because they typically metastasize by tumor emboli into the blood stream, most commonly into the lungs. It is important to rule out lung metastasis at the time of diagnosis and provide continuous follow-up with serial chest radiographs. The European Medical Society of
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PERIOSTEAL OSTEOSARCOMA OF THE MANDIBLE
FIGURE 5. Tumor morphology is similar to that of a chondromyxoid fibroma-like osteosarcoma with malignant cells (arrow) producing osteoid (hematoxylin and eosin stain; magnification, 200). Meyliker and Strauss. Periosteal Osteosarcoma of the Mandible. J Oral Maxillofac Surg 2015.
Oncology has recommended follow-up every 6 weeks to 3 months in years 1 and 2 after diagnosis, every 2 to 4 months in years 3 and 4, every 6 months in years 5 to 10, and every 6 to 12 months thereafter. Each visit should include a history, a physical examination, and a chest radiograph. Additional radiographs of the primary tumor site are recommended every 4 months until the end of year 4. Late metastases can occur beyond 10 years after diagnosis and there is no universally accepted stopping point for tumor surveillance.10 Although the differential diagnosis consisted of benign lesions, which would require simple excision, this case stresses the importance of a histologic diagnosis. Furthermore, the clinician should be able to differentiate between conventional intraosseous osteosarcomas and surface osteosarcomas. Treatment and follow-up should be similar to current management of intraosseous osteosarcomas; however, the clinician should be aware that literature on head and neck surface osteosarcoma is limited and more research is needed.
References 1. Geschickter CF, Copeland MM: Parosteal osteoma of bone: A new entity. Ann Surg 133:790, 1951 2. Okada K, Unni KK, Swee RG, et al: High grade surface osteosarcoma: A clinicopathologic study of 46 cases. Cancer 85:1044, 1999 3. Wold LE, Unni KK, Beabout JW, et al: High-grade surface osteosarcomas. Am J Surg Pathol 8:181, 1984 4. Minic AJ: Periosteal osteosarcoma of the mandible. Int J Oral Maxillofac Surg 24:226, 1995 5. Dwinnell LA, Dahlin DC, Ghormley RK: Parosteal (juxtacortical) osteogenic sarcoma. J Bone Joint Surg Am 36:732, 1954 6. Unni KK, Dahlin DC, Beabout JW: Parosteal osteogenic sarcoma. Cancer 37:2466, 1976 7. Klein MJ, Siegal GP: Osteosarcoma anatomic and histologic variants. Am J Clin Pathol 125:555, 2006 8. Yoon JH, Yook JI, Kim HJ, et al: Periosteal osteosarcoma of the mandible. J Oral Maxillofac Surg 63:699, 2005 9. Bertoni F, Dallera P, Bacchini P, et al: The Istituto Rizzoli-Beretta experience with osteosarcoma of the jaw. Cancer 68:1999, 1991 10. Bielack S, Carrle D, Casali PG: Osteosarcoma. ESMO clinical recommendations for diagnosis, treatment and follow-up. Ann Oncol 20:137, 2009
