Benign osteoblastoma of the mandible: Report of a case

Benign osteoblastoma of the mandible: Report of a case

743 HAUG ET AL ported. Despite the advantages of using autogenous calvarial bone grafts for craniofacial reconstructive surgery, the potential for s...

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ported. Despite the advantages of using autogenous calvarial bone grafts for craniofacial reconstructive surgery, the potential for serious complications related to graft harvest must be appreciated. Surgeons involved in calvarial graft harvest must be familiar with cranial surgical technique, and should be capable of managing intraoperative complications at the donor site. References 1. Cinberg JZ, Rosenbaum FA, Lowrie C, et al: Calvarial grafts for midface rehabilitation. Arch Otolaryngol 111:434,1985 2. Edwards MSB, Ousterhout DK: Autogeneic skull bone grafts to reconstruct large or complex skull defects in children and adolescents. Neurosurgery 20:273, 1987 3. Lauritzen C, Lilja J, Vallfors B: The craniofacial approach to trauma. Ann Plast Surg 17:503, 1986 4. Petroff MA, Burgess LPA, Anonsen CK, et al: Cranial bone grafts for posttraumatic facial defects. Laryngoscope 97:1249, 1987

J Oral Maxillofac 48:743-748,

5. Tessier P: Autogenous bone grafts taken from the calvarium for facial and cranial applications. Clin Plast Surg 9:531, 1982 6. Kapp JP: Nonseptic venous occlusive disease, in Wilkins RH, Rengachary SS (eds): Neurosurgery. New York, NY McGraw-Hill, 1985, p 1300 7. Donaghy RMP: Surgical management of lesions of the dural venous sinuses, in Schnidek HH, Sweet WH (eds): Operative Neurosurgical Techniques: Indications, Methods, and Results. Philadelphia, PA, Grune & Stratton, 1982. p 729 8. Browder J, Browder A, Kaplan HA: The venous sinuses of the cerebral dura mater. Arch Neurol 26:175, 1972 9. Maxwell RE, Chou SN: Parasagittal and falx meningiomas, in Schnidek HH, Sweet WH (eds): Operative Neurosurgical Techniques: Indications, Methods, and Results. Phildelphia, PA, Grune & Stratton, 1982, p 503 10. Warwick R. Williams PL: Grav’s Anatomv led 351. Philadelphia, PA, Saunders, 1973: p 693 ’ 11. Ojemann RG: Meningiomas: Clinical features and surgical management, in Wilkins RH, Rengachary SS (eds): Neurosurgery. New York, McGraw-Hill, 1985, p 635 12. Wilkinson HA: Autogeneic skull bone grafts. Neurosurgery 21:760, 1987 (letter)



Benign Osteoblastoma

of the Mandible:

Report of a Case RICHARD H. HAUG, DDS,* CHRISTOPHER HAUER, MD,? ANTHONY J. DE CAMILLO, DDS,* AND MARIA ARANETA, MD-f Benign osteoblastoma is a rare tumor of bone representing less than 1% of all tumors of the maxillofacial region. There is a slight predilection for the mandible in the 30 documented osteoblastomas of the jaws. More frequent sites for this tumor are the vertebral column, sacrum, long bones, and calvarium. A small predilection in males exists. The age of occurrence ranges from 5 to 37 years, with an average of 16.5. Clinically, patients have pain and swelling. The tumor contains a well-vascularized, osteoblastic connective tissue stroma. Osteoclasts may also be present. Osteoid with varying degrees of calcification, as well as immature bone, is noted. I-5 Received from Cleveland Metropolitan General Hospital and the Case Western Reserve University, Cleveland, OH. * Assistant Professor of Surgery. t Assistant Professor of Pathology. $ Chief Resident, Division of Oral and Maxillofacial Surgery, Cleveland Metropolitan General Hospital. Address correspondence and reprint requests to Dr Haug: Department of Surgery, Cleveland Metropolitan General Hospital, 3395 Scranton Rd, Cleveland, OH 44109. 0 1990 geons




of Oral

and Maxillofacial


Because osteoblastoma has a pleomorphic histologic appearance, and can therefore present a diffrcult diagnostic problem, the incisional biopsy must contain representative areas of the lesion. This report discusses a case of benign osteoblastoma of the jaw which exemplifies the diagnostic dilemma that these tumors can pose to both the surgeon and pathologist. The differential diagnosis will be discussed from a clinical, radiologic, and histological perspective. Report of a Case A 3S-year-old white woman was seen in the oral and maxillofacial surgery clinic with a complaint of dull pain and swelling in the left side of her face. She related that she had had a large facial mass removed from the same site 4 years previously. The remainder of her history was noncontributory. Examination revealed a firm 4.0 X 4.0 cm painful mass in the left preauricular area (Fig 1). The patient had normal function of the facial musculature and no paresthesia, but did have trismus, with a vertical opening of 2.0 cm. A panoramic radiograph showed a well-demarcated radiolucent lesion of the left superior ramus, condyle, and coronoid process (Fig 2). A computed tomography (CT)


FIGURE 1. Frontal view of patient showing extensive facial deformity secondary to tumor expansion.

scan showed a well-defined,

variably calcified lesion approximately 3.5 cm in diameter involving the majority of the left ramus (Fig 3). A peripheral rim of calcification was noted. Review of the records of the patient’s surgical procedure 4 years previously indicated a preoperative biopsy diagnosed as chronic osteomyelitis. The postoperative histologic diagnosis was central giant cell granuloma with myxomatous changes. An incisional biopsy was performed under local anesthesia. A portion of the thin outer cortex was removed and a soft, grey, gritty material was removed from the center of the lesion. The histologic diagnosis was benign fibroosseous lesion. A second, more extensive incisional biopsy was performed, A diagnosis of benign fibroosseous lesion was again reported. The excision of the lesion was an extensive undertaking because of its size and proximity to the great vessels and base of the skull. A submandibular incision and hemicoronal incision, facilitated by a zygomatic arch osteotomy after the method described by Obwegeser, was performed.6 The specimen was removed with a l.O-cm free bony margin and one plane of soft tissue overlying the tumor. Reconstruction was performed immediately using a free rib graft.

FIGURE 2. Panoramic radiograph showing a well-demarcated radiolucent lesion of the left superior ramus, condyle, and coronoid process.


FIGURE 3. CT scan with contrast showing extent of tumor and its proximity to the great vessels.

Microscopic examination of the tumor revealed a thin fibro-osseous capsule surrounding a well-vascularized stroma containing osteoid tissue (Fig 4). Partially calcified trabeculae were also noted. Extensive osteoblastic activity was noted, with single strands of plump osteoblasts lining the trabeculae (Fig 5). Coarse woven and fine lamellar bone was also seen, as were osteoclasts and multinucleated giant cells (Fig 6). The histologic diagnosis was benign osteoblastoma.

Discussion Diagnosis of benign sive, as our case report

osteoblastoma can be eluillustrates. Preoperative and

postoperative histologic examination at the time of the first resection led to diagnoses of chronic osteomyelitis and giant cell granuloma, lesions with histologic similarities to benign osteoblastoma. These

FIGURE 4. Low-power photomicrograph showing tumor with libro-osseous capsule surrounding a well-vascularized stroma containing osteoid tissue. (Hematoxylin-eosin stain. Original magnification, X4.0)


FIGURE 5. Photomicrograph showing plump osteoblasts rimming trabeculae in single strands (arrowheads), osteoid, and osteoclasts within lacunae. (Hematoxylin-eosin stain. Original magnification, X40.)

erroneous diagnoses resulted in inadequate surgical treatment and subsequent recurrence. Moreover, despite examination of two separate preoperative specimens prior to the second resection, a diagnosis of benign osteoblastoma could not be made. A number of lesions of the jaw of both osseous and mensenchymal odontogenic origin can have similar overlapping demographic, radiographic, and histological characteristics (Table 1). Some of these lesions can have a vascular stroma, and show the presence of fibrous tissue in various stages of calcification; these features can also be seen in benign osteoblastoma. Adequate representative sections of the entire lesion must be submitted to assure adequate histologic diagnosis and thus guide the surgical management. Osteoblastoma and osteoid osteoma can be considered as almost identical tumors in terms of both their demographic and histological profiles. The essential gross difference between the lesions is size. Osteoid osteomas can be distinguished from benign osteoblastoma by clinical and radiographic examination, as they are usually less than 2.0 cm in diameter.‘33*7,8 Microscopically, osteoid osteoma contains a central nidus of bone, generally lacks giant cells, and is not as well vascularized as osteoblastoma. Osteogenic sarcoma is the most important condition to consider in the differential diagnosis. The radiograph will show a less well-demarcated lesion with some radiopacity. Histologically, osteogenic sarcoma contains atypical osteoblasts, with malignant bone formation, and may have a sarcomatous stroma as well as show anaplastic cartilage. These features distinguish it from osteoblastomas.2.7-9 Ossifying fibroma has a similar demographic, clinical, and radiographic appearance to osteoblastoma. Histologically the two lesions may be similar

745 because both can form immature woven bone. The ossifying fibroma, however, will be less vascular, more fibrous, and have less osteoblastic activity. 2,3,7,10 Condensing osteitis can be differentiated radiographically and histologically. It will most frequently be associated with roots of teeth. Sclerotic bone will be noted histologically.’ Bone in Paget’s disease is generally less vascular, with less osteoblastic activity. The abnormal, tilelike pattern of bone formation that characterizes Paget’s disease microscopically will distinguish it from benign osteoblastoma. Radiographically, Paget’s disease also shows greater calcification. Clinically, the serum alkaline phosphatase level will be elevated. Additional characteristics may be noted such as paresthesia, simian gait, and facies lionisis. The target population is generally older.’ Fibrous dysplasia will show multilocular radiolucencies in the early stages and a ground glass appearance in the more mature stages. Histologically, there will be osteoid within fibrous tissue which is composed of woven bone with fibrous stroma and C-shaped disoriented bony trabeculae, which in the face may have osteoblastic rimming. These features may cause confusion with ossifying fibroma. Ossifying fibroma, however, will have a higher degree of osteoblastic rimming of trabeculae.“*3*7 Chrondroblastoma is found in a younger population and rarely occurs in the jaw. The histopathologic appearance differs significantly in that polyhedral cells with a chondromatous matrix are present. The lesion may possess multinucleated giant cells but is devoid of any significant osteoblastic activity.7 Cementoblastoma is found usually in association

FIGURE 6. Well-vascularized fibrous stroma between trabeculae of varying degrees of calcification. Rimming of trabeculae by single strands of osteoblasts (arrowheads A), multinucleated giant cells (arrowheads B), and osteocytes within lacunae can be seen. (Hematoxylin-eosin stain. Original magnification, x 10.)



Radical excision

Conservative excision

Atypical osteoblasts, osteoid and bone; sarcomatous stroma with neoplastic osteoid, hyperchromatic nuclei; mitoses; anaplastic cartilage; giant cells Cellular fibrosis; irregular trabeculae, bone and osteoid; osteoblastic, osteoclastic, tibroblastic activity Sclerotic bone; fibrous pattern; osteoblastic and osteoclastic activity; mosaic bone Osteoblastic and osteoclastic activity; fibrous marrow with mosaic bone Irregular trabeculae; osteoid within fibrous tissue; woven bone within fibrous stroma

Poorly demarcated radiolucency with speckled radiopacity

Well-delineated radiolucency with radiopaque material Poorly demarcated radiopacity

Cotton wool radiopacity

Multiple locular radiolucencies; later, ground glass

Pain, swelling, paresthesia

Local swelling, vital teeth

Occasional pain or swelling Swelling; elevated alkaline phosphatase Local swelling

2nd-3rd decade; male predilection

3rd decade; female:male 211

Over 20 years

40 or older; no sex predilection 3rd decade; female:male 2:l

Mostly long bones, rare in jaws, mandible: maxilla 2: 1

Maxilla:mandible 2:l; rare outside jaws

Both jaws, usually

All bones; maxilla > mandible

Any bone; maxilla: mandible 2: 1

Ossifying sarcoma

Ossifying tibroma

Condensing osteitis

Paget’s disease

Fibrous dysplasia


Conservative excision

Highly vascular stroma; trabeculae rimmed with osteoblasts, osteoid and immature calcified bone

Circumscribed sclerotic lesion with central radiolucent nidus <2.0 cm

Swelling and severe local pain

Cosmetic recontouring







2nd-3rd decade; slight male predilection


Long bones most common, but affects all bones

rich is with giant

Osteoid osteoma

Highly vascular stroma, in osteoblasts; osteoid abundant and rimmed osteoblasts; may have cells

Well-circumscribed radiolucency; some radiopacities >2.0 cm

Swelling and pain


2nd-3rd decade; slight male predilection


Mostly vertebral column Rarely jaws


Benign osteoblastoma

Radiographic Appearance

Age and Sex



Differential Diagnosis of Benign Osteoblastoma of Jaws


Table 1.

Multilocular “soap bubble” radiolucency

Slow-growing swelling

Usually 1st or 2nd decade

1st or 2nd decade; male:female 2:1

Any age; no sexual predilection 2nd or 3rd decade; no sexual predilection

Vertebral column and long bones predominate

Uncommon, usually tooth-bearing areas of jaws; mandible most frequent location



Aneurysmal bone cyst

Giant cell granuloma

Odontogenic fibroma


Well demarcated, usually associated with a tooth

Slow-growing, local swelling

Young and middle-aged adults

Mandible greater than maxilla Unilocular or multilocular radiolucency

Firm, nontender, painless swelling

Slow and Ill-defined asymptomatic; radiolucent or pain, loculation paresthesia. loose teeth when rapid

Radiolucent with radiopaque foci

Local swelling

Well-demarcated areas of mottled, opaquebone fused to tooth

Cementifying fibroma

Local swelling

2nd or 3rd decade; male predilection

Cystic radiolucency

Found in assocation with tooth, usually molars

Local swelling


Ist, 2nd. 3rd decade; male:female 2:1

Long bones; rare in jaws


Local excision

Excision 1.0 cm border

Fibroblasts with occasional strands of odontogenic epithelium Abundant basophilic stroma with stellate cells

Prominent, plump spindle cell stroma; multinucleated giant cells, blood vessels


High recurrence





Osteoid proliferation, in avascular stroma; vascular channels with pools of blood; honeycombed vascular space, with fibrous connective tissue


Local excision

Fibroblastic stroma with foci of calcification



Tooth extraction

Conservative excision

Encapsulated mass of cementumlike tissue; osteocytes in lacunae; multinucleated giant cells, vascular connective tissue; uncalcified tissue at periphery

Polyhedral cells with chondromatous matrix; occasional multinucleated giant cells; sometimes associated with aneurysmal bone cyst



with teeth. The radiographic appearance shows mottled opaque bone fused to teeth. An encapsulated mass of cementumlike tissue differentiates this lesion histologically.3,7 Cementifying fibroma will possess demographic characteristics similar to the benign osteoblastoma. A fibroblastic stroma with a focus of cementoid tissue is present. No osteoblasts or bone are present.7 Odontogenic fibroma is a rare tumor of odontogenie mesenchyme. Radiographically, a wellcircumscribed mass will be found associated with roots or crowns of teeth. Histologically, fibroblasts are noted in great density, with occasional strands of odontogenic epithelium which distinguishes it from osteoblastoma. l1 Odontogenic myxoma is a rare mesenchymal tumor in the jaws of young people. It grows slowly and may displace teeth or stop their eruption. Multilocularity is noted radiographically. Histologically, an abundance of mucoid connective tissue with few stellate cells distinguishes this lesion from osteoblastoma.” Aneurysmal bone cyst is usually found in a younger age group than osteoblastoma. Nevertheless, radiographically and clinically it is similar. This lesion has a highly vascular stroma, with fibrous connective tissue and osteoid proliferation. The histologic hallmark that distinguishes aneurysmal bone cyst from benign osteoblastoma and other fibro-osseous lesions is honeycombed vascular spaces lined with connective tissue containing pools of blood.‘,” Giant cell granuloma, as with the 12 previous le-

J Oral Maxillofac


sions, has a similar demographic, radiographic, and clinical appearance as osteoblastoma. This lesion has large numbers of blood vessels and multinucleated giant cells. The histologic feature distinguishing it from osteoblastoma is the presence of plump spindle cells in the stroma, with giant cells intimately associated with blood vessels. Bone formation occurs predominantly at the periphery of the lesion. These lesions can also simulate osteogenic sarcoma and must be carefully examined.2*8-‘0 References 1. El-Mofty S , Refai H: Benign osteoblastoma of the maxilla. J Oral Maxillofac Sum 4760.62 1989 2. Kent JN, Castro HF, Girotti WR: Benign osteoblastoma of the maxilla. Oral Surg Oral Med Oral Path01 27:209, 1%9 3. Greer RO, Berman DN: Osteoblastoma of the jaws: Current concepts and differential diagnosis. J Oral Surg 36:304, 1978 4. Smith RA, Hansen LS, Resnick D, et al: Comparison of the osteoblastoma in gnathic and extragnathic sites. Oral Surg Oral Med Oral Path01 54:285, 1982 5. Greene GW, Natiella JR, Spring PN: Osteoid osteoma of the jaws. Oral Surg Oral Med Oral Path01 26342, 1968 6. Obwegeser HL: The temporal approach to the TMJ, the orbit, and retromaxillary-infracoronal region. Head Neck Surg 7:185, 1985 7. Farman AG, Nortje CJ, Grotepass F: Periosteal benign osteoblastoma of the mandible: Report of a case and review of the literature pertaining to benign osteoblastic neoplasms of the jaws. Br Oral Surg 14: 12, 1976 8. Marsh BW, Bonfglio M, Brady LP, et al: Benign osteoblastoma. J Bone Joint Surg 57A:l, 1975 9. Lichtenstein L: Benign osteoblastoma. Cancer 9:1044, 1956 10. Dahlin DC, Johnson EW: Giant, osteoid osteoma. J Bone Joint Surg 36A:559, 1954 11. Hoffman S, Jacoway JR, Knolls SO: Intraosseous and periosteal tumors of the jaws. Armed Forces Inst Path01 Second Series Fast 24:126, 1985



/so/a ted Granulocytic



Received from the University of Oviedo, Oviedo, Spain. * Associate Professor of Oral and Maxillofacial Surgery. t Head, Department of Oral and Maxillofacial Surgery. $ Associate Professor of Pathology. Address correspondence to Dr Rodriguez: Department of Oral and Maxillofacial Surgery, N.S. de Covadonga Hospital, Celestino Villamil s/n, 33005-Oviedo, Spain. 0 1990 American geons



of Oral and Maxillofacial



Granulocytic sarcoma (chloroma) is a localized tumor mass composed of immature cells of the granulocytic series.’ The lesion was first described by Bums in 1811.* In 1853, King3 used the term chloroma because the tumor often exhibited a greenish color that faded on exposure to the air, which was produced by the presence of myeloperoxidase (verdoperoxidase) in the tumor cells. The term granulocytic sarcoma (GS) seems more appropriate because the tumor is not always green, is