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SQUAMOUS CELL CARCINOMA AS OSTEOMYELITIS

J Oral Maxillofac 46X118-1122.

Surg

1990

Invasive Squamous Cell Carcinoma Mandible Presenting as a Chronic Osteomyelitis:

of the

Report of a Case PATRICK J. VEZEAU, DDS,* GERARD F. KOORBUSCH, AND MICHAEL FINKELSTEIN, DDS, MS+ A systematic approach for the diagnosis of oral squamous cell carcinoma is important because of the poor overall 5-year survival rate.’ Patients with chronic swelling of the head and neck, even in the presence of pain or drainage, may have a malignant neoplasm.2,3 This is particularly germane when appropriate treatment for a presumed infection (including culture-guided antibiotic therapy) elicits limited or no clinical improvement. This article describes a case of invasive squamous cell carcinoma of the mandible that presented with features suggestive of chronic osteomyelitis. Report of a Case The patient, a 41-year-old white man, was seen on consultation from the medical psychiatry unit for right-sided facial swelling and dysphagia. Review of the patient’s record showed admission 9 days previously after an apparent alcohol-related fall from a third-story construction site. The patient had sustained left radius and left tibial plateau fractures that had been treated with closed reduction and immobilization. Additionally, the patient had been seen at the University College of Dentistry 1 month previously, where a noncomplicated extraction of the mandibular right second molar was performed after endodontic therapy did not decrease pain and swelling in the region. A panoramic radiograph taken immediately before the ex-

Received from The University of Iowa Hospitals and Clinics, Iowa City, IA. * Resident, Division of Oral and Maxillofacial Surgery, - . Department of Hospital Dentistry. ‘t Assistant Professor. Division of Oral and Maxillofacial Surgery, Department of Hospital Dentistry. $ Associate Professor, Department of Oral Pathology/Oral Diagnosis. Address correspondence and reprint requests to Dr Koorbusch: Division of Oral and Maxillofacial Surgery, Department of Hospital Dentistry, The University of Iowa Hospitals and Clinics, E202 General Hospital, Iowa City, IA 52242. 0 1990 American

Association

geons 027%2193/90/481

O-0020$3.00/0

of Oral

and Maxillofacial

Sur-

DDS, MBA,t

traction demonstrated a small periapical radiolucency without other osseous changes (Fig 1). The patient had a long history of tobacco and ethanol abuse and had sustained severe ethanol withdrawal symptoms during his current hospitalization. This resulted in transfer to the medical psychiatry unit, where large doses of intravenous benzodiazepines and barbiturates were used to treat delirium tremens. Physical examination showed a thin white man who appeared older than his stated age. There was a discrete, firm, tender nonerythematous swelling on the buccal aspect of the right mandible. No exudate or fluctuance were noted, and the recent extraction site was well healed. Vital signs were stable, and review of laboratory values were as follows: white blood count 8,400lp.L without significant left shift, red blood count 3,500,OOO pL, hemoglobin concentration 10.7 g/dL. hematocrit 34%. blood urea nitrogen 9 mg/dL, serum protein 6.0 g/dL, albumin 3.3 g/dL, alkaline phosphatase 354 IU/L, amylase 76 U/L, lipase 123 U/L, lactic dehydrogenase 386 III/L. Radiographs of the chest were negative, the electrocardiogram was normal, and the human immunodeficiency virus and hepatitis A/B antibody screens were negative. A panoramic radiograph demonstrated that the recent extraction site was without obvious pathologic change. Therapy was initiated with oral penicillin, 500 mg four times daily, and 1 day later the patient was brought to the oral and maxillofacial surgery clinic, where aspiration of the right mandibular buccal and submandibular spaces was nonproductive. An extraoral approach to the right submandibular region and blunt exploration of this area produced a small amount of purulent exudate, which grew coagulase-negative staphylococci on aerobic culture. A Penrose drain was placed, and intravenous penicillin, 2 million units, was administered every 4 hours along with twice daily drain irrigations. Two days later, the swelling had spread to the right submandibular and sublingual spaces, and the first molar was exquisitely tender to percussion. The tooth was extracted and the involved fascial spaces were reexplored. A submentalto-submandicular Penrose drain was placed with little resultant drainage. Cultures again grew coagulase-negative staphylococci. Intravenous antibiotics, irrigation of the drain, and extraoral heat applications resulted in a slow decrease in the patient’s swelling and discomfort. Penicillin was discontinued 3 days later and intravenous clindamycin, 300 mg three times daily, was begun. The drains were nonproductive and were subsequently removed

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VEZEAU ET AL

FIGURE 1. Radiograph of the mandibular right molar region prior to extraction of the second molar.

FIGURE 3. Radiograph showing of osteolytic bony change in region of first molar socket.

with normal healing of the incision sites. Computerized tomography (CT) performed after drain removal demonstrated a homogeneous mass extending from the medial surface of the right mandibular body to the right submandibular space, interpreted to be consistent with a hematoma (Fig 2). By the 3rd postoperative week, the swelling remained brawny and there was limited mandibular opening. Tuberculosis and mumps screens were negative. A repeated panoramic radiograph 1 week later showed an irregular l-cm radiolucency inferior to the first-molar extraction site, with loss of trabecular bone posterior to this area (Fig 3). A presumptive diagnosis of chronic osteomyelitis was made. A repeat of the CT scan exhibited osteolysis of the right mandibular body and ramus, with resorption of the buccal and lingual bony cortices (Fig 4). A technicium-99 bone scan demonstrated increased uptake from the right angle to the left parasymphysis region, consistent with osteomyelitis (Fig 5).

The patient was brought to the operating room for a planned extraoral debridement. Arch bars and maxillomandibular fixation were applied, and three transcutaneous biphase appliance pins were placed. A generous elliptical skin wedge was excised from the right mandibular area, and blunt dissection to the mandibular body showed markedly fibrotic, necrotic, and avascular tissue. The mandibular buccal cortical plate was firm, but had a motheaten appearance. The right facial vessels were fibrotic. Microscopic examination of frozen sections was consistent with squamous cell carcinoma. A Penrose drain was placed and the biphase pins and maxillomandibular fixation were removed. The patient’s immediate postoperative course was unremarkable, and 2 days later he was taken to the operating room by the otolaryngology service who performed a tracheostomy. Pan-endoscopic examination was at-

FIGURE 2. Soft-tissue enhanced coronal CT showing homog-

FIGURE 4. Bone-enhanced mandibular body.

enous soft-tissue swelling.

CT showing osteolysis

of right

SQUAMOUS CELL CARCINOMA AS OSTEOMYELITIS

FIGURE 5. Frontal (A) and right lateral (B) technetium-99 bone scan of involved region of the right mandible.

tempted, but abandoned because of the limited range of mandibular motion. Examination of the oropharynx and nasopharynx showed only some false vocal cord and arytenoid edema, without evidence of tumor. A fullmouth extraction with alveoloplasty and primary closure was done, followed by a resection of the right mandible, floor of mouth, inferior portion of the parotid gland, and radical neck dissection. Closure of the resultant surgical defect was done via a pectoralis major cervico-facial advancement flap. Postoperatively, the patient made good progress, and a 2-month course of external beam radiation to the tumor bed and neck was initiated. The radiation dose incident to the tumor bed was 6,480 cGy, with additional exposure to the posterior cervical lymph nodes and anterio-inferior neck. The patient was last examined 3 months postoperatively, after he had sustained a fall with head injuries. CT showed significant cortical atrophy without evidence of intracranial hematoma. One month later, the patient expired at a local hospital, reportedly as a result of massive infection of the surgical site. Evidence of lung metastasis was noted on chest radiographs. An autopsy was not performed.

peripheral osteoclastic activity near regions of invading squamous cell tumor, often with a dense lymphocytic infiltrate in the intervening connective tissue. Several lymph nodes were almost entirely replaced by tumor. The final diagnosis was poorly differentiated invasive squamous cell carcinoma. No definite primary site was indicated, although the mandible was thought to be the most likely site. Tumor staging was listed as T,N,M,.

Discussion The origin of mandibular carcinomas traosseous, extension from an adjacent tumor, or metastatic. Mucosal changes with squamous cell carcinoma were not multiple examinations of this patient.

can be insoft-tissue associated evident on Limited

HISTOLOGIC FINDINGS Tumor was found in the mandibular body; submandibular, sublingual, and parotid glands, extending to the epidermis in one region; and in the mucosa of the lateral pharyngeal wall. Lymph node metastasis was noted in one superficial and two superior deep cerical lymph nodes. The tumor consisted of nests and cords of large, atypical cells with pale eosinophilic cytoplasm, and pleomorphic, hyperchromatic nuclei with numerous abnormal mitotic figures (Fig 6). Larger nests exhibited central necrosis (Fig 7). Keratin formation was present in some areas. The connective tissue stroma exhibited foci of dense lymphocytic infiltrate, with a prominent desmoplastic response. The medullary mandibular bone showed marked

FIGURE 6. Photomicrograph of primary tumor. Nuclei of tumor cells are variable in size, shape, and staining characteristics (hematoxylin-eosin, original magnification x 160).

VEZEAU ET AL

FIGURE 7. Photomicrograph of primary tumor. An island of squamous cell carcinoma demonstrates necrosis of tumor cells in its central portion (hematoxylin-eosin, original magnification x 25).

pharyngoscopy at the time of tumor resection failed to elicit mucosal change above the level of the false vocal cords. Nevertheless, the extension of the tumor as examined microscopically was from lateral pharyngeal wall to epidermis; therefore, mucosal, skin, or salivary gland origin cannot be definitively ruled out. Similarly, although repeated chest radiographs were negative, and the patient did not demonstrate any signs or symptoms of another primary tumor site, an aero-digestive tract origin remains a possibility. Appropriate workup for such a lesion may include upper and lower gastrointestinal tract examination, magnetic resonance imaging, CT, liver/spleen scan, and nuclear medicine studies. In this case, origin in the mandible seems probable because of the chronicity of the tumor, local invasion, and ipsilateral lymph node involvement. Considering the lack of clinically apparent mucosal lesions and major salivary gland symptoms, an intraosseous primary site must be considered a strong possibility. Primary intraosseous carcinoma of the jaws (PIOC), as defined by the World Health Organization (WHO), is a squamous cell carcinoma arising within the jaws, presumably from odontogenic epithelial remnants.4 The WHO definition excludes those tumors having an initial connection with oral mucosa, or arising in an odontogenic tumor or in a cyst wall. Elzay’s modification of this classificatiot3 groups all intraosseous nonmetastatic squamous cell carcinomas together as PIOC, then subclassifies them as to histologic origin (ie, arising in ameloblastoma, odontogenic cyst, or de novo). His critical review of the literature for PIOC found 5 of 12 reported lesions exhibiting keratin formation, with palisading found in 7 lesions. Presence of

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keratin did not correlate with recurrence, metastasis, or survival. This reported case might qualify for inclusion in the modified PIOC classification, because the small radiolucency originally seen at the apex of the second molar could represent a tumor primary site (arising de novo, ex-ameloblastoma, or ex-odontogenic cyst). Signs and symptoms of chronic osteomyelitis and malignancy of the jaws can often be similar, posing a challenge in diagnosis for the clinician.6-‘1 Both entities can cause localized, prolonged swelling, trismus, and pain. A history of one or more tooth extractions is common. The partial remission of this patient’s symptoms with extractions, incision and drainage, and antibiotic therapy further obscured the definitive diagnosis. Radiographic changes associated with osteomyelitis and malignant disease may be similar in appearance characterized by a motheaten appearance on plane and panoramic views and cortical bone destruction on plane films and CT. However, malignancies are typically more localized, eroding adjacent teeth and the contents of the inferior alveolar neurovascular canal.12 Technicium-99 bone scintigraphy has been useful for detecting regions of osteoblastic activity that may be associated with both chronic osteomyelitis and malignancies of bone.‘2-‘5 In both processes, the extent of technicium uptake generally exceeds the radiographic extent of the lesion, although the size of the actual lesion is smaller than the image produced by the scan. However, a bone scan may demonstrate poor uptake (a “cold” spot) if the regional blood supply is compromised by thrombosis or periosteal necrosis. This may be indicative of rapid, aggressive growth, Therefore, differentiation of malignancy from chronic osteomyelitis by conventional radiography or bone scanning may be difficult because of the similar appearance of such lesions. Gallium-67 radionucleide studies are thought to be beneficial for identifying regions of inflammation, as gallium uptake sites are associated with leukocytic uptake and with several proteins found in regions with inflammation.“j Gallium scans have been shown to identify regions of acute head and neck infection, with somewhat less correlation in chronic infection.” However, gallium uptake is relatively nonspecific, and also occurs in nasal and oral mucosa, lacrimal and salivary glands, spleen, marrow, liver, and regions of inflammation not associated with infection. Additionally, poor sensitivity has been noted for head and neck tumors. “-*’ Indium- l-labeled leukocytes have been reported to be more specific markers of regions of inflammation, with less accumulation in other regions. How-

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SQUAMOUS CELL CARCINOMA AS OSTEOMYELITIS

ever, indium-labeled leukocytes have been thought to be no more specific than gallium for identification of chronic osteomyelitis, car although some studies have demonstrated indium uptake with some malignancies. 22323The use of technicium scintigraphy and gallium scanning together in maxillofacial lesions has been shown to be of value for differentiation of chronic osteomyelitis from other processes. l4 A negative Te 99 scan mitigates against malignancy or osteomyelitis, whereas a positive technicium and gallium scan (or indium-labeled leukocyte) may be suggestive of osteomyelitis. A positive technicium scan with an equivocal or negative gallium/indium scan may indicate a neoplasm, infection with little inflammation, or healing bone. The use of gallium scanning or labeled leukocytes for diagnostic imaging of this case may have been of diagnostic benefit, although acute inflammation associated with necrosis and purulence at the tumor site may have caused a positive gallium or indium uptake, further obscuring the diagnosis. Clinical differential diagnosis of lesions of the maxillofacial region that present with pain may be difficult, with diagnostic confirmation not possible until microscopic examination is performed. This patient had significant risk factors for the development of squamous cell carcinoma; however, clinical examination failed to show a mucosal lesion in the affected region. The clinical and radiographic appearance suggested chronic osteomyelitis, with some response to therapy directed towards this presumptive diagnosis. Management of similar lesions should include evaluation for malignancy to avoid a delay in diagnosis. Summary A case of invasive squamous cell carcinoma of the mandible initially thought to be chronic osteomyelitis is reviewed. Discussion of the origin of the tumor and several imaging techniques used for differentiating maxillofacial infection from neoplasm are discussed. The difftculty of diagnosis of chronic painful maxillofacial lesions illustrates the need for a high degree of suspicion concerning any inflammatory lesion not responding to appropriate therapy. Accurate diagnosis and treatment of such lesions most often necessitates microscopic examination .

References 1. Komblut AD, Wixson D, Kirchner PT: The clinical diagnosis of oral malignant tumors. Otolaryngol Clin North Am 12:57, 1979 2. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology (4th ed). Philadelphia, Saunders, pp 113-I IS, 1983 3. Sellars SL: Epidemology of oral cancer. Otolaryngol Clin North Am 12:45, 1979 4. Pindborg JJ, Kramer IRH, Torlon H: Histologic Typing of Odontogenic Tumors, Jaw Cysts, and Allied Lesions. Geneva, World Health Organization, pp 35-36, 1972 5. Elzay RP: Primary intraosseous carcinoma of the jaw. Review and update of odontogenic carcinomas. Oral Surg 54:299, 1982 6. Rubin RL, Maloney PL, Doku HC: Squamous cell carcinoma of the floor of the mouth with extensive mandibular involvement. Oral Surg 39:184, 1975 7. McGowan RH: Primarv Intra-alveolar carcinoma: A difftcult diagnosis. Br J OralSurg 18:259, 1980 8. Lindquist C, Teppo L: Primary intraosseous carcinoma of the mandible. Int J Oral Maxillofac Surg 15:209, 1986 9. Rushkin JD, Cohen DM, Davis LF: Primary intraosseous carcinoma: Report of two cases. J Oral Maxillofac Surg 46:425, 1988 10. Van Wyk CW, Padayachee A, Nortje CA, et al: Primary intraosseous carcinoma involving the anterior mandible. Br J Oral Maxillofac Surg 25:427, 1987 11. Waldron CA, Mustoe TA: Primary intraosseous carcinoma of the mandible with probable origin in an odontogenic cyst. Oral Surg 671716, 1984 12. Hofer B, Hardt N, Voeyeli E, et al: A diagnostic approach to lvtic lesions of the mandible. Skeletal Radio1 14:164. 1985 13. Robinson CB , Higginbotham-Ford EA: Determination of sequestrum activity by SPECT with CT correlation in chronic osteomyelitis of the head and neck. J Otolaryngol 15:279, 1986 14. Shafer RB, Marlette JM, Browne GA, et al: The role of Tc-99m phosphate complexes and gallium-67 in the diagnosis and management of maxillofacial disease: Concise communication. J Nut Med 22:8, 1981 15. Al-Sheikh W, Sfakidnakis G, Mnaymneh W, et al: Subacute and chronic bone infections: Diagnosis using In-l 11, Ga67, and Te-99m MDP bone scintigraphy, and radiography. Radiology 155:501, 1985 16. Neumann RD, Hoffer PB: Gallium for Detection of Malignant Disease, in Diagnostic Nuclear Medicine (2nd ed). Baltimore, Williams & Wilkins, 1982, pp 1076-1077 17. Neumann RD. Hoffer PB: Gallium and Infection in Diagnostic Nuclear Medicine (2nd ed). Baltimore, Williams & Wilkins, pp 1111-1118 18. Teats CD, Preston DF, Boyd CM: Gallium-67 citrate imaging in head and neck tumors. Report of a cooperative group. J Nucl Med 21:622, 1980 19. Teats CD, Bray ST, Williamson BRJ: Tumor Detection with Ga-67 citrate: A literature survev_ (1970-19751. Clin Nucl Med 3:456 20. Hoffer P: Status of gallium-67 in tumor detection. J Nucl Med 21:394, 1980 2 1. Coleman RE: Radiolabeled Leukocytes, Nuclear Medicine Annual. New York, Rakem Press, pp 119-137, 1982 22. Lamki LM. Kas LP. Mavnie TP: Localization of indium- 111 leukocytes in noninfected neoplasms. J Nucl Med 29: 1921, 1988 23. Schmidt KG, Rasmussen JW, Wedebye IM, et al: Accumulation of indium-11 1 labeled granulocytes in malignant tumors. J Nucl Med 29:479, 1988

Invasive squamous cell carcinoma of the mandible presenting as a chronic osteomyelitis: report of a case.

A case of invasive squamous cell carcinoma of the mandible initially thought to be chronic osteomyelitis is reviewed. Discussion of the origin of the ...
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