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Radiology Quiz Case 1: Diagnosis

Radiology Quiz Case 1: Diagnosis Diagnosis: Primary intraosseous odontogenic carcinoma (PIOC) Primary intraosseous odontogenic carcinoma is a rare squamous cell carcinoma that arises within the jaws and develops from remnants of odontogenic epithelium.1It is 1 of the 4 types of odontogenic carcinomas that arise within the jaws. In 1989, Waldron and Mustoe2created a widely accepted and frequently cited histologic classification for odontogenic carcinomas. View LargeDownload Most authors reserve the term PIOCfor squamous cell carcinomas of odontogenic origin that arise either de novo (type 3) or in association with a preexisting odontogenic cyst (type 1). The histologic features of PIOC are not pathognomonic, and a definitive diagnosis is often difficult; therefore, few cases have been reported in the literature. The diagnostic criteria for PIOC are (1) the absence of an initial connection with the overlying mucosa or skin and (2) exclusion of metastasis from a distant primary tumor by physical and radiographic examination during at least 6 months of follow-up.3-5To et al4reviewed the literature regarding metastasis to the jaw and concluded that (1) investigation of the primary tumor should include chest radiography and (2) the patient must receive at least 6 months of follow-up. Metastatic carcinoma of the mandible occurs mainly from the breast, thyroid, kidneys, and lungs.4 The mean age of patients with PIOC is 52.3 years (age range, 4-81 years). The male to female ratio is 2.5:1.0, and a background of alcohol or tobacco abuse is usually not present.5The total number of reported cases is difficult to determine owing to insufficient data to support the diagnosis in some published cases.3However, the total number of reported PIOC cases is approximately 134, including 82 cases of PIOC type 1 and 52 cases of PIOC type 3.6Both types are more frequently found in the mandible, with a predilection in the posterior region of the mandible, similar to benign tumors and odontogenic cysts,3but they have also been found in the maxilla. In their review of the world literature, Thomas et al5found that 77% of PIOCs were located in the posterior mandible, 8.5% in the anterior mandible, and 11.4% in the maxilla. The tumor typically presents as a painful swelling in the jaw area, but it may be asymptomatic and found on routine panoramic radiography, as in the present case. Clinically, homolateral positive neck nodes have been noted in 28% of patients at the time of diagnosis, with the majority located in the submandibular triangle.7Imaging has been found to be one of the most effective methods for detecting PIOCs8; however, they can present as radiolucent lesions with great variation, either unilocular or multilocular, with an ill-defined or well-defined but noncorticated border.6They are generally lytic, with evidence of destructive changes to the bone. It is important to note that even benign-appearing osseous lesions may contain small areas that indicate a malignant nature. The differential diagnosis of radiolucent lesions of the mandible includes carcinoma, osteosarcoma, invasive lesions of the oral mucosa, distant metastasis, periapical lesions, cysts of the globullomaxillary region, dentigerous cysts, primordial cysts, odontogenic keratocysts, and ameloblastoma. During radiographic evaluation of lesions of the jaw, malignant changes should be considered if a radiolucent area shows jagged or irregular margins with indentations and indistinct borders suggestive of invasion and destruction by tumor or if serial occlusal radiographs show rapid enlargement of the radiolucent area.9Panoramic radiography can be useful in cases of gross disease, but it is of limited value in evaluating margins and extension and invasion of the tumor. Computed tomography can reveal soft-tissue masses, aid in delineating the extent of the malignant lesion, and disclose invasion of adjacent anatomical regions.9However, its usefulness may be limited by the presence of intraoral metal prostheses.10 The treatment of choice for PIOC is surgery and/or postoperative radiation therapy.5,7However, there is no overall treatment consensus, as there are no comprehensive comparisons because of the rarity of the disease. Overall, PIOC portends a poor prognosis. Zwetyenga et al7recommend surgical treatment combined with postoperative radiotherapy for 2 reasons: (1) PIOC is similar to locally advanced squamous cell carcinoma of the oral mucosa, particularly T4 gum carcinoma, which often requires the same aggressive treatment; and (2) although the results of their comparison of survival according to the treatment modalities were not statistically significant, the trend that they analyzed favored radical surgery followed by postoperative radiotherapy. In their review, 47% of the patients were treated with surgery alone and 38% were treated with surgery plus radiotherapy. Neck dissection was performed in 69.7% of the patients, 43% of whom had histologic evidence of nodal disease. Patients undergoing radical surgery and postoperative radiotherapy had 2- and 3-year survival rates of 61.3% and 40.9%, respectively, whereas the remaining patients had rates of 59.7% and 31.3%. Thomas et al5found that overall survival rates were 75.7%, 62.1%, and 37.8% at 1, 2, and 3 years, respectively. However, their calculations did not separate the patients who were treated with surgery alone and those who were treated with surgery plus adjuvant radiation therapy. In our case, the findings of the original outpatient biopsy were inconclusive, showing either squamous cell carcinoma or mucoepidermoid carcinoma. The patient underwent a radical composite surgical resection in which a fibular osseocutaneous free-tissue transfer was used for reconstruction. Because of the 43% rate of occult metastasis found by Zwetyenga et al,7a selective neck dissection at ipsilateral levels 1 through 4 was performed. Gross pathologic examination revealed a 2.2-cm, firm, tan mass with a necrotic center that was entirely contained within the bone and no evidence of a cystic component. Histologic examination demonstrated a moderately well-differentiated squamous cell carcinoma (Figure 4) with no evidence of lymph node metastasis and negative surgical margins. Adjuvant radiotherapy was not recommended given the patient's pathologically negative nodal status, and there has been no evidence of recurrent disease to date. Figure 4. View LargeDownload Return to Quiz Case 1. References 1. Kramer IRHPindborg JJShear M International Histological Classification of Tumours: Histological Typing of Odontogenic Tumours. 2nd ed. Heidelberg, Germany Springer-Verlag1992; 2. Waldron CAMustoe TA Primary intraosseous carcinoma of the mandible with probable origin in an odontogenic cyst. Oral Surg Oral Med Oral Pathol 1989;67 (6) 716- 724PubMedGoogle ScholarCrossref 3. Suei YTanimoto KTaguchi AWada T Primary intraosseous carcinoma: review of the literature and diagnostic criteria. J Oral Maxillofac Surg 1994;52 (6) 580- 583PubMedGoogle ScholarCrossref 4. To EHWBrown JSAvery BSWard-Booth RP Primary intraosseous carcinoma of the jaws: three new cases and review of the literature. Br J Oral Maxillofac Surg 1991;29 (1) 19- 25PubMedGoogle ScholarCrossref 5. Thomas GPandey MMathew A et al. Primary intraosseous carcinoma of the jaw: pooled analysis of world literature and report of two new cases. Int J Oral Maxillofac Surg 2001;30 (4) 349- 355PubMedGoogle ScholarCrossref 6. Chaisuparat RColetti DKolokythas AOrd RANikitakis NG Primary intraosseous odontogenic carcinoma arising in an odontogenic cyst or de novo: a clinicopathologic study of six new cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101 (2) 194- 200PubMedGoogle ScholarCrossref 7. Zwetyenga NPinsolle JRivel J et al. Primary intraosseous carcinoma of the jaws. Arch Otolaryngol Head Neck Surg 2001;127 (7) 794- 797PubMedGoogle Scholar 8. Kaffe IArdekian LPeled MMachtey ELauffer D Radiological features of primary intra-osseous carcinoma of the jaws: analysis of the literature and report of a new case. Dentomaxillofac Radiol 1998;27 (4) 209- 214PubMedGoogle ScholarCrossref 9. Cavalcanti MGPVeltrini VCRuprecht AVincent SDRobinson RA Squamous-cell carcinoma arising from an odontogenic cyst—the importance of computed tomography in the diagnosis of malignancy. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100 (3) 365- 368PubMedGoogle ScholarCrossref 10. Lin YJChen CHWang WCChen YKLin LM et al. Primary intraosseous carcinoma of the mandible. Dentomaxillofac Radiol 2005;34 (2) 112- 116PubMedGoogle ScholarCrossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Otolaryngology - Head & Neck Surgery American Medical Association

Radiology Quiz Case 1: Diagnosis

Archives of Otolaryngology - Head & Neck Surgery , Volume 135 (2) – Feb 16, 2009

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Publisher
American Medical Association
Copyright
Copyright © 2009 American Medical Association. All Rights Reserved.
ISSN
0886-4470
eISSN
1538-361X
DOI
10.1001/archoto.2008.533-b
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Abstract

Diagnosis: Primary intraosseous odontogenic carcinoma (PIOC) Primary intraosseous odontogenic carcinoma is a rare squamous cell carcinoma that arises within the jaws and develops from remnants of odontogenic epithelium.1It is 1 of the 4 types of odontogenic carcinomas that arise within the jaws. In 1989, Waldron and Mustoe2created a widely accepted and frequently cited histologic classification for odontogenic carcinomas. View LargeDownload Most authors reserve the term PIOCfor squamous cell carcinomas of odontogenic origin that arise either de novo (type 3) or in association with a preexisting odontogenic cyst (type 1). The histologic features of PIOC are not pathognomonic, and a definitive diagnosis is often difficult; therefore, few cases have been reported in the literature. The diagnostic criteria for PIOC are (1) the absence of an initial connection with the overlying mucosa or skin and (2) exclusion of metastasis from a distant primary tumor by physical and radiographic examination during at least 6 months of follow-up.3-5To et al4reviewed the literature regarding metastasis to the jaw and concluded that (1) investigation of the primary tumor should include chest radiography and (2) the patient must receive at least 6 months of follow-up. Metastatic carcinoma of the mandible occurs mainly from the breast, thyroid, kidneys, and lungs.4 The mean age of patients with PIOC is 52.3 years (age range, 4-81 years). The male to female ratio is 2.5:1.0, and a background of alcohol or tobacco abuse is usually not present.5The total number of reported cases is difficult to determine owing to insufficient data to support the diagnosis in some published cases.3However, the total number of reported PIOC cases is approximately 134, including 82 cases of PIOC type 1 and 52 cases of PIOC type 3.6Both types are more frequently found in the mandible, with a predilection in the posterior region of the mandible, similar to benign tumors and odontogenic cysts,3but they have also been found in the maxilla. In their review of the world literature, Thomas et al5found that 77% of PIOCs were located in the posterior mandible, 8.5% in the anterior mandible, and 11.4% in the maxilla. The tumor typically presents as a painful swelling in the jaw area, but it may be asymptomatic and found on routine panoramic radiography, as in the present case. Clinically, homolateral positive neck nodes have been noted in 28% of patients at the time of diagnosis, with the majority located in the submandibular triangle.7Imaging has been found to be one of the most effective methods for detecting PIOCs8; however, they can present as radiolucent lesions with great variation, either unilocular or multilocular, with an ill-defined or well-defined but noncorticated border.6They are generally lytic, with evidence of destructive changes to the bone. It is important to note that even benign-appearing osseous lesions may contain small areas that indicate a malignant nature. The differential diagnosis of radiolucent lesions of the mandible includes carcinoma, osteosarcoma, invasive lesions of the oral mucosa, distant metastasis, periapical lesions, cysts of the globullomaxillary region, dentigerous cysts, primordial cysts, odontogenic keratocysts, and ameloblastoma. During radiographic evaluation of lesions of the jaw, malignant changes should be considered if a radiolucent area shows jagged or irregular margins with indentations and indistinct borders suggestive of invasion and destruction by tumor or if serial occlusal radiographs show rapid enlargement of the radiolucent area.9Panoramic radiography can be useful in cases of gross disease, but it is of limited value in evaluating margins and extension and invasion of the tumor. Computed tomography can reveal soft-tissue masses, aid in delineating the extent of the malignant lesion, and disclose invasion of adjacent anatomical regions.9However, its usefulness may be limited by the presence of intraoral metal prostheses.10 The treatment of choice for PIOC is surgery and/or postoperative radiation therapy.5,7However, there is no overall treatment consensus, as there are no comprehensive comparisons because of the rarity of the disease. Overall, PIOC portends a poor prognosis. Zwetyenga et al7recommend surgical treatment combined with postoperative radiotherapy for 2 reasons: (1) PIOC is similar to locally advanced squamous cell carcinoma of the oral mucosa, particularly T4 gum carcinoma, which often requires the same aggressive treatment; and (2) although the results of their comparison of survival according to the treatment modalities were not statistically significant, the trend that they analyzed favored radical surgery followed by postoperative radiotherapy. In their review, 47% of the patients were treated with surgery alone and 38% were treated with surgery plus radiotherapy. Neck dissection was performed in 69.7% of the patients, 43% of whom had histologic evidence of nodal disease. Patients undergoing radical surgery and postoperative radiotherapy had 2- and 3-year survival rates of 61.3% and 40.9%, respectively, whereas the remaining patients had rates of 59.7% and 31.3%. Thomas et al5found that overall survival rates were 75.7%, 62.1%, and 37.8% at 1, 2, and 3 years, respectively. However, their calculations did not separate the patients who were treated with surgery alone and those who were treated with surgery plus adjuvant radiation therapy. In our case, the findings of the original outpatient biopsy were inconclusive, showing either squamous cell carcinoma or mucoepidermoid carcinoma. The patient underwent a radical composite surgical resection in which a fibular osseocutaneous free-tissue transfer was used for reconstruction. Because of the 43% rate of occult metastasis found by Zwetyenga et al,7a selective neck dissection at ipsilateral levels 1 through 4 was performed. Gross pathologic examination revealed a 2.2-cm, firm, tan mass with a necrotic center that was entirely contained within the bone and no evidence of a cystic component. Histologic examination demonstrated a moderately well-differentiated squamous cell carcinoma (Figure 4) with no evidence of lymph node metastasis and negative surgical margins. Adjuvant radiotherapy was not recommended given the patient's pathologically negative nodal status, and there has been no evidence of recurrent disease to date. Figure 4. View LargeDownload Return to Quiz Case 1. References 1. Kramer IRHPindborg JJShear M International Histological Classification of Tumours: Histological Typing of Odontogenic Tumours. 2nd ed. Heidelberg, Germany Springer-Verlag1992; 2. Waldron CAMustoe TA Primary intraosseous carcinoma of the mandible with probable origin in an odontogenic cyst. Oral Surg Oral Med Oral Pathol 1989;67 (6) 716- 724PubMedGoogle ScholarCrossref 3. Suei YTanimoto KTaguchi AWada T Primary intraosseous carcinoma: review of the literature and diagnostic criteria. J Oral Maxillofac Surg 1994;52 (6) 580- 583PubMedGoogle ScholarCrossref 4. To EHWBrown JSAvery BSWard-Booth RP Primary intraosseous carcinoma of the jaws: three new cases and review of the literature. Br J Oral Maxillofac Surg 1991;29 (1) 19- 25PubMedGoogle ScholarCrossref 5. Thomas GPandey MMathew A et al. Primary intraosseous carcinoma of the jaw: pooled analysis of world literature and report of two new cases. Int J Oral Maxillofac Surg 2001;30 (4) 349- 355PubMedGoogle ScholarCrossref 6. Chaisuparat RColetti DKolokythas AOrd RANikitakis NG Primary intraosseous odontogenic carcinoma arising in an odontogenic cyst or de novo: a clinicopathologic study of six new cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101 (2) 194- 200PubMedGoogle ScholarCrossref 7. Zwetyenga NPinsolle JRivel J et al. Primary intraosseous carcinoma of the jaws. Arch Otolaryngol Head Neck Surg 2001;127 (7) 794- 797PubMedGoogle Scholar 8. Kaffe IArdekian LPeled MMachtey ELauffer D Radiological features of primary intra-osseous carcinoma of the jaws: analysis of the literature and report of a new case. Dentomaxillofac Radiol 1998;27 (4) 209- 214PubMedGoogle ScholarCrossref 9. Cavalcanti MGPVeltrini VCRuprecht AVincent SDRobinson RA Squamous-cell carcinoma arising from an odontogenic cyst—the importance of computed tomography in the diagnosis of malignancy. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100 (3) 365- 368PubMedGoogle ScholarCrossref 10. Lin YJChen CHWang WCChen YKLin LM et al. Primary intraosseous carcinoma of the mandible. Dentomaxillofac Radiol 2005;34 (2) 112- 116PubMedGoogle ScholarCrossref

Journal

Archives of Otolaryngology - Head & Neck SurgeryAmerican Medical Association

Published: Feb 16, 2009

Keywords: radiology specialty

References