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Indian Journal of Cancer
Medknow Publications on behalf of Indian Cancer Society
ISSN: 0019-509X EISSN: 1998-4774
Vol. 46, Num. 3, 2009, pp. 231-233

Indian Journal of Cancer, Vol. 46, No. 3, July-September, 2009, pp. 231-233

Original Article

Gnathic osteosarcomas: A 10-year multi-center demographic study

Professor OMF Surgery, Trauma Research Center, BMSU, Tehran
Correspondence Address:Professor OMF Surgery, Trauma Research Center, BMSU, Tehran
motamedical@lycos.com

Code Number: cn09049

PMID: 19574676
DOI: 10.4103/0019-509X.52958

Abstract

Background: Osteosarcomas (OS) of the jaws are uncommon lesions representing 6-8% of skeletal OS. We assessed the characteristics, demographics, prevalence clinical and histopathological findings and distribution of gnathic OS relative to non-gnathic OS in four major treatment centers.
Materials and Methods: This study assessed 13 gnathic OS patients of 98 OS patients from four major referral centers during 1996-2007. The age distribution, gender, involved site, clinical findings, signs, symptoms, grade and sub-types were assessed. Hematoxylin-eosin, Picrosirius red, Ponceau trichrome, Masson trichrome and osteoid staining methods were used.
Results:
Of the 98 OS lesions, 85 (86.8%) involved the skeleton, the youngest patient was 6 and the oldest 60 years old; 13 lesions (13.2%) involved the jaws (seven mandibular and six maxillary) and the youngest and oldest patients were 15 and 50 years-old, respectively. Non-gnathic OS was more prevalent between the ages of 11 and 20 years (avg. 15 years) and was common in the distal femur and proximal tibia, presenting most frequently with pain and swelling. OS of the jaws, however, presented more than 10 years later than non-gnathic OS, being more prevalent between the ages of 20 and 30 years (avg. 27 years). OS of the jaws most frequently involved the mandibular body and the posterior maxillary alveolar ridge, presenting frequently with pain, swelling and loosening of teeth. Two patients with gnathic OS died during the 10-year follow-up period (15.3%).
Conclusion:
Prevalence of OS of the jaws was about twice as high as that reported in other studies and presented later than non-gnathic cases. Pain and swelling were common signs and symptoms in this disease. The mixed sub-type was the most common sub-type of gnathic OS.

Keywords: Demographics, jaws, osteosarcoma, subtypes

Introduction

Osteosarcomas (OS) of the jaws are uncommon and represent 6-8% of all OS. [1],[2],[3] OS of the jaw most often presents in the third and fourth decades of life. [1],[2],[3],[4],[5],[6] The mean age of patients with OS of the jaw is about 33 years, which is 10-15 years older than the mean age for OS of long bones. [2],[3],[4],[7],[8],[9],[10] As is seen in extra-gnathic locations, a slight male predominance is noted. [1],[3],[7],[8],[11] The maxilla and mandible are involved, with about an equal frequency. [1],[2],[7] Mandibular OS arises more frequently in the posterior part. [1],[2],[7] Maxillary lesions are discovered more commonly in the posterior alveolar ridge and sinus floor or palate. [1] Swelling and pain are the most common signs and symptoms. [1],[3],[7],[9],[11] Loosening of teeth, paresthesia and nasal obstruction are also noted. [1],[3],[9]

The demographic characteristics, distribution and sub-types of OS vary in different countries, assessment of which may aid treatment. Information regarding gender, age, signs and symptoms may be useful in early diagnosis and management and prevent further damage to hard and soft tissues. We assessed files and specimens diagnosed with OS from 1996 to 2007 at four major referral centers to compare with the body of literature on the subject.

Materials and Methods

A 10-year retrospective study assessed data collected from 98 OS patients from 1996 to 2007. Information regarding age, distribution, gender, involvement site, prevalence, signs, symptoms and sub-types were documented from four major referral centers. Thirteen of these 98 lesions were jaw lesions (seven mandibular and six maxillary) and the youngest and oldest patients were 15 and 50 years-old, respectively. Eighty-five were non-gnathic cases, the youngest and oldest patients being 6 and 60 years old, respectively. The age distribution, gender, involved site, clinical findings, signs, symptoms, grade and sub-type of lesions were assessed. Hematoxylin-eosin, Picrosirius red, Ponceau trichrome, Masson trichrome and osteoid staining methods were used.

Results

In our study, OS of the jaw was more prevalent between 20 and 30 years of age (avg. 27 years of age) while non-gnathic OS was more prevalent between the ages of 11 and 20 years (avg. 15 years of age). OS of the jaws comprised 13.3% of all OS of the body. The most frequent signs and symptoms accompanying gnathic OS were swelling and pain (69.2%), followed by loosening of the teeth (15.4%), widening of the periodontal ligament (15.4%), ulceration (7.7%) and metastases (15.4%). The common signs and symptoms of the non-gnathic cases were also swelling and pain followed later by weight loss, weakness, fever and anorexia. The most frequent sites of involvement in gnathic OS were the body of the mandible in the region of molar teeth and the posterior region of the maxilla involving the paranasal sinuses. The most frequent sites for non-gnathic cases were the distal femur and the proximal tibia areas. With regard to microscopic morphology of the jaw lesions, the distribution of histopathologic subtypes were osteoblastic (23.0%), fibroblastic (7.7%), telangiectatic (7.7%), small cell (7.7%) and mixed (53.8%). Cases that displayed more than one histopathologic sub-type microscopically were classified under the mixed sub-type. During the 10-year follow-up period, two patients with gnathic OS died.

Discussion

OS of the jaws is uncommon and represents 6-8% of all skeletal OS. [1],[2] But, in this study, it was somewhat greater (13.3%). Guo reported a higher incidence of OS patients in Asia than in America and stated that OS occurred more in the flat bones in Americans than in Asians. [1] Our data showed OS of the jaws to be more prevalent between the ages of 20 and 30 years (avg. 27 years of age), slightly less than that reported by Patel et al ., whose patients ranged in age from 6 to 64 years (avg. 31 years). [2] In his study, the mandible was the primary site in 18 patients (41%), the maxilla in 20 patients (45%) and the skull in six patients (14%).[2] Nissanka reported the mean age for OS of the jaws to be 34.1 years in 19 patients (11 mandibular lesions and eight maxillary lesions). [8] In Ogunlewe′s study, the mean age of patients at presentation was 27.2 years (range 11-70 years), corresponding to our study. [9] The mean age for OS of the long bones has been reported to be 10-15 years lesser than OS of the jaws. [2],[3] The mean age of non-gnathic OS in our patients was 15 years (12 years less than gnathic OS).

Immunohistochemical methods alone are not sufficient for a diagnosis of OS. The diagnosis is based on correlated clinical, radiographic and histopathological findings. [1],[9],[11],[12],[13] Gnathic OS usually has a high microscopic grade in most cases, and the grade is higher in males. [1],[3],[7] Hematoxylin-eosin, Picrosirius red, Ponceau trichrome, Masson trichrome and osteoid staining methods were used. We had two cases of grade II (15.4%), eight cases of grade III (61.6%) and three cases of grade IV (23.0%) gnathic OS. There were no cases of grade I gnathic OS.

With regard to microscopic morphology, some authors have reported the most common morphology of OS to be of the mixed sub-type, as was the case in our study. [6] However, in Nissanka′s [8] study, the osteoblastic variant was the most common histopathological sub-type in gnathic OS while in Ogunlewe′s study, the most common histologic subtype was chondroblastic (47%), followed by the fibroblastic type (35.3%). [9] In all these studies, however, the cases are too few to be statistically significant. The trichrome method of staining undecalcified tissues according to Masson is good for staining decalcified bone sections. Masson′s method stains mineralized bone (blue) and osteoid (red). [14] To distinguish between osteoid and other tissue components that can be mistaken for osteoid, osteoid from both normal osteogenesis and OS is disclosed by the Picrosirius-polarization method as a three-dimensional network of randomly arranged, thin, short, collagenous structures that shine against a dark background. These morphologic features can be used as a precise diagnostic criterion for the differential diagnosis between osteoid and other materials that resemble osteoid by other staining techniques. This precise characterization of osteoid is of great importance because the presence of osteoid is used as a criterion for the differential diagnosis of OS. Not only does this method permit the precise characterization of osteoid but it is also useful for studying collagen distribution in OS. [15] With modified Ponceau trichrome staining, bone formation tissues showed a homogenous, orange-red color inter-blended with blue. From osteoid to mature bone, the color changes from orange-red, light blue to dark blue. Fibrotic tissue stained blue with striated appearance. Cartilage was not stained. The Picrosirius red method gives bone-forming tissues a homogenous staining. Along with bone maturation, from osteoid tissue to mineralized bones, the color changes from light red, yellow, orange-red, red to dark purple. The cartilage demonstrates a homogenous light red color. Fibrous tissue stains red inter-blended with yellow. The modified Ponceau trichrome and Picrosirius red staining methods are better than Masson trichrome to demonstrate bone formation tissue in OS. [16]

An important early sign of gnathic OS is symmetrical widening of the periodontal ligaments of one or several teeth. This feature presents before any other radiographic feature and is of paramount importance because this feature is seen only in malignancies, scleroderma and acrosclerosis. [3] This finding along with pain and swelling is helpful in the early diagnosis of OS of the jaws. In our study, pain and swelling were the most common presenting signs and symptoms. The common signs and symptoms of the non-gnathic cases were also swelling and pain, followed later by weight loss, weakness, fever and anorexia.

The treatment of choice for primary OS of the jaws remains to be surgery primarily, followed by chemotherapy and radiotherapy. Although the literature states that the 5-year survival rate is better for OS of the jaw, some more recent literature has survival rate better in OS of the long bones. [3] An individualized approach has been stated to offer the highest likelihood of survival in this patient population. [12] Multi-disciplinary treatment of craniofacial OS within a multi-center setting resulted in long-term survival in well over two-thirds of affected patients in Jasnau′s study. [12] Involvement of extragnathic sites and failure to achieve and maintain local surgical control are strong negative prognostic factors. [13] Although demographic data differs in various reports, further studies are needed to see if differing demographic data from different societies influence treatment outcomes in patients with OS. Early diagnosis and primary surgical treatment have been shown to increase the 5-year survival rate in OS. [1],[3],[9],[11]

References

1.Guo W, Xu W, Huvos AG, Healey JH, Feng C.Comparative frequency of bone sarcomas among different racial groups. Chin Med J (Engl) 1999;112:1101-4.   Back to cited text no. 1  [PUBMED]  
2.Patel SG, Meyers P, Huvos AG, Wolden S, Singh B, Shaha AR, et al . Improved outcomes in patients with osteogenic sarcoma of the head and neck. Cancer. 2002;95:1495-503.  Back to cited text no. 2    
3.Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. Philadelphia: WB Saunders; 2002. p. 574-8.  Back to cited text no. 3    
4.Matsuzaka K, Shimono M, Uchiyama T, Noma H, Inoue T. Lesion related to the formation of bone, cartilage or cementum arising in the oral area: a statistical study and review of the literature. Bull. Tokyo. Dent Coll. 2002,;43:173-80.  Back to cited text no. 4    
5.Regezi JA, Sciubba JJ. Oral Pathology Clinical Pathologic Correlations. W.B Saunders, Philadelphia, 3rd ed., 1999; pp 397-404.  Back to cited text no. 5    
6.Nora FE, Unni KK, Pritchard DJ, Dahlin DC. Osteosarcoma of extragnathic craniofacial bones. Mayo Clin Proc 1983;58:268-72.  Back to cited text no. 6  [PUBMED]  
7.Slootweg PJ, Muller H. Osteosarcoma of the jaw bones. Analysis of 18 cases. J Oral Maxillofac Surg 1985;13:158-66.  Back to cited text no. 7    
8.Nissanka EH, Amaratunge EA, Tilakaratne WM. Clinicopathological analysis of osteosarcoma of jaw bones. Oral Dis 2007;13:82-7.   Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Ogunlewe MO, Ajayi OF, Adeyemo WL. Osteogenic sarcoma of the jaw bones: a single institution experience over a 21-year period. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101:76-81.   Back to cited text no. 9    
10.Bennett JH, Thomas G, Evans AW, Speight PM. Osteosarcoma of the jaws: a 30-year retrospective review. Oral Surg Oral Med Pathol Oral Radiol Endod. 2000;90; 323-32.  Back to cited text no. 10    
11.Chindia ML. Osteosarcoma of the jaw bones. Oral Oncol 2001;37:545-7.  Back to cited text no. 11  [PUBMED]  [FULLTEXT]
12.Nathan SS, Gorlick R, Bukata S, Chou A, Morris CD, Boland PJ, et al . Treatment algorithm for locally recurrent osteosarcoma based on local disease-free interval and the presence of lung metastasis. Cancer 2006;107:1607-16.   Back to cited text no. 12  [PUBMED]  [FULLTEXT]
13.Jasnau S, Meyer U, Potratz J, Jundt G, Kevric M, Joos UK, et al . Craniofacial osteosarcoma: Experience of the cooperative German-Austrian-Swiss osteosarcoma study group. Oral Oncol 2008;44:286-94.  Back to cited text no. 13  [PUBMED]  [FULLTEXT]
14.Asonova SN, Migalkin NS. Use of Masson's trichrome method for staining decalcified bone tissue. Arkh Patol 1996;58:66-7.  Back to cited text no. 14    
15.Junqueira LC, Assis Figueiredo MT, Torloni H, Montes GS. Differential histologic diagnosis of osteoid. A study on human osteosarcoma collagen by the histochemical picrosirius-polarization method. J Pathol 1986;148:189-96.  Back to cited text no. 15    
16.Li Q, Gong XQ, Ma FC, Zhao YL, Zhu XH. Application of histochemical staining in diagnosis of osteosarcomas, Zhonghua Zhong Liu Za Zhi 2005;27:489-91.   Back to cited text no. 16    

Copyright 2009 - Indian Journal of Cancer

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