View metadata, citation and similar papers at core.ac.uk brought to you by CORE provided by Elsevier - Publisher Connector Oral Oncology EXTRA (2006) 42, 247– 250 available at www.sciencedirect.com journal homepage: http://intl.elsevierhealth.com/journal/ooex CASE REPORT Chondrosarcoma of the mandibular symphysis Takayuki Shirato a, Kojiro Onizawa a, Kenji Yamagata a, Hiroshi Yusa a, Tatsuo IIjima b, Hiroshi Yoshida a,* a Department of Oral and Maxillofacial Surgery, Institute of Clinical Medicine, University of Tsukuba, 1-1-1 Tennoudai, Tsukuba, Ibaraki 305-8575, Japan b Department of Pathology, Institute of Basic Medical Science, University of Tsukuba, 1-1-1 Tennoudai, Tsukuba, Ibaraki 305-8575, Japan Received 8 March 2006; received in revised form 27 March 2006; accepted 3 April 2006 KEYWORDS Summary A rare case of chondrosarcoma of the mandibular symphyseal region in a 39-year- Chondrosarcoma; old man is presented. The patient complained of a recurring and growing mass on the labial side Mandible; of the mandibular gingiva extending from the right lateral incisor to the left canine. Radio- Mandibular; graphs showed irregularly shaped osteoblastic lesion with radiopacity corresponding to the Symphysis mass. MR images showed the lesion to be well-defined and adjacent to the labial cortex without bone marrow invasion. The mass was histologically diagnosed as grade 2 chondrosarcoma. Nine- teen months after marginal resection with tumour-free margins, there was no evidence of recurrence or metastasis. c 2006 Elsevier Ltd. All rights reserved. Introduction might persist in the mandibular symphysis from its develop- ment,4,9,10 to our knowledge, only 12 cases of chondrocar- comas arising at the mandibular symphyseal region have Chondrosarcomas are rare malignant tumours characterized 2,11 by the formation of cartilage, but not bone, by tumour been reported, and in none of theses cases was the tu- cells. Chondrosarcomas of the maxillofacial region, mour’s growing behaviour described. This report presents accounting for 1–3% of all chondrosarcomas of the entire a new case of chondrosarcoma that appeared to have arisen body,1,2 arise predominately in the maxilla with a predilec- intraosseously and extended peripherally along the external tion for the anterior maxillary region,3 and occur at lower surface of the labial cortex of the mandibular symphysis. incidence in the mandible.1,4–7 The preferred site in the mandible is the molar region, and they infrequently occur Case report in the ramus, condyle, coronoid process, or symphysis.8 Although remnants of Meckel’s cartilage or fibrocartilage A 39-year-old man was referred to the Division of Oral and Maxillofacial Surgery, Tsukuba University Hospital complain- * Corresponding author. Tel./fax: +81 29 853 3050. ing of a painless mass on the anterior mandibular ridge, E-mail address: [email protected] (H. Yoshida). which had been present for 6 months. The patient had 1741-9409/$ - see front matter c 2006 Elsevier Ltd. All rights reserved. doi:10.1016/j.ooe.2006.04.002 248 T. Shirato et al. undergone excision of the mass 2 months before by a gen- eral dental practitioner, but the removed specimen had not been submitted for pathological analysis. The mass had recurred and gradually grew in size. Physical examina- tion showed no abnormal sensitivity of the lower lip and no lymphadenopathy in the neck. Intraorally, a well-de- fined, nodular mass was found on the labial side of the man- dibular gingiva extending from the right lateral incisor to the left canine, and extending superiorly to the alveolar ridge. The mass was elastic hard and covered with normal- appearing mucosa. An interdental space was found between the left central and lateral incisors, which had been present for 1 year (Fig. 1). The teeth in the region showed slight loosening and responded to vital testing. A panoramic radiograph revealed the a widened inter- dental space between the left central and lateral incisors Figure 1 Clinical appearance showing a mass on the labial that erupted parallel to each other, and an irregularly side of the anterior mandibular region and an interdental space shaped osteoblastic lesion with radiopacity corresponding between the left central and lateral incisors. to the interdental space (Fig. 2A). Occlusal radiography Figure 2 (A) Panoramic radiograph showing a widened interdental space between the right central and lateral incisors that erupted parallel to each other, and an irregularly shaped osteoblastic lesion with radiopacity corresponding to the interdental space. (B) Occlusal radiograph showing an irregular, slightly radiopaque extrusion consistent with the location of the mass. (C) CT scans showing a slightly radiopaque lesion on the outer labial cortex in the anterior mandibular region. (D) MR images revealed that the lesion was well-defined and adjacent to the labial cortex without bone marrow invasion. Chondrosarcoma of the mandibular symphysis 249 Figure 3 (A) Histologic feature of biopsy specimen. Spindle-shaped to stellate tumour cells with enlarged, oval nuclei and occasional mitotic figures proliferated in myxomatous stroma and focally formed hyaline cartilage with irregular ossification (H & E staining, ·200). (B) Histologic feature of the resected specimen. Clusters of enlarged chondrocytes with hyperchromatic nuclei, binucleation and nuclear pleomorphism were observed in a myxoid background. Mitosis was occasionally presented (H & E staining, ·400). (C) The labial alveolar bone was partially invaded with tumour, but almost all of cortical bone bordering the lesion was intact (H & E staining, ·40). showed an irregular, slight radiopaque extrusion consistent the tumour, but almost all of the cortical bone bordering with the location of the mass (Fig. 2B). CT scans revealed a the lesion was intact (Fig. 3C). The periphery of the tumour slightly radiopaque lesion on the outer surface of the labial was well-demarcated and covered with a capsule of soft tis- cortex in the anterior mandibular region (Fig. 2C). MR sue. Partial calcification and hyperplasia of bone were images showed the lesion to be well-defined and adjacent found in the tumour. The surgical margins were free of tu- to the labial cortex without bone marrow invasion mour cells. Nineteen months after the surgery, the patient (Fig. 2D). These findings suggested the tumour was localized is still free of disease, with no evidence of recurrence or dis- subperiosteally. Total body bone scintigraphy did not show tant metastasis clinically and radiographically. any abnormal accumulation except in the mandibular sym- physeal region. A biopsy specimen revealed the spindle-shaped to stellate Discussion tumour cells that had enlarged, oval nuclei and occasional mitotic figures with a distinct nucleolus, proliferated in the Chondrosarcomas of the mandible most commonly present myxomatous stroma and focally formed hyaline cartilage as a painless swelling or mass of long duration, and pain, with irregular ossification (Fig. 3A). These histologic findings paresthesia, trismus, and loosening of the teeth are associ- were consistent with grade 2 chondrosarcoma. ated with the progression of the disease. Radiographically, After the biopsy, the lesion expanded rapidly. One month the appearance of the lesion varies from ill-defined radiolu- after the initial presentation, a marginal resection was per- cency to obvious radiopacity, but these findings are not formed between the left second premolar and the right first pathognomonic.12 CT and MRI are quite valuable in deter- premolar regions under general anesthesia, because the mining the nature and extent of the lesion,8,11 but a defin- mandibular basal bone appeared radiographically to be itive diagnosis needs to be made histologically. unaffected. Histologic examination of the resected speci- Tumours whose histological differentiation from chon- men showed clusters of enlarged chondrocytes with hyper- drosarcoma is difficult and important are chondroma and chromatic nuclei, binucleation, and nuclear pleomorphism chondroblastic osteosarcoma. Chondroma predominantly in a myxoid background. Mitosis was occasionally presented occurs in small bone,13 and is extremely rare in the jaw (Fig. 3B). The labial alveolar bone was partially invaded by and facial bones.1 Accordingly, cartilaginous tumours of 250 T. Shirato et al. the jaws should be usually interpreted as malignant rather tumour-free margins, suggesting a favourable prognosis. than benign.1 It is important to differentiate chondrosar- However, long-term follow-up is essential, because chon- coma from chondroblastic osteosarcoma in the jaw, be- drosarcomas show a wide variation in time of recurrence cause the reported prognosis for the former is more and metastasis.8,18 favourable than for the later.1 Chondrosarcomas are com- posed purely of hyaline cartilage and fulfill cytologic malig- References nant criteria.14 Osteosarcomas are arranged in lobules composed of cytologically malignant cells, with spindling 1. Saito K, Unni KK, Wollan PC, Lund BA. Chondrosarcoma of the of the tumour cells toward the periphery of the lobules. jaws and facial bones. Cancer 1995;76(9):1550–8. 13 Osteoid is usually present between the spindle cells, and 2. Izadi K, Lazow SK, Solomon MP, Berger JR. Chondrosarcoma a pattern of chondroid lobules with bone formation is also of the anterior mandible. A case report. N Y State Dent J observed.1 The absence of osteoid and neoplastic bone 2000;66(7):32–4. in the case presented here ruled out chondroblastic 3. Martis C. Chondrosarcoma of the mandible: report of case. J osteosarcoma. Oral Surg 1978;36(3):227–30. In the present case, a mass was noticed on the anterior 4. Fronstin MH, Hutcheson JB, Sanders HL. Chondrosarcoma of the mandibular ridge 6 months after the occurrence of a wid- mandibular symphysis. Report of a case. Oral Surg Oral Med ened interdental space between the left central and lateral Oral Pathol 1968;25(5):665–9. 5. Weiss Jr WW, Bennett JA. Chondrosarcoma: a rare tumor of the incisors. The direction of the roots of these incisors, which jaws. J Oral Maxillofac Surg 1986;44(1):73–9.