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      A spindle cell carcinoma presenting with osseous metaplasia in the gingiva: a case report with immunohistochemical analysis

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          Abstract

          Background

          Spindle cell carcinoma (SpCC) is a rare, high malignant variant of squamous cell carcinoma (SCC), which shows biphasic proliferation of conventional SCC component and malignant spindle shape cells with sarcomatous appearance.

          Methods

          A case of Spindle cell carcinoma with bone-like calcified materials, occurring at the mandibular molar region of 71-years-old Japanese male patient was presented with gross finding, histological findings and MRI image. To identify the characteristics of the bone-like materials, immunohistochemistry were performed.

          Results

          Histologically, the cancer cells were composed of spindle cells and epithelial cells which form nests with prominent keratinization. Histological findings showed typical histology of the SpCC, however, as an uncommon finding, spatters of calcified, bone-like materials were observed in between the cancer cells. Immunohistochemistry revealed that cancer cells were positive for cytokeratins and vimentin to a varying degree and negative for Desmin, S-100, Osteopontin, BMP-2 or BMP-4. These findings implied that the calcified materials were formed by metaplasia of the stromal cells.

          Discussion

          Bone-like materials formation by osseous and/or cartilaginous metaplasia of the stroma in the carcinoma has been reported. However, the detailed mechanism of these metaplasia and affection on the clinical feature, prognosis and therapies are not well established. In summary, we presented an unique case of SpCC, which has not been described in the literature.

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          Most cited references28

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          Spindle cell (sarcomatoid) carcinomas of the larynx: a clinicopathologic study of 187 cases.

          Laryngeal spindle cell (sarcomatoid) carcinomas are uncommon tumors, frequently misdiagnosed as reactive lesions or mesenchymal malignancies. The records of 187 patients with tumors diagnosed as laryngeal spindle cell (sarcomatoid) carcinoma were retrieved from the files of the Otorhinolaryngic Tumor Registry of the Armed Forces Institute of Pathology. There were 174 men and 13 women, 35-92 years of age (average, 65.6 years). Nearly all patients experienced hoarseness (n = 165 [88%] patients) for a mean duration of 11.0 months. Patients admitted to smoking (n = 162 [87%] patients) and/or alcohol use (n = 90 [48%] patients). Most tumors were glottic (n = 132 [71%]), T1 (n = 111 [59%]), 1 and polypoid (n = 185 [99%]), with a mean tumor size of 1.8 cm. Histologically, squamous cell carcinoma (n = 157 [84%]) was noted, ulcerated, and blended with the spindle cell component, which was most frequently arranged in a storiform pattern (n = 92 [49%] tumors). Foci of benign or malignant cartilage and/or bone (n = 13 [7%]) were noted in the spindle cell component. All patients were treated with surgery (n = 90 [48%] patients) or surgery with radiation (n = 97 [52%] patients). Recurrences developed in 85 (45%) patients. Overall, T1 glottic tumors managed by complete surgical eradication had the best outcome (mean follow-up, 7.8 years).
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            Monoclonal origins of malignant mixed tumors (carcinosarcomas). Evidence for a divergent histogenesis.

            Malignant mixed tumors (carcinosarcomas) are examples of unusual neoplasms whose occurrences have been observed in increasingly diverse sites but whose pathogenesis remains a complete mystery. Two antithetical hypotheses that have been advanced to explain the histogenesis of these tumors include the convergence hypothesis, which proposes an origin from two or more stem cells (multiclonal hypothesis), and the divergence hypothesis, which proposes an origin from a single totipotential stem cell that differentiates into separate epithelial and mesenchymal directions (monoclonal hypothesis). To test these hypotheses, a novel strategy for the determination of clonality from as few as 100 tumor cells obtained by enzymatic digestion of either fresh or formalin-fixed, paraffin-embedded tissues and cell sorting was used that exhibited the polymerase chain reaction (PCR) in amplifying a 511-bp region located within the first intron of the human hypoxanthine phosphoribosyl transferase gene, a site that contains inactive X chromosomal obligately methylated HpaII/MspI sites and single-base allelic polymorphisms in 5% females. Carcinoma cells gated on the basis of fluorescein isothiocyanate (FITC)-anti-cytokeratin and sarcoma cells gated on the basis of FITC-antivimentin or FITC-anti-desmin were sorted to homogeneity on FACSTAR and then subjected to genomic DNA extraction and Hpa II digestion before PCR amplification and subsequent analysis of the product on denaturing gradient gel electrophoresis. The comigrations of the single homoduplexes generated from both the carcinoma cells and sarcoma cells in six different malignant mixed tumors obtained from four different organs indicated clonal identity and monoclonality in all cases. These findings of monoclonality were confirmed independently by two other methods of clonality determination. The findings of a monoclonal origin of carcinosarcomas support the single totipotential stem-cell-divergence hypothesis.
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              Spindle cell carcinoma of the oral cavity and oropharynx: factors affecting outcome.

              Spindle cell carcinoma (SpCC) is considered to be a rare variant of squamous cell carcinoma (SCC). The behavior of such tumors is unclear. The aim of this study was to elucidate the treatment and outcome of oral and oropharyngeal SpCC. All the medical records of patients with the diagnosis of SpCC in the oral cavity and oropharynx in our hospital from 1994 to 2005 were reviewed. The clinical features, treatments and survival of the patients were evaluated. Within the 11-year study period, 18 patients were diagnosed with oral and oropharyngeal SpCC. There were 3 cases of AJCC (American Joint Committee on Cancer) stage I, 3 of stage II, 2 of stage III, 9 of stage IV, and 1 case without definite staging. Twelve patients died of their diseases. The median overall survival time was 8.89 months. The 1-year overall survival rate was 36.7% and the 3-year overall survival rate was 27.5%. In the early stage group, the 1-year and 3-year survival rates were both 100%. In the late stage group, the 1-year survival rate was 9%, and the 3-year survival rate was 0%. The factors influencing overall survival were tumor grade, lymph nodes, metastasis, stage, vascular invasion and distant recurrence. A high local recurrence rate (73.3%) and distant metastasis rate (33.3%) were observed. The behavior of SpCC seems to be more aggressive than that of SCC at a similar stage. Setting wider safety margins (> 2 cm) during surgical intervention is suggested. In the case of locoregional recurrence, salvage operation showed some benefit. Seeking an effective chemotherapy protocol is important for the control of distant recurrence.
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                Author and article information

                Journal
                Head Face Med
                Head & Face Medicine
                BioMed Central
                1746-160X
                2008
                1 December 2008
                : 4
                : 28
                Affiliations
                [1 ]Department of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Okayama, 700-8525, Japan
                [2 ]Department of Oral and Maxillofacial Radiology, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Okayama, 700-8525, Japan
                [3 ]Department of Oral and Maxillofacial Surgery and Biopathological Science, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Okayama, 700-8525, Japan
                Article
                1746-160X-4-28
                10.1186/1746-160X-4-28
                2627831
                19040765
                1a1c8754-ac3a-4f97-a2f7-7bd959282928
                Copyright © 2008 Katase et al; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 1 August 2008
                : 1 December 2008
                Categories
                Case Report

                Orthopedics
                Orthopedics

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