A spindle cell carcinoma presenting with osseous metaplasia in the gingiva: a case report with immunohistochemical analysis
© Katase et al; licensee BioMed Central Ltd. 2008
Received: 01 August 2008
Accepted: 01 December 2008
Published: 01 December 2008
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.
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.
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.
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.
Spindle cell carcinoma (SpCC), also known as sarcomatoid carcinoma or pseudosarcoma, of head and neck is a rare neoplasm. SpCC is known as a high malignant variant of squamous cell carcinoma, which is composed of conventional squamous cell carcinoma component, either in-situ and/or invasive and malignant spindle component with sarcomatous appearance. Although it is generally accepted that SpCC is a monoclonal epithelial neoplasm [1–5], and the sarcomatous components are derived from squamous epithelium with divergent mesenchymal differentiation , the diagnosis, classification and management of this tumor infrequently may become subject matter deluded of its histological variety in sarcomatous components. These sarcomatous components commonly resemble to fibrosarcoma or malignant histiocytoma [7, 8], and while rare, foci resembling to chondrosarcoma and/or osteosarcima differentiation may be observed .
This is a case report of a spindle cell carcinoma of gingival mucosa presenting with bone-like calcified materials.
A 71-year-old Japanese male patient was referred to the Okayama University Hospital, complaining the swelling at the left side of the mandibular molar region. The lesion was 44 × 29 mm in size, exophytic with rough and irregular surface. The patient's medical and family histories were unremarkable.
No tumor cells were observed around the surgical margin, and lymph node metastasis was not observed. No recurrence or metastasis was detected since surgery so far.
Antibodies used for the present case
Roche Diagnostics (Germany)
Santa Cruz (USA)
Santa Cruz (USA)
SpCC is a rare variant of squamous cell carcinoma. Its most frequently affected sites is larynx, however, it may infrequently occur in various organs; gingiva [2, 10], tongue [11, 12], upper aerodigestive tract including hypopharynx and nasal cavity [6, 13, 14], esophagus, skin and breast .
It is generally understood that the diagnosis of SpCC requires the demonstration of both components . In accord with this criterion, typical histology of SpCC was observed in the present case, which was composed of conventional squamous cell carcinoma component and spindle shape cells with sarcomatous appearance.
However, the existence of bone-like calcified materials was the uncommon, controversial finding for SpCC in oral mucosa. To the best of our knowledge, this finding was not described in previous report. In the immunohistochemical examination, both epithelial component and spindle shape cell component showed positive reaction for cytokeratins to a varying degree, but vimentin positive cells were limited to the spindle shape cell component. These results were consistent to the previous reports [1, 14, 17–19]. The cells around the materials were vimentin-positive low activity cells, and cancer cells did not show positive reaction for neither BMP-2 nor BMP-4, which indicates these materials were formed by mesenchymal metaplasia of the stromal cells, but not by the tumor cells itself.
Although the incidence of osseous metaplasia is rare finding in oral SpCC, it is occasionally observed in that of larynx . While also rare, the formation of bone-like and/or cartilage-like materials by the metaplasia of stromal cells is often reported in various kind of carcinomas, such as laryngeal cancer , esophageal cancer [21, 22], colon cancer [23–25], lung cancer , and breast cancer . The histogenesis of the osseous and/or cartilaginous metaplasia in the carcinoma has been come up to debate so far. The biological mechanism of these metaplastic changes is considered to be caused by stromal activation associated with human-host interface . Although some report implies the formation of these materials is related to radiation therapy , the mechanism of stromal metaplasia is still unclear. Moreover, the affection of these metaplastic bone or cartilage on the clinical features, prognosis, and response to radiation therapy or chemotherapy are not well established.
In summary, we reported a unique of SpCC with calcified bone-like materials in the gingiva, which is a very rare case in the literature.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
Spindle cell carcinoma
The work was supported by Grant-in-Aid for Scientific Research (B) No.20791337 and (C) No.19592109 from the Japanese Ministry of Education, Culture, Sports, Science and Technology. Written consent for publication was obtained from the patients or their relative. The authors would like to thank Ms. Kazuko Funakoshi for the expert technical assistance in histological preparations.
- Takata T, Ito H, Ogawa I, Miyauchi M, Ijuhin N, Nikai H: Spindle cell squamous carcinoma of the oral region. An immunohistochemical and ultrastractual study on the histogenesis and differential diagnosis with a clinicopathological analysis of six cases. Virchows Arch. 1991, 419: 177-182. 10.1007/BF01626345.View ArticleGoogle Scholar
- Leventon GS, Evans HL: Sarcomatoid squamous cell carcinoma of the mucous membranes of the head and neck: a clinicopathological study of 20 cases. Cancer. 1981, 48: 994-1003. 10.1002/1097-0142(19810815)48:4<994::AID-CNCR2820480424>3.0.CO;2-M.View ArticlePubMedGoogle Scholar
- Guarino M, Tricomi P, Giordano F, Cristofori E: Sarcomatoid carcinomas: pathological and histopathogenetic considerations. Pathology. 1996, 28: 298-305. 10.1080/00313029600169224.View ArticlePubMedGoogle Scholar
- Thompson L, Chang B, Barsky SH: Mompclonal origins of malignant mixed tumors (carcinosarcomas). Evidence for a divergent histogenesis. Am J Surg Pathol. 1996, 20: 277-285. 10.1097/00000478-199603000-00003.View ArticlePubMedGoogle Scholar
- Torenbeek R, Hermsen MA, Meijer GA, Baak JP, Meijer CJ: Analysis by comparative genomic hybridization of epithelial and spindle cell components in sarcomatoid carcinoma and carcinosarcoma: histogenetic aspects. J Pathol. 1999, 189: 338-348. 10.1002/(SICI)1096-9896(199911)189:3<338::AID-PATH429>3.0.CO;2-Q.View ArticlePubMedGoogle Scholar
- Batsakis JG, Suarez P: Sarcomatoid carcinomas of upper aerodigestive tracts. Adv Anat Pathol. 2000, 7: 282-293. 10.1097/00125480-200007050-00002.View ArticlePubMedGoogle Scholar
- Lewis JE, Olsen KD, Sebo TJ: Spindle cell carcinoma of the larynx: review of 26 cases including DNA content and immunohistochemistry. Hum Pathol. 1997, 28: 664-673. 10.1016/S0046-8177(97)90175-1.View ArticlePubMedGoogle Scholar
- Thompson LD, Wieneke JA, Miettinen M, Heffner DK: Spindle cell (sarcomatous) carcinoma of the larynx: a clinicopathologic study of 187 cases. Am J Surg Pathol. 2002, 26: 153-170. 10.1097/00000478-200202000-00002.View ArticlePubMedGoogle Scholar
- Lambert PR, Ward PH, Berci G: Pseudosarcoma of the larynx: a comprehensive analysis. Arch Otolaryngol. 1980, 106: 700-708.View ArticlePubMedGoogle Scholar
- Munakata R, Cheng J, Nakajima T, Saku T: Spindle cell carcinoma of the gingiva: report of an autopsy case. J Oral Pathol Med. 1998, 27 (4): 180-184.View ArticlePubMedGoogle Scholar
- Sherwin RP, Strong MS, Vaughn CW: Polypoid and junctional squamous cell carcinoma of the tongue and larynx with spindle cell carcinoma ("pseudosarcoma"). Cancer. 1963, 16: 51-60. 10.1002/1097-0142(196301)16:1<51::AID-CNCR2820160108>3.0.CO;2-2.View ArticlePubMedGoogle Scholar
- Chen YK, Lin CC, Chen CH, Yan YH, Lin LM: Spindle cell carcinoma of the tongue: Case report and immunohistochemistry study. Oral Med Pathol. 1998, 3: 51-54. 10.3353/omp.3.51.View ArticleGoogle Scholar
- Altrabulsi B, Carrizo F, Luna MA: Spindle basaloid squamous carcinoma of the upper aerodigestive tract: immunohistochemical and clinicopathological study of three cases. Ann Diagn Pathol. 2006, 10: 149-153. 10.1016/j.anndiagpath.2005.09.010.View ArticlePubMedGoogle Scholar
- Ellis GL, Langloss JM, Heffner DK, Hyams VJ: Spindle-cell carcinoma in the upper aerodigestive tract. An immunohistochemical analysis of 21 cases. Am J Surg Pathol. 1987, 11: 335-342. 10.1097/00000478-198705000-00001.View ArticlePubMedGoogle Scholar
- Su HH, Chu ST, Hou YY, Chang KP, Chen CJ: Spindle cell carcinoma of the oral cavity and oropharynx: Factors affecting outcome. J Chin Med Assoc. 2006, 69: 478-483.View ArticlePubMedGoogle Scholar
- Banes L, Eveson JW, Reichart P, Sidransky D, Eds: World Health Organization Classification of Tumors. Pathology and Genetics of Head and Neck Tumors. 2005, IARC Press: Lyon, 127-
- Meijer JW, Ramaekers FC, Manni JJ, Slooff JJ, Aldeweireldt J, Vooys GP: Intermediate filament of proteins in spindle cell carcinoma of the larynx and tongue. Acta Otolaryngol. 1988, 106: 306-313. 10.3109/00016488809106441.View ArticlePubMedGoogle Scholar
- Slootweg PJ, Roholl PJ, Muller H, Lubsen H: Spindle cell carcinoma of the oral cavity and larynx. Immunohistochemical aspects. J Craniomaxillofac Surg. 1989, 17: 234-236.View ArticlePubMedGoogle Scholar
- Gupta R, Singh S, Hedau S, Nigam S, Das BC, Singh I, Mandal AK: Spindle cell carcinoma of head and neck: an immunohistochemical and molecular approach to its pathogenesis. J Clin Pathol. 2007, 60: 472-475. 10.1136/jcp.2005.033589.View ArticlePubMedGoogle Scholar
- Marioni G, Altavilla G, Marino F, Marchese-Ragona R, Lelli-Mami G, Staffieri A: Squamous cell carcinoma of the larynx with osteosarcima-like stromal metaplasia. Acta Otolaryngol. 2004, 124: 870-873. 10.1080/00016480410017288.View ArticlePubMedGoogle Scholar
- Sugai T, Oikawa M, Uesugi N, Habano W, Jiao YF, Nakamura S, Hatakeyama S, Suhara M, Hatafuku K: Esophageal squamous cell carcinoma characterized by extensive chondroid differentiation. Pathol Int. 2000, 50: 514-519. 10.1046/j.1440-1827.2000.01067.x.View ArticlePubMedGoogle Scholar
- Kwatra KS, Prabhakar BR, Jain S, Grewal JS: Sarcomatoid carcinoma of the esophagus with extensive areas of osseous differentiation: a case report. Indian J Pathol Microbiol. 2003, 46: 49-51.PubMedGoogle Scholar
- Alper M, Akyürek N, Patiroğlu TE, Yüksel O, Belrenli O: Heterotopic bone formation in two cases of colon carcinoma. Scand J Gastroenterol. 2000, 35: 556-558. 10.1080/003655200750023859.View ArticlePubMedGoogle Scholar
- Ansari MQ, Sachs IL, Max E, Alpert LC: Heterotopic bone formation in rectal carcinoma. Case report and literature review. Dig Dis Sci. 1992, 37: 1624-1629. 10.1007/BF01296512.View ArticlePubMedGoogle Scholar
- Kypson AP, Morphew E, Jones R, Gottfried MR, Seigler HF: Heterotopic ossification in rectal cancer: Rare finding with a novel proposed mechanism. J Surg Oncol. 2003, 82: 132-136. 10.1002/jso.10181.View ArticlePubMedGoogle Scholar
- Hara H, Iwabuchi K, Shinada J, Yoshimura H, Kameya T: Pulmonary adenocarcinoma with heterotopic bone formation. Pathol Int. 2000, 50: 910-913. 10.1046/j.1440-1827.2000.01125.x.View ArticlePubMedGoogle Scholar
- Kijima Y, Umekita Y, Yoshinaka H, Owaki T, Sakamoto A, Yashida H, Aikou T: A case of breast carcinoma with cartilaginous and osseous metaplasia. Breast Cancer. 2006, 13: 214-219. 10.2325/jbcs.13.214.View ArticlePubMedGoogle Scholar
This article is published under license to 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.