Primary intraoral leiomyosarcoma of the tongue: an immunohistochemical study and review of the literature (original) (raw)
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Oral leiomyosarcomas: report of two cases with immunohistochemical profile
Oral Surgery Oral Medicine Oral Pathology Oral Radiology and Endodontology, 2007
Leiomyosarcoma of the oral cavity is a very rare tumor associated with aggressive clinical behavior and low survival. In this paper, we report 2 cases of leiomyosarcoma, affecting the gingival mucosa of a 54-year-old female and the maxillary bone of a 63-year-old male. Histologically, the tumors were composed of variably oriented fascicles of spindle-shaped cells with cigar-shaped nuclei and eosinophilic cytoplasm. The lesions were treated by surgical resection. Immunoreactivity to anti-vimentin, anti-smooth muscle actin, anti-desmin, anti-laminin, and anti-musclespecific actin antibodies were found; conversely, the tumor cells were negative for anti-S100 and AE1/AE3 proteins. This report emphasizes the role of immunohistochemical study for correct diagnosis of leiomyosarcoma. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:e50-e55)
2013
Purpose: Leiomyosarcoma (LMS) rarely occurs in the head and neck region. These tumors present with a wide range of clinical features, so the diagnosis is predicated on conventional microscopic findings coupled with immunohistochemical analysis. Patients and Methods: Clinical and histologic data of 7 patients with LMS of the head and neck were recorded retrospectively. In addition to routine immunohistochemistry, staining for cell cycle regulator proteins p16 and p21 was performed. Results: Five LMSs (4 intraoral, 1 dermal cheek) occurred primarily in the oral and perioral region. Two LMSs (parietal and sinonasal) were diagnosed as metastases originating from the uterus and pelvis. Treatment of the primary LMSs consisted of radical tumor resection with clear margins. Distant metastases from LMSs were irradiated or excised as palliative treatment. Three of 5 patients (60%) with primarily excised LMS developed recurrence after an average of 7 months, with lung metastases occurring after 17 months. In 1 patient, cervical lymph node metastases were detected after 10 months. Of all patients, 5 died after an average survival period of 2.4 years. The mean survival period of the 5 patients with primary LMS of the head and neck was 3.3 years. All tumors were positive for vimentin and a-smooth muscle actin, with 57% of tumors showing positive nuclear expression of p16 and 71% of p21. Lack of p16 nuclear expression was associated with a shorter mean survival time (1.3 vs 4.3 yr for p16 positivity). Conclusion: Lung and cervical lymph node metastases often occur in LMS of the head and neck. Presurgical staging, including gynecologic examination, whole-body computed tomography, and sometimes positron-emission or computed tomography, to rule out LMS metastasis is mandatory. Surgical resection of the tumor should be given top priority. Lack of p16 reactivity may have a prognostic value for LMS because it was related to a trend toward poorer survival.
Oral Oncology, 2002
Leiomyosarcoma of the oral cavity is a very rare tumor that is associated with aggressive clinical behavior and low survival. In this paper, we report two new cases of leiomyosarcoma affecting the mandibular gingiva and mandible of a 35-year-old male and the mandible of a 51-year-old female. Given the difficulty in the histopathologic discrimination between benign and malignant smooth muscle tumors and the absence of reliable histologic parameters for prognostication of leiomyosarcomas, we evaluated the diagnostic and prognostic value of various immunohistochemical and molecular markers. By means of immunohistochemistry and quantitative real-time PCR analysis, we detected protein expression of PCNA, bcl-2, CDK4, p53 and MDM2 in both our cases and MDM2 amplification in our second case. The literature, pertinent to oral leiomyosarcoma and to molecular analysis of smooth muscle tumors, is reviewed. #
Leiomyosarcomas of the oral cavity. Report of four cases and review of the literature
Journal of Cranio-Maxillofacial Surgery, 1993
SUMMAR Y. Leiomyosarcomas (LMS) make up 7 % of all soft tissue sarcomas. In the oral cavity, the LMS is rare due to the paucity of smooth muscle in that region. Four cases of intraoral LMS are reported, plus 34 cases found in the literature reviewed. There were 24 males and 14 females. The age range at presentation was 10 months to 88 years with no predilection for any particular age group. The commonest presenting symptom was a mass. The intraoral LMS occurred most commonly in the jaws (59 % of cases). The mass was painful in 61% of cases. Followup of the case reports was assessed. Recurrence occurred in 36 % of cases. Distant metastases occurred in 39 % of cases, most commonly to the lungs. Cervical nodal metastasis was reported in 15 % of cases. The 5-year survival rate determined on cases with adequate follow-up (n = 13) was 23 % free of disease, 8 % alive with disease and 69 % who died of disease. In comparison with stage I and stage I1 tongue squamous cell carcinomas, the intraoral LNIS is very aggressive. It is best treated surgically, early and aggressively.
Lingual Leiomyosarcoma: A Histopathological Case Report
Cureus
Leiomyosarcomas are rare malignant mesenchymal neoplasms originating from smooth muscle cells. Although leiomyosarcoma is commonly located in the female reproductive system, gastrointestinal tract, and subcutaneous tissues, it is a rare entry in the head and neck area, probably due to the scarcity of smooth muscle tissue in this topographical region. Herein we present a histopathological case report of a 60-yearold male with a slow-growing painless mass on the middle third of the right lateral lingual margin, with focal ulceration measuring 1x1.5cm. After gross excision, histopathology revealed pleomorphic spindle cells, some with bizarre nuclei and abundant pathological mitotic activity with a tendency to grow in a fascicular pattern. As the patient had the demographic characteristic and risk factors for oral cavity squamous cell carcinoma, a spindle-shaped variety (sarcomatoid) variety was suspected, and immunohistochemistry with a broad set of antibodies was used to prove the histogenetic group of the tumor. As the tumor was pancytokeratin and desmin negative, focally positive for caldesmon, and positive for smooth muscle actin, the diagnosis of pleomorphic leiomyosarcoma of the tongue was established.
American Journal of Otolaryngology, 2005
Laryngeal leiomyosarcoma (LMS) is definitely considered a rare occurrence: our exhaustive literature review disclosed to date only 45 cases (including the new case we have reported). The morphological diagnosis of laryngeal LMS may be problematic on conventional light microscopy especially in small laryngeal specimens. In the past, LMSs of the larynx were frequently confused with other spindle-cell malignant tumors. Nowadays, immunohistochemical investigations are considered necessary to distinguishing LMS from other spindle-cell tumors. On immunostaining, LMS is usually positive for muscle-specific actin and negative for cytokeratins and epithelial membrane antigen. In most cases, immunohistochemical study allows reliable diagnosis of LMS, but it can produce ambiguous or inconclusive results when the tumor cells lack specific immunohistochemical reactivity. In these controversial cases, LMS diagnosis has to be substantiated by electron microscopy. Distinguishing laryngeal LMS from other spindle-cell malignancies plays a role not only in academic interest. Confusion in the diagnosis of a spindle-cell laryngeal malignancy could result in inappropriate clinical management and inadequate treatment. D
Leiomyosarcoma of the Tongue With Multiple Metastases: A Case Report and Review of Literature
Journal of Oral and Maxillofacial Surgery, 2012
Leiomyosarcoma of the tongue is an extremely rare malignant mesenchymal neoplasm and only 25 cases have been described in the English-language literature, 22 of which were primary lesions. A case is presented of a 54-year-old female patient diagnosed with primary leiomyosarcoma of the tongue with lung and multiple soft tissue metastases. This case is especially unusual because widespread soft tissue metastases resulted, involving the lung and skeletal muscle in 3 separate sites. Unfortunately, curative treatment was not possible. Chemotherapy using a combination of ifosfamide and doxorubicin was, therefore, provided as palliation, with a good response after a 3-week cycle of therapy. Currently available literature on primary and secondary leiomyosarcoma of the tongue was reviewed for a better understanding of this rare mesenchymal neoplasm. To the investigators' knowledge, there are no other reported cases in the literature that have previously been managed with chemotherapy alone.
Leimyosarcoma of the buccal mucosa and review of literature
Journal of Oral and Maxillofacial Pathology, 2013
Leiomyosarcoma (LMS) is an uncommon malignant spindle cell tumor of the head and neck region. The occurrence is particularly rare in the buccal mucosa of the oral cavity. It is a rapidly growing tumor with aggressive behavior and poor prognosis. Method: This article presents a rare case of primary leimyosarcoma of the buccal mucosa in a 35 year old female and retrospective analysis of primary oral LMS published in the English literature since past 20 years is done. Diagnosis was confirmed by immunohistochemistry profile showing positivity for vimentin, smooth muscle actin (SMA), high proliferative index displayed by Ki-67, focal positivity for pan-CK and negativity for S-100. Conclusion: Based on the presence of malignant spindle cells showing positivity for vimentin and SMA, a diagnosis of leiomyosarcoma was made.
Craniomaxillofacial Trauma & Reconstruction, 2016
Leiomyosarcomas (LMSs) are rare malignant mesenchymal tumors which show smooth-muscle differentiation. Most LMSs involving the oral tissues primarily affect the maxillary sinus, the maxillary or mandibular bone. We present a case of LMS of the mandibular alveolar mucosa, arising in a 12-year-old male child from Ethiopia. A malignant spindle cell-like neoplasm was diagnosed on clinical and radiographic findings as well as on incisional biopsy. The tumor was resected with wide margins. The following histopathologic examination with additional immunohistochemical studies secured the diagnosis LMS. Microscopically, the spindle-shaped tumor cells were arranged in an interlacing fascicular pattern and contained oval to elongated, blunt-ended (cigar-shaped) nuclei. The immunohistochemical examination showed immunoreactive tumor cells for vimentin, actin, desmin, and H-caldesmon, which is pathognomonic for LMS. Immunohistochemical studies are mandatory to differentiate the LMS from other si...
Annals of Diagnostic Pathology, 2002
Solitary fibrous tumors are rare in extrapleural sites and extremely rare in the oral cavity. We report a case of a solitary fibrous tumor arising from the tongue of a 70-year-old woman. The tumor measured 1.6 cm in maximum diameter and consists of spindle-shaped cells distributed in a haphazard pattern. Immunohistochemical studies show strong positivity for CD34 and bcl-2, and weak positivity for desmin. Smooth muscle actin and S-100 protein are negative. Electron microscopy shows uniform neoplastic spindle cells with mesenchymal features. The differential diagnosis for spindle cell neoplasms in the tongue is discussed.