Oral leiomyosarcomas: report of two cases with immunohistochemical profile (original) (raw)
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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. #
Oral Oncology, 2000
Leiomyosarcoma is a relatively uncommon mesenchymal tumor that exhibits smooth-muscle dierentiation. We report a new case of leiomyosarcoma involving the tongue of a 67-year-old male. Histologically, the tumor was composed of variably oriented fascicles of spindle-shaped cells with`cigar-shaped' nuclei and eosinophilic cytoplasm, containing occasional PAS-positive granules. Atypical mitotic ®gures and necrotic foci were frequently detected. Consistent desmin, a-smooth muscle-speci®c and sarcomeric actin, and vimentin immunoreactivity was demonstrated in the tumor cells, whereas cytokeratins, CD 30, CD 31, CD 34, CD 45, CD 68, EMA, GFAP, HMB 45 and S-100 protein were negative. The patient underwent wide surgical excision of the tumor and is alive and disease-free at a 5-year follow-up. This report emphasizes the diculties in the dierential diagnosis of these uncommon tumors in an intra-oral location. #
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.
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.
Gingival Leiomyosarcoma in a Young Woman: Case report and literature review
Sultan Qaboos University Medical Journal [SQUMJ]
Leiomyosarcoma (LMS) is a rare mesenchymal malignancy, of which 3–10% of cases occur in the head and neck region. We report a 22-year-old woman who was referred to the University Hospital of Siena, Italy, in 2016 with an ostensibly benign asymptomatic lump on the mandibular gingiva. The lesion grew rapidly, causing otalgia in the right ear. An excisional biopsy was performed and primary LMS was diagnosed histologically. Subsequently, the patient underwent radical re-excision of the perilesional mucosa, a partial bone resection and the extraction of four teeth. No recurrences or metastases were detectable at a 20-month follow-up. This report discusses the differential diagnosis of LMS with regards to other benign and malignant lesions and reviews the recent literature on primary and secondary oral LMS. Due to its innocuous clinical features—including its asymptomatic nature and presentation at a young age—this aggressive malignancy can go undetected; therefore, an early histopatholog...
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...
Leiomyosarcomas of the oral cavity: report of a radiation-associated and a metastatic case
Oral and Maxillofacial Surgery, 2011
Background Leiomyosarcoma is rare in the oral cavity, where it may arise as primary, radiation-associated, or metastatic tumor. This article reports two cases of oral leiomyosarcoma, discussing the range of clinicopathological features and the significance of these presentations. Case report One case is a radiation-associated leiomyosarcoma arising in the tongue of a 71-year-old male occurring 22 years after radiation therapy for tonsil squamous cell carcinoma that was surgically treated. The other one is a mandible metastasis from a retroperitoneal widespread leiomyosarcoma in a 69-year-old man, who was treated by surgery and chemotherapy but died from the disease. Discussion Post-radiotherapy sarcomas of the oral cavity and oral metastasis from soft tissue sarcomas are very uncommon, but based on patient's clinical history, they should be considered by oral health care providers in order to allow an early diagnosis and proper and timely management. Finally, to the best of our knowledge, this seems to be the first reported case of tongue leiomyosarcoma arising in a previously irradiated field.
Leiomyosarcoma of mandible: A case report and review of literature
groups, one wherein each tumor type is characterized by a unique simple recurrent genetic abnormality such as a chromosomal translocation and the other in which highly complex genetic abnormalities are present (1). LMS belong to the latter group and are malignant neoplasms of smooth muscle, which most frequently occur in the uterus or retroperitoneum but can occur throughout the body (1). Women are more affected than men because of the uterine association. The prognosis is poor, with a high percentage of recurrence or metastasis. The most common sites of metastasis include the lungs, bone, brain and the lymph node. This neoplasm is an extremely rare mesenchymal lesion in the oral cavity, with unusual bone location because of paucity of smooth muscle in that site (2). The immunohistochemical analysis for cytoskeletal proteins such as actin, vimentin, desmin, Ki-67 and ultrastructural examination has been widely used for the diagnosis of LMS (3, 4). The aim of this article is to describe a rare case of LMS of the mandible with immunohistochemical analysis that was useful in making the final diagnosis. CASE REPORT A 65-year-old woman reported to our institute with chief complaint of pain and swelling on right facial region since 5 years. Patient was apparently all right 5 years ago, and then she noticed a small swelling on same region which gradually increased to present size. There were no associated symptoms accept intermittent dull pain. On examination a growth was observed with multiple nodules extending from corner of the mouth to angle of mandible measuring about 10X7 cm in size (Fig 1). Intraorally the lesion appeared as homogenous mass over right vestibular area extending from central incisor to molar area with central necrosis at body region. On palpation swelling was soft to firm in consistency with no tenderness and skin was attached to underlying structure. No lymph nodes involvement was found. Otherwise patient was healthy and all hematological and biochemical investigations were under normal limits.
Medicina oral, patología oral y cirugía bucal, 2008
Leiomyoma, a benign neoplasia arising from smooth muscle is an uncommon neoplasia of the oral cavity. The most common histological subtype in the oral cavity is the vascular one. To supplement information on vascular leiomyoma of the oral cavity (VLOC), we present cases of VLOC describing their clinical, histological, and immunohistochemical characteristics. Case reports. Five cases of VLOC (3 females; 2 males) from the Clinical and Experimental Pathology Laboratory, Dental School, National Autonomous University of México, are included. The most frequent clinical characteristic of VLOC was a single, asymptomatic, slow growing nodule. The age average of the cases was 40.6, however 3 out of our 5 cases were < or = 40 years old at the moment of their diagnosis. The lesions were composed of fusiform cells arranged in bundles or fascicles. The neoplastic cells were characterized by eosinophilic cytoplasm and tapered nuclei. The presence of vascular spaces was prominent in all cases. T...
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.