Intravascular papillary endothelial hyperplasia of the oral cavity

To examine the pathogenesis of intravascular papillary endothelial hyperplasia (IPEH), a relatively uncommon benign, non-neoplastic vascular lesion, clinicopathological and immunohistochemical studies were performed. Paraffin-embedded tissue specimens of 78 vascular lesions were examined histologically, and 9 cases of IPEH were investigated immunohistochemically using antibodies against CD34, vimentin, factor VIII antigen, α-smooth muscle actin (α-SMA), podoplanin, CD105, and ki-67 antigen. A thrombus or ulcer was found near the sites of all IPEH specimens. Histologic examination revealed papillary proliferated endothelial cells located toward the lumen of enlarged blood vessels. Immunohistochemistry showed that CD34, α-SMA, and factor VIII antigen were positive in lining endothelial cells. Vimentin was positive in the mesenchymal components. Immunohistochemical staining for podoplanin and CD105 was partially positive. Labeling index was 4.7 to 9.2 in ki-67-positive cases. IPEH is believed to result from reactive proliferation of blood endothelial cells that is caused by an abnormal process of organization in thrombosed blood vessels. The pathogenesis of IPEH might be related to inflammation or mechanical stimulus such as irritation.     

Non-functioning adrenal cortical carcinoma presenting with metastasis to the tongue

We report a case of non-functioning adrenal cortical carcinoma (ACC) presenting with metastatic disease to the tongue, which is an extremely uncommon onset for this neoplasm. Histologically, the lesion had the appearance of an anaplastic neoplasm, and a panel of immunohistochemical markers including vimentin, MART-1, S100 protein, HMB-45, smooth muscle actin, common muscle actin, desmin, CD31, CD34, CD68, EMA and cytokeratins, was helpful in excluding melanoma, as well as other mesenchymal and epithelial neoplasms.     

Solitary fibrous tumour of the oral cavity: clinicopathological and immunohistochemical characterization of three cases

Solitary fibrous tumour (SFT) is an uncommon mesenchymal neoplasm rarely located in the oral cavity. To characterize further oral SFT, we describe three new cases. Each tumour originated in the buccal mucosa of a middle-aged/elderly patient. Histological examination showed well-circumscribed tumours with densely cellular areas alternating with hypocellular areas in a variedly collagenous, vascular stroma. Mast cells were abundant. The spindle-shaped, neoplastic cells immunostained strongly for CD34 antigen and vimentin and weakly for bcl-2, but not for epithelial cell markers, alpha-smooth muscle actin, or neurofilament or S-100 proteins. Compatible with the virtual absence of mitoses and of marked nuclear atypia, the overall frequency of proliferating cells expressing Ki-67 was low. The expression of CD34 was useful in the differential diagnosis. The consistent location in the cheek and expansion of one tumour after local trauma does not preclude a traumatic element in the development of oral SFT.     

Fibrosarcoma originating in the mandible

Fibrosarcoma is a malignant mesenchymal neoplasm of fibroblasts that rarely affects the oral cavity and can cause local recurrences or metastasis. In this article we describe a case of primary fibrosarcoma in the mandible in a 16-year-old girl. Microscopically the tumor showed an intense proliferation of spindle-shaped cells, varying little in size and shape and arranged in parallel bands, partly crossing each other, the mitotic activity was increased and there was nuclear pleomorphism. Immunohistochemically the cells only showed immunoreactivity for vimentin and negativity for S-100 protein, CD 68, cytokeratin cocktail, HMB-45, CD34, pan actin HHF 35, desmin, smooth muscle actin and epithelial membrane antigen (EMA). Based on clinical, radiological, histological and immunohistochemical findings the final diagnosis was high-grade intra-osseous fibrosarcoma. The treatment choice was radical surgery with mandibular reconstruction. After one year and nine months of the treatment the patient displayed multiple metastases. Radiation therapy and chemotherapy were used as adjuvant treatment. Unfortunately, the girl died two years after initial diagnosis.     

Immunocytochemical study of giant cell fibroma

Giant cell fibroma (GCF) is a non-neoplastic lesion of the oral mucosa. The origin of stellate and multinucleate cells of GCF is not well known. The purpose of the present article was to investigate the immunoreactivity of these cells for leukocyte common antigen, vimentin, tryptase, HLA-DR, alpha-smooth muscle actin, CD68, and S-100. The results showed positive staining only for vimentin. This suggests that the stellate and multinucleate cells of GCF have a fibroblast phenotype.     

Benign fibrous histiocytoma in the condylar process of the mandible: Case report

A 48-year-old man was referred for investigation of an asymptomatic radiolucent lesion in the mandible. The margin was partly irregular, and there was no peripheral sclerosis. The tumour was composed of histiocytic cells, spindle cells, and fibrous tissue. Immunohistochemical analysis showed that the tumour cells stained for CD68 and vimentin, and not for cytokeratin, smooth muscle actin, S-100 protein, or CD34. The tumour was therefore diagnosed as a benign fibrous histiocytoma.     

Angiomyolipoma of the upper lip: case report and review of the literature

Angiomyolipoma (AML) is a hamartomatous growth that usually affects the kidney. One third of patients with AML present with manifestations of tuberous sclerosis. Oral AML is rare with only 6 cases reported in the English-language literature. In the present case, AML was located in the upper lip of a 43 year-old woman. Clinically, it presented as a firm nodule, well circumscribed and measuring 1 x 2 cm. It was surgically excised. Histopathological analysis showed a lesion composed of an admixture of smooth muscle cells, blood vessels, and adipose tissue. The immunohistochemical study revealed positivity for vimentin, smooth muscle actin, pan specific muscle actin and desmin. CD68, CD34 and mast cell antibodies showed focal immunoreactivity. S100 protein, Ki-67, and HMB-45 were negative. Based on these histological and immunohistochemical features the diagnosis was of oral AML. No recurrence was observed after 2 years of follow-up.     

粘液表皮样癌组织发生的免疫组织化学研究

为探讨涎腺液表皮样癌的组织发生,本文采用三种特异性抗体:上皮肤抗原(EMA);肌动蛋白(actin);波形蛋白(vimentin)对25例涎腺粘液表皮样癌进行免疫组织化学研究。结果发现,粘液表皮样癌中三种肿瘤细胞对EMA反应阳性,部分表皮样细胞和中间细胞对actin和Vimentin反应阳性,粘液细胞反应阴性。研究结果表明,粘波表皮样癌是上皮源性的肿瘤,其中表皮样细胞和中间细胞具有肌上皮细胞的分化特征。提示,肿瘤性腺上皮与肿瘤性肌上皮细胞同时参与了该肿瘤的组织发生。     

Epithelioid hemangioendothelioma of the facial bone

The epithelioid hemangioendothelioma is an uncommon vascular neoplasm. Numerous sites of involvement are possible, but these tumors most commonly arise in the soft tissues, liver, and lung. We report a case of epithelioid hemangioendothelioma presenting as a single lesion on the nasal portion of the maxilla. Histologically, the tumor within bone and skin exhibited cords and nests of plump, epithelioid-appearing cells exhibiting rudimentary vascular differentiation within a myxohyaline stroma. Immunohistochemical reactivities for factor VIII-related antigen, CD31, CD34, and vimentin were demonstrated. Wide local excision with close clinical follow-up seems to be the treatment of choice for these tumors.     

Nodular fasciitis and solitary fibrous tumor of the oral region: tumors of fibroblast heterogeneity

OBJECTIVE: Fibroblastic proliferations of the oral cavity are extremely varied, yet they share certain features-spindle cell morphology, collagen synthesis, and fasciculation. Nodular fasciitis is a cellular fibroblastic lesion, uncommonly located in the oral submucosa, that shows smooth muscle actin (SMA) immunoreactivity. Solitary fibrous tumor expresses a CD34 fibroblast phenotype. The aim of this study is to report instances of nodular fasciitis and solitary fibrous tumor in the orofacial region and investigate immunohistochemical markers to compare and contrast fibroblastic phenotypic heterogeneity in these tumors. STUDY DESIGN: Seven benign cellular fibrogenic tumors initially diagnosed as nodular fasciitis over a 10-year period were examined. Immunohistochemical markers, including S-100 protein, SMA, CD68, CD34, and vimentin, were used to further characterize these lesions. RESULTS: All tumors occurred in adults, and the buccal mucosa was found to be the favored site. The spindle cells in these tumors showed phenotypic heterogeneity both within and between tumors. All were vimentin-reactive and harbored small populations of CD68-positive macrophage/dendrocytes. Five tumors were SMA-positive and CD34-negative; the tumor in one case was SMA-negative and CD34-positive, and that in another was SMA-positive and CD34-positive. CONCLUSION: Although rare, nodular fasciitis and solitary fibrous tumor arise in oral submucosa, usually in the cheek. The histopathologic features and immunomarkers indicative of myofibroblastic differentiation are seen in nodular fasciitis, whereas solitary fibrous tumor is CD34-positive; however, one instance was found to be positive for both markers. All of these cases harbored subpopulations of CD68-positive cells. Immunomarkers are a valuable adjunct in differentiating nodular fasciitis from solitary fibrous tumor, yet some tumors may harbor heterogeneous fibroblast phenotypes.     

Peripheral giant cell granuloma. An immunohistochemical and ultrastructural study

OBJECTIVE: To study the nature of multinucleated and mononuclear cells from peripheral giant cell granuloma (PGCG).
MATERIALS AND METHODS: Formalin-fixed, paraffin-embedded sections of 40 cases of PGCG were immunohistochemically stained for vimentin, alpha I-antichymotrypsin, CD68, S-100 protein, lysozyme. leucocyte common antigen (LCA), factor VIII-retated antigen and muscle cell actin. Six cases of PGCG were also studied by transmission etectron microscopy.
RESULTS: Vimentin, alpha I-antichymotrypsin and CD68 were expressed in both the mononuclear and multinucleated giant cells. Dendritic mononuclear cells, positive for 5–100 protein, were noted in 67.5% of the lesions, whereas lysozyme and leucocyte common antigen were detected in occasional mononuclear cells. Ultrastructural examination showed mononuclear cells with signs of phagocytosis and sometimes interdigitations with similar cells. Others presented non-specific characteristics and the third type exhibited cytoplasmic processes and occasional Birbeck granules. Some multi-nucleated giant cells showed oval nuclei, abundant mitochondria and granular endoplasmic reticulum whereas others presented with irregular nuclei and a great number of cytoplasmic vacuoles.
CONCLUSIONS: lmmunohistochemical and ultrastructural results suggest that PGCGs of the jaws are composed mainly of cells of the mononuclear phagocyte system and that Langerhans cells are present in two thirds of the lesions.     

Intraoral superficial angiomyxoma

Superficial angiomyxoma is an unusual benign myxoid tumor of the trunk, head and neck, extremities, and genitalia. We report the third case of intraoral superficial angiomyxoma, which occurred within the buccal mucosa of a 37-year-old woman. The symptomless slow-growing mass was present for 2 years. The lobulated, paucicellular myxoid tumor showed prominent vascularity, stromal inflammation, including neutrophils, and immunopositivity for CD34, vimentin, and muscle-specific actin. Cure is effected by localized excision, with follow-up, owing to the high recurrence rate of the cutaneous tumors, which has not been substantiated for the few reported intraoral cases. Superficial angiomyxoma does indeed occur intraorally and should be included in the differential diagnosis of myxoid intraoral soft tissue neoplasms.     

Phenotypic characterisation of stellate and giant cells in giant cell fibroma by immunocytochemistry

The origin of the stromal, stellate and multinucleate cells in oral giant cell fibroma is unclear. Sixteen giant cell fibromas were stained immunocytochemically for keratin (MNF 116), vimentin, S-100 protein, neurofilaments, glial fibrillary acidic protein, α-smooth muscle actin, desmin, CD31 (PECAM-1). CD68, Factor XIIIa and prolyl 4-hydroxylase (5B5). In all cases positive staining was found with vimentin and prolyl 4-hydroxylase, indicating a functional fibroblast phenotype. Reactivity for Factor XII la was seen in two cases and in only one was a small number of giant cells stained, suggesting that the majority of oral giant cell fibromas are unrelated to the histologically similar fibrous papule of the nose or facial angiofibroma.     

Inflammatory myofibroblastic tumor of the alveolar mucosa of the mandible

Inflammatory myofibroblastic tumor is a rare lesion composed of myofibroblastic spindle cells accompanied by inflammatory infiltrate. The objective of this paper is to report an uncommon case of inflammatory myofibroblastic tumor located in the alveolar mucosa of the mandible. A 33-year-old male presented an asymptomatic tumoral lesion, firm, pedunculated, pink-colored, covered by smooth mucosa, with focal ulceration, measuring 30x20x20 mm, located in the left posterior alveolar mucosa. Clinical diagnosis was soft tissue tumor. An excisional biopsy was made. Microscopic examination showed compact fascicular spindle cells proliferation with a diffuse inflammatory infiltrate of plasma cells, lymphocytes, and eosinophils. Large ganglion-like cells were observed. The lesional cells were immunopos-itive to vimentin, a-smooth muscle actin, muscle specific actin, and CD68. Negative immunostain was observed to S-100, Bcl-2, Ki-67, desmin, CD34, and cytokeratin. A diagnosis of inflammatory myofibroblastic tumor was performed. After 28 months of follow-up there was no recurrence. Although no evidence of oral inflammatory myofibroblastic tumor recurrence or malignant transformation has been reported, it has been observed that in inflammatory myofibroblastic tumor of other regions a prolonged follow-up is necessary after surgical excision.     

CD34 expressing ameloblastic fibrosarcoma arising in the maxilla: a new finding

Ameloblastic fibrosarcoma (AFS) is a rare malignant tumor of the jaw. The malignant mesenchymal component of AFS has been described as 'fibroblast-like', although little is known about the immunophenotype, except for vimentin expression. Here, we present a case of AFS in a 62-year-old woman. The mesenchymal component displayed the features of either dermatofibrosarcoma protuberans or fibrosarcoma, and was positive for CD34. This is the first reported case of CD34 expressing AFS in the maxilla.     

Endovascular papillary angioendothelioma (Dabska tumor) of the tongue: report of a case

Endovascular papillary angioendothelioma (EPA), known as Dabska tumor, is a very rare vascular neoplasm that usually occurs in the skin or subcutis of infants and young children. There is no previous report of EPA appearing in the oral cavity. Here, we present an exceptionally rare case of EPA of the tongue in a 67-year-old man. A well-defined, reddish tumor measuring 11 mm x 8 mm x 7 mm existed at the submucosal area of the left tongue margin. Microscopic examination of an excisional biopsy specimen revealed the endothelioid tumor cells showing a papillary growth pattern, such as blood vessel-like structures. Immunohistochemical studies showed positive reactivities for CD31, CD34, alpha-smooth muscle actin, and factor VIII-related antigen in most of the tumor cells and CD68 in some tumor cells. Based on these observations, the tumor was diagnosed as an EPA.     

Central giant cell granulomas of the jaws: phenotype and proliferation-associated markers

Central giant cell granulomas (CGCGs) are jaw tumors of unknown origin that often exhibit an aggressive, though unpredictable, clinical course. The purpose of this study was to determine the immunoprofile of the mononuclear cells that seem to be responsible for the biologic behavior of these tumors. Numbers of cells in cell cycle were also determined and compared in clinically aggressive and non-aggressive CGCGs. Sixteen aggressive and 12 non-aggressive CGCGs were immunohistochemically stained with antibodies to CD34, CD6S. factor XHIa, ct-smooth muscle actin. prolyl 4-hydroxylase, Ki-67. and p53 protein. Cell populations and numbers of cells in cell cycle were determined through microscopic quantitative assessment. CD34-positive ceils were limited to support vessels. CD68-positive mononuclear cells constituted a small population of cells in ail tumors. With two exceptions, factor XIIIa-positive cells were rarely seen. Alpha-smooth muscle actin staining was present in approximately half the tumors, and occasionally large numbers of positive cells were seen. Most mononuclear cells were positive for fibroblast-associated antigen. No phenotypic differences were detected between aggressive and non-aggressive tumors. P53 protein did not appear to be overexpressed in CGCGs. Ki-67 staining showed that only mononuclear cells were in cell cycle, and that there were no differences between aggressive and non-aggressive tumors. We conclude that CGCGs are primarily fibroblastic (and myofibroblastic) tumors in which macrophages appear to play a secondary role. Tumor cells show no differentiation toward endothelial cells or macrophage-related dendrocytes (factor XIIIa). Cellular phenotypes and numbers of cells in cell cycle are similar in both aggressive and non-aggressive tumors.     

Intraosseous epithelioid hemangioendothelioma of the mandible: a case report with an immunohistochemical study

Epithelioid hemangioma is the prototype of a group of vascular tumors characterized by epithelioid endothelial cells. Hemangioendothelioma of bone is a rare lesion that constitutes less than 0.5% of primary malignant skeletal tumors. We report and discuss a case of epithelioid hemangioendothelioma arising intraosseously in the anterior portion of the mandible in a 76-year-old woman. The case was treated successfully by wide resection. Radiographically, the tumor mass showed osteolysis and expansion. Histologically, the tumor showed invasive and destructive growth, although it lacked frequent mitotic figures and severe atypia. On immunohistochemical study, tumor cells exhibited characteristics of mesenchymal and endothelial origin, i.e., strong to moderate immune reactivity against vimentin, factor VIII-related antigen (F8RA), Ulex europaeus agglutinin type 1 lectin (UEA-1), and CD 34, but not against keratin, epithelial membrane antigen (EMA) or S-100 protein (S100). The proliferating cell nuclear antigen (PCNA)-positive cell index was 27.5%. These pathological findings suggested a borderline malignant potential for this tumor. Thus, clinically, wide resection with or without dissection of regional lymph nodes is recommended.     

Actin versus vimentin in myoepithelial cells of salivary gland tumors: A comparative study

Vimentin versus actin expression was immunohistochemically studied in myoepithelial cells of 24 salivary gland tumors in which the participation of myoepithelial cells as a tumoral component has been postulated: two basal cell adenomas, seven pleomorphic adenomas, two myoepitheliomas, seven adenoid cystic carcinomas (two tubular, four cribriform, one solid), six polimorphous low-grade adenocarcinomas. Immunostaining was carried out in formalin-fixed tissue serial sections (3 μm) by the avidin-biotin method, using the antibody vimentin (Dako Corp., Carpenteria, Calif.) and the antibody HHF35 anti-muscle actin (Enzo Biochemical, N.Y.). Our results have confirmed positive staining for vimentin in all salivary tumors studied, although in some tumors it was only in focal areas. The staining for the HHF35 antibody to muscle actin was only consistently found in the adenoid cystic carcinomas of the tubular and cribriform patterns. This study suggests that actin is at least somewhat replaced by vimentin in neoplastic tumoral cells. Therefore vimentin can be used to define the participation and distribution of myoepithelial cells in these tumors.     

原发咽旁滑膜肉瘤1例

本文报告1例咽旁滑膜肉瘤,患者男,15岁,以左咽旁无痛性逐渐增生包块1月就诊。专科检查发现左侧咽旁有一大小约8 cm×4 cm×3 cm肿物,蒂部位于左扁桃体上隐窝。影像学检查见左侧口咽不规则肿块,与左舌根后份分界不清,增强扫描明显不均匀强化。免疫组织化学检查：上皮细胞膜抗原（+）,细胞角蛋白（CK）19（+）,CD7（+）,波形蛋白（+）,CK10（-）,钙黏蛋白（+）,B淋巴细胞瘤-2（-）,CD2（-）,CD10（-）,CD138（+）,CD99（+）,白细胞共同抗原（+）,Ki-67（20%+）。病理诊断为咽旁滑膜肉瘤。