S-100 Negative Granular Cell Tumor of the Oral Cavity.

Classic granular cell tumor is a mesenchymal neoplasm that commonly occurs on the skin, but is not infrequently found in the oral cavity, primarily on the dorsal tongue. Diagnosis is usually straightforward with hematoxylin and eosin stained slides. Immunohistochemical studies on classic granular cell tumor shows positive immunostaining for S-100 and vimentin, while CD68 is variably positive. We report a case of otherwise unremarkable oral granular cell tumor that was immunohistochemically negative for S-100, and positive for vimentin and CD68, and discuss the differential diagnosis. The results of the immunohistochemical studies in our case are compared with those of classic S-100 positive oral granular cell tumors, as well as cutaneous and oral S-100 negative granular cell tumors. Classic S-100 positive granular cell tumors and S-100 negative granular cell tumors of the oral cavity can only be distinguished by immunohistochemical studies; however, the necessity of this distinction is unclear, as both are benign lesions in which recurrence is unlikely.

Vulvovaginal-gingival Lichen Planus: Association with Lichen Planopilaris and Stratified Epithelium-specific Antinuclear Antibodies.

Vulvovaginal-gingival lichen planus (VVG-LP) consists of a triad of symptoms: vulval, vaginal and gingival lichen planus lesions. The aim of this study was to analyse the prevalence of lesions in various anatomical locations in patients with VVG-LP. The study included 126 consecutive patients with lichen planus. Sixteen (12.7%) patients fulfilled the criteria of VVG-LP. In 12/16 (75%) patients with VVG-LP scalp lesions were also observed. Stratified epithelium-specific antinuclear antibodies (SES-ANA) and anti-ΔNp.3α antibodies were detected in 10/16 (75%) patients with VVG-LP and in 15/110 (13.6%) patients with other forms of lichen planus (p < 0.05). In conclusion, VVG-LP is frequently associated with lichen planopilaris. The new entity may be termed "vulvovaginal-gingival-pilar lichen planus" and our study indicates that SES-ANA is a marker of this type of lichen planus with extensive, severe and refractory-to-therapy involvement of the mucous membranes, skin and scalp.

Retrospective analysis of nuclear protein in testis (NUT) midline carcinoma in the upper aerodigestive tract and mediastinum.

OBJECTIVE: Nuclear protein in testis (NUT) midline carcinoma (NMC) is a very aggressive tumor with limited survival, recently recognized as a subset of poorly differentiated squamous cell carcinoma. A simple chromosomal translocation results in NUT overexpression and malignant transformation. This study used immunohistochemistry to retrospectively diagnose and characterize NMC cases. STUDY DESIGN: Immunoperoxidase staining was performed according to a standard protocol and interpreted independently by two pathologists. Scores were based on nuclear staining with monoclonal NUT antibody (C52B1) in the tumor cells. RESULTS: Fifty-one poorly differentiated carcinoma cases with material available for testing were retrieved. Average patient age was 54.9 years (range: 16-82), with 20 women and 31 men. A single NMC case (2%) was retrospectively diagnosed in a 26-year-old man with a left maxillary sinus/nasal cavity tumor; he died of his disease 18 months after presentation, despite treatment. CONCLUSIONS: These results support inclusion of NUT antibody in diagnostic immunohistochemical panels for poorly differentiated carcinomas of the upper aerodigestive tract.

Localized juvenile spongiotic gingival hyperplasia: a report of 3 cases.

Localized juvenile spongiotic gingival hyperplasia (LJSGH) is a recently described benign condition that affects the gingiva of children and young adults. Clinically distinctive, LJSGH presents as a localized area of erythema on the attached gingiva, with a subtly papillary surface architecture. The lesions are generally biopsied because of the lack of resolution with conservative oral hygiene therapeutic measures and esthetic concerns. The histopathology has a characteristic appearance of subtle papillary epithelial hyperplasia, acute inflammation, and numerous engorged capillary vascular spaces in the lamina propria, although clinical correlation is necessary to make the diagnosis. The purposes of this paper were to: introduce localized juvenile spongiotic gingival hyperplasia to the pediatric dental community; document its clinical and histologic features and treatment and the follow-up of three cases; and discuss the most common clinical differential diagnoses.

Lymphadenoma of the salivary gland: clinicopathological and immunohistochemical analysis of 33 tumors.

Lymphadenomas (LADs) are rare salivary gland tumors. Their clinicopathologic characteristics and etiopathogenesis are poorly understood. We examined 33 LADs in 31 patients (17 women and 14 men) aged 11-79 years (median 65 years). There were 22 sebaceous LADs in 21 patients (9 women and 12 men) and 11 non-sebaceous LADs in 10 patients (8 women and 2 men). Two patients had synchronous double tumors. Twenty-six tumors (79%) arose in parotid, three in the neck, and two each in submandibular gland and oral cavity. Extraparotid tumors were seen in 2 of 21 (10%) patients with sebaceous and 4 of 10 (40%) patients with non-sebaceous LADs. Seven of twenty-three (30%) patients had immunosuppressive therapy for unrelated diseases. The tumors were well circumscribed, encapsulated (n=28, 84%) painless masses, varying in size from 0.6 to 6 cm (median 2.2). The cut surfaces were gray-tan to yellow, homogeneous and multicystic (n=24, 72%). The epithelial cells were basaloid, squamous and glandular, forming solid nests, cords, tubules, and cysts. Sebaceous differentiation was restricted to sebaceous lymphadenoma. The epithelial cells expressed basal cell markers (p63, 34BE12, and/or CK5/6, 18/18, 100%) and the luminal glandular cells expressed CK7 (12/12, 100%). Myoepithelial cells were absent (n=10/16, 63%) or focal. The lymphoid stroma was reactive, with germinal centers in 28 (84%). There was no evidence of HPV (0/11), EBV (0/7), and HHV-8 (0/8). Malignant transformation to sebaceous and basal cell adenocarcinoma was seen in one patient each. None of the 11 patients with follow-up (1-8 years) recurred. In summary, sebaceous and non-sebaceous LADs are benign, encapsulated, solid and cystic tumors affecting older adults. Non-sebaceous LADs affect women and extraparotid sites more frequently than sebaceous LADs. Altered immune status may have a role in their etiopathogenesis. Multiple synchronous tumors, origin in buccal mucosa, and malignant transformation may rarely occur.

Chronic ulcerative stomatitis: evidence of autoimmune pathogenesis.

OBJECTIVES: Chronic ulcerative stomatitis is a condition characterized by chronic, painful oral ulcers, whose pathogenesis is unknown. Patients demonstrate specific IgG autoantibodies against ΔNp63α, an epithelial nuclear transcription factor. The aim of this study was to investigate the role of patient autoantibodies in the disease pathogenesis. METHODS: Three-dimensional in vitro tissues consisting of a fully differentiated, multilayer epithelium that mimics its in vivo counterpart were incubated with serum from patients with chronic ulcerative stomatitis. RESULTS: Our results show a subepithelial detachment of the epithelium at the basement membrane interface, mimicking the oral ulcerations that are seen clinically. Expression of basement membrane proteins Type IV collagen and laminin-5 was unaltered, whereas the expression of α6ß4 integrins, hemidesmosome components that attach basal keratinocytes to the basement membrane, was reduced, as determined by immunohistochemistry. CONCLUSION: These results give evidence that patient autoantibodies are pathogenic; and support an autoimmune pathogenesis in chronic ulcerative stomatitis.

A clinico-pathologic correlation: primary Ewing's sarcoma of the mandibular body-ramus.

A 15-year-old Asian male was referred to the department of oral and maxillofacial surgery at Tufts University School of Dental Medicine (TUSDM) for evaluation of a radiolucent lesion on the posterior left mandible. The patient was initially evaluated by his primary dentist for pain of two months' duration associated with teeth #17 and 18. Radiographic examination was performed and the panoramic radiograph revealed a radiolucent lesion involving the left mandibular body and ascending ramus, along with resorption of the roots of the predetermined teeth. Considering odontogenic infection, the patient was placed on antibiotics and referred to TUSDM for further evaluation and treatment.

Sensitivity of direct tissue fluorescence visualization in screening for oral premalignant lesions in general practice.

Various specialty clinics and research centers have conducted studies of direct tissue fluorescence visualization as a screening technique for oral premalignant lesions and early oral squamous cell carcinoma (OSCC). The effectiveness of the VELscope in a private practice setting is unknown. This pilot study is the first report to assess the VELscope system as a screening adjunct among lower-risk populations seen by a primary care clinician in a general practice setting. This study involved a retrospective comparison of two oral cancer screening examination protocols conducted on a presumably low-risk patient population seen in a private general dentistry practice. For one year, all patients age 12 or older received oral examinations, according to a standard oral cancer screening protocol. The following year, the same population was examined according to the same protocol with the addition of direct tissue fluorescence visualization using the VELscope. Screening with incandescent light examination yielded a prevalence of mucosal abnormalities of 0.83%, none of wich were premalignant. Screening with incandescent light examination combined with direct tissue fluorescence visualization yielded a 1.3% prevalence of mucosal abnormalities; based on surgical biopsy and histopathologic examination, 83% of these were potentially premalignant epithelial dysplasia.

Plasma cell mucositis of the oral cavity: report of a case and review of the literature.

Plasma cell mucositis (PCM) is a rare plasma cell proliferative disorder of the upper aerodigestive tract with an unknown etiology. Including the present case, only 22 cases have been reported in the English-language literature. PCM affects adult patients at an average age of 56.6 years. Clinical features are an intensely erythematous mucosa with papillomatous, cobblestone, nodular, or velvety surface changes. Symptoms include oral pain of long duration, dysphagia, persistent hoarseness, and pharyngitis. The majority of cases have a history of autoimmune or immunologically mediated disease. The histopathologic features of a dense, submucosal plasma cell infiltrate are not specific and must be differentiated from other reactive and neoplastic conditions. Diagnosis of PCM depends on clinical pathologic correlations. The present case was complicated by evidence of a monoclonal plasma cell population. The significance of this case is the differentiation of a benign disease from one that is potentially life threatening.

Ewing sarcoma of the mandibular condyle: multidisciplinary management optimizes outcome.

BACKGROUND: Ewing sarcoma (ES) is a rare, primary malignancy of bone that occurs in childhood and early adolescence. Improved methods of diagnosis and treatment have dramatically increased survival over the last 20 years. Treatment mainstays are chemotherapy and surgical tumor resection. ES usually occurs in long bones of the axial skeleton; however, it may rarely arise in facial structures, particularly the mandible. In these cases, resection presents a challenging postsurgical reconstruction. METHODS AND RESULTS: We present the clinical findings and management of a case of ES that developed in the left mandibular condyle of a 15-year-old female. Chemotherapy and segmental mandibulectomy were used to achieve local control. An innovative temporomandibular joint reconstruction was successfully accomplished using a microvascular fibular free flap and conchal cartilage graft. CONCLUSION: Multidisciplinary management in diagnosis, treatment, and restoration of function produced an optimal result that eliminated disease and preserved aesthetics and quality of life.

The prevalence and significance of anatomic variance in the mandibular symphysis: a retrospective study.

As computerized tomography (CT) becomes more widespread in implant treatment planning, dentists are bound to encounter more uncharacteristic anatomical conditions in the jaws. This retrospective study examined the prevalence of one such anatomic variance on 78 CT scans of human mandibles. Irregular heterogeneous circular radiopacities with internal radiolucencies were present in a majority (97.4%) of the CT scans examined; however, the phenomenon only appeared pronounced in 28.9% of this group. Bone radiodensity was higher in the regions with the phenomenon as compared with adjacent bone without the phenomenon, which suggested no adverse indications for implant placement.

Differentiation of mucous membrane pemphigoid subgroups with confocal imaging.

OBJECTIVE: Mucous membrane pemphigoid is an immune-mediated subepithelial blistering disease consisting of immunologically heterogeneous subgroups. Differentiation between these subgroups is important because they differ in prognosis. This study uses oral mucosal pemphigoid specimens to investigate the utility of computer-aided fluorescence overlay antigen mapping and laser scanning confocal microscopy to differentiate subgroups of mucous membrane pemphigoid. STUDY DESIGN: Thirty oral mucosal biopsy specimens were cryosectioned and immunostained, although only 13 could be analyzed due to technical difficulties. In vivo bound antibodies and molecular markers of the basement membrane zone were differentially labeled with fluorescent antibodies. Fluorescent signals were imaged, and the spatial localization of in vivo bound antibodies was compared with the markers and analyzed. RESULTS: In vivo bound IgG antibodies colocalized with beta4-integrin in 3 cases, with laminin-5 in 8 cases, and with collagen VII in 2 cases. CONCLUSION: Fluorescence overlay antigen mapping and laser scanning confocal microscopy are useful techniques to differentiate pemphigoid subgroups in oral biopsy specimens.

Clinical and immunopathologic findings in oral lichen planus pemphigoides.

Lichen planus pemphigoides (LPP) is a rare, acquired, immunobullous disorder of skin that occasionally involves oral mucous membranes. Clinical, histologic, and immunopathologic findings of the oral manifestations of LPP are described. Clinical features are lichenoid striae, erosions, and ulcerations involving gingiva and buccal mucosae. Histopathologic features are similar to those of ora lichen planus. Direct immunofluorescence demonstrates linear deposits of immunoglobulin G and complement component C3 along the basement membrane with fibrillar deposits of fibrin at the epithelial/lamina propria junction. Fluorescence overlay antigen mapping and laser scanning confocal microscopy of the biopsy specimen exhibits colocalization of in situ antibodies with beta4 integrin, a marker of the keratinocyte basal plasma membrane and upper lamina lucida, consistent with the location of the bullous pemphigoid antigens. This case report describes a case of LPP that presented exclusively as an oral condition. Lichen planus pemphigoides should be considered in the clinical differential diagnosis of vesiculoerosive oral mucosal diseases.

Necrotizing sialometaplasia associated with bulimia: case report and literature review.

Necrotizing sialometaplasia (NSM) is a self-limiting disorder affecting mainly the minor salivary glands. The significance of NSM resides in its clinical and histopathological resemblance to carcinoma. Few cases of NSM associated with eating disorders have been reported to date. We present here the clinical features and histomorphology of an additional case of bulimia-associated NSM closely mimicking an invasive carcinoma. A high index of suspicion and good communication between clinician and pathologist are essential in recognizing this entity and preventing unnecessary surgical therapy.

A diagnostic challenge: anterior variant of mandibular lingual bone depression.

Mandibular lingual bone depressions are considered to be developmental anomalies and most clinicians are familiar with the posterior variant (known as Stafne's bone cavity) that occurs in lingual mandibular molar regions, adjacent to the submandibular gland and below the mandibular canal. The anterior variant of the mandibular lingual bone depression is an asymptomatic bone cavity that occurs adjacent to the sublingual gland in the anterior mandible. Radiographically, it appears as a well-corticated radiolucency that retains a normal trabecular pattern internally. CT imaging is diagnostic and avoids surgical and sialographic procedures. This article reviews radiographic and CT features of a case involving the anterior variant of mandibular lingual bone depression.