Oral Surgery Oral Medicine Oral Pathology Oral Radiology最新文献

Introduction

GLI1-altered epithelioid soft tissue tumor is a recently recognized entity featuring GLII-1 fusions or amplifications. It has a predilection for the head and neck area, particularly in the tongue, and appears to have a malignant potential. Herein we report an additional case of GLI1-altered soft tissue tumor of the tongue in a patient with growth retardation and epilepsy.

Materials and Methods

A 9-year-old boy presented with a lobulated sessile reddish nodule at the dorsum of the tongue of unknown duration. His medical history was significant for growth hormone insufficiency and epilepsy with centrotemporal spikes. Magnetic resonance imaging showed an avidly enhancing lesion at the midline dorsum of the tongue. An incisional biopsy of the lesion was performed.

Results

Microscopic examination of the lesion revealed a multilobulated submucosal mass of epitheloid to ovoid cell proliferation with a perivascular distribution around delicate and branching blood vessels. The tumor cells are small to medium with clear to amphophilic cytoplasm. They are arranged in fascicles, cords, and reticular patterns and separated by richly vascular stroma with frequent prominent capillary-sized vasculature. Protrusion of tumor cells into vascular spaces is focally seen. Immunohistochemistry showed cytoplasmic positivity of the tumor cells to GLUT-1 and B-Catenin and negativity to S100, GFAP, Pan-CK, SMA, WT-1, CD-31, CD-34, and HMB-45. Ki-67 is around 10-15%. RNA-sequencing confirmed the presence of ACTB::GLI1 fusion.

Conclusion

GLI1-altered epithelioid soft tissue tumor is a unique neoplasm with unknown lineage and variable immunoreactivity. Therefore, molecular testing is indicated to confirm the diagnosis. GLI1-altered epithelioid soft tissue tumors have the potential for local recurrence and distant metastases; thus, it is best regarded as low-grade sarcoma and warrants a long-term follow-up after surgical excision.

Introduction

BFOLJ represent a heterogeneous group of conditions with overlapping clinico-radiographic and histopathologic features despite diverse etiopathogenesis and biologic behavior. Overall, BFOLJ are subclassified into cemento-osseous dysplasia (COD), (cemento-)ossifying fibroma (OF) and fibrous dysplasia (FD). Herein, we present our institutional experience regarding the epidemiologic and clinicopathologic characteristics of BFOLJ.

Material and methods

Archived BFOLJ cases diagnosed over a 22-year period (2000-2022) were retrieved from the electronic laboratory databases of Oral Pathology Consultants at The OSU. Available information regarding patient age and gender, anatomic location, lesion focality, radiographic appearance, and histopathologic diagnosis was recorded and used for analysis.

Results

A total of 518 BFOLJ cases were identified with 440 (85%) affecting women and 78 (15%) men (F:M=5.6:1; age range=4-92y, mean=40.5y). The mandible was involved in 86% and the maxilla in 14% of the cases with the posterior mandible being the most common site (304, 57%). Among 123 BFOLJ with available clinico-radiographic information, 101 (82%) appeared unifocal and 22 (18%) showed multifocality with 63% of the cases presenting as well-defined, mixed radiopacity/radiolucency, 23% as unilocular radiolucency and 14% as radiopacity. Most BFOLJ were diagnosed as COD (449, 86.7%; F:M=7.8:1; age range=11-92y, mean=43y) further subcategorized as focal (292, 65%), florid (46, 10.25%), periapical (19, 4.25%), and COD NOS (92, 20.5%). Other diagnoses included OF (33, 6.4%; F:M=1:1; age range=4-47y, mean=26y), FD (28, 5.4%; F:M 3:1, age range=8-66y, mean=31y), juvenile OF (7, 1.3%) and renal osteodystrophy (1, 0.2%). Thirty (5.8%) BFOLJ were associated with secondary lesions, chiefly traumatic bone cyst (18, 60%), followed by central giant cell lesion (4, 13.3%) and xanthoma of bone (3, 10%).

Conclusions

BFOLJ are relatively uncommon and largely represent forms of COD with a strong predilection for the posterior mandible of middle-aged women. Histopathologic examination and clinico-radiographic correlation are required for accurate diagnosis and proper management.

Introduction

Neurofibroma is a benign tumor that originates from the peripheral nerve sheath. In its localized form, the tumor most often presents as a superficial, solitary lesion arising anywhere on the body. Oral localized neurofibromas are common. The diffuse and plexiform growth patterns of neurofibroma are associated with neurofibromatosis 1, while the localized form is not. Histologically, neurofibromas are widely variable. One rare form is the lipomatous neurofibroma. This more recently described variant is characterized by the presence of intratumoral fat in clear excess of entrapped adipose tissue. Typically, this variant is cutaneous and affects the head and neck- only a handful of cases have been reported in the oral cavity. Here, we report four cases of intraoral lipomatous neurofibromas.

Materials and Methods

Cases for this series were retrieved from the archives of Oral Pathology Laboratory, Inc.

Results

Four cases were identified, two of which our laboratory have previously presented. Age at presentation ranged from 18 to 62 with a median of 35. Two male patients and two female patients were affected, with three of the tumors presenting on the hard palate and one presenting on the dorsal tongue. All tumors presented as a raised mass. No patients had a history of neurofibromatosis type 1. Immunohistochemical studies completed on the cases reveal S100 positivity, and negativity for CD99, CD34, and BCL2. Clinically, these tumors were often biopsied under suspicion of a salivary gland neoplasm.

Conclusion

Five cases of intraoral lipomatous neurofibroma have been reported to date- one by Shimoyama et al. and the four present cases. The histologic features identified here are concordant with previously described cases, both intraoral and cutaneous. This case series expands the known spectrum of this lesion. Greater recognition of this variant may help to identify the driving forces behind this histopathologic finding.

Introduction

Malakoplakia is a rare inflammatory disorder which typically occurs in immunocompromised patients secondary to impaired bactericidal activity of macrophages. Histopathologically, it is characterized by a dense infiltrate of histiocytes with eosinophilic cytoplasm and Michaelis-Gutmann bodies, which are basophilic, round inclusions within macrophages. Malakoplakia commonly affects the genitourinary and gastrointestinal tracts but has been reported in other organs. Oral cavity involvement, however, is exceptionally rare. Here we present the first reported case of maxillary malakoplakia in an immunosuppressed patient.

Materials and Methods

This case was submitted to the Oral Pathology Laboratory, Inc. at New York-Presbyterian Queens.

Case Report

A 66-year-old male with a history of lung transplant presented with a non-healing extraction site of the left maxilla. To rule out a diagnosis of malignant neoplasm, the submitting clinician performed an incisional biopsy. Multiple pieces of tan-brown soft tissue measuring 2.3 × 2.0cm in aggregate were submitted for histopathologic examination.

Microscopic examination revealed a lesion composed of diffuse sheets of ovoid to spindle cells containing foamy cytoplasm. Numerous round, basophilic inclusions, consistent with Michaelis-Gutmann bodies, were noted within the cells. The inclusions were positive with von Kossa stain, indicating the presence of calcium. Immunohistochemical studies for CD68 were strongly positive, confirming a dense macrophage infiltrate. GMS and PAS stains were negative for the presence of microorganisms.

Conclusions

This is the first reported case of malakoplakia affecting the maxilla. Forty-nine previous cases of malakoplakia affecting the head and neck region have been published, with only 9 intraoral cases reported. Malakoplakia should be considered in the differential diagnosis of patients presenting with non-healing surgical sites, especially in those with a history of immunosuppression. Early diagnosis and prompt treatment are essential for successful management of this rare disease.

Introduction

Foreign body gingivitis (FBG) is an inflammatory reaction associated with the presence of foreign material in the gingival tissues. Si is the element most prevalent in the foreign particles and is typically found in nature as SiO2 (silica). Silica is a major component of toothpaste, pumice, polishing paste, and carborundum. We have previously reported a significant association between the presence of foreign particles and epithelial dysplasia. Herein, we aim to evaluate the clinical and histologic features of additional gingival lesions of FBG with significant epithelial changes to better characterize the histologic spectrum of the host response and epithelial findings.

Materials and Methods

Fourteen gingival lesions showing FBG and significant epithelial changes were retrieved from the archives of the Texas A&M Oral Pathology Services. Clinical records and deidentified microscopic slides were reviewed.

Results

Both sexes were affected equally, with an average age of 68.7 years. There was a strong predilection for the posterior gingiva (92.9% of cases), and the lesions were clinically suspected to be leukoplakia, proliferative verrucous leukoplakia, or dysplasia in the majority of cases. Microscopically, the predominant inflammatory cell type was lymphocytes followed by plasma cells, and the pattern of inflammation was lichenoid in 64.2%. A verrucous architecture was seen in 57.1% of the cases and varying degrees of dysplasia in 71.4 %.

Conclusion

Although the effects of silica microparticles in oral tissues are poorly understood, they have been shown to cause significant changes such as silicosis and carcinoma in the lung. Given the recent increase in the prevalence of gingival carcinomas and the wide use of nano- and microparticles in dental and household products, the biological effects of these particles in oral epithelium need to be better elucidated. Further documentation and evidence are needed, and pathologists should evaluate and document all gingival premalignant lesions for foreign material.

Introduction

Plasma cell gingivitis has been recognized and described for many decades and has generally been associated with sensitivity reactions to various oral healthcare products or food flavoring agents. Plasma cell gingivitis is recognized as clinical lesions that are flat to slightly raised, erythematous, and diffuse but confined to the attached gingival surfaces. Whereas plasma cell mucositis has been a term utilized when lesions extend beyond the attached gingival surfaces. 3 abstracts on plasma cell lesions of the oral cavity were presented at last year's meeting and anecdotal discussions amongst oral pathologists revealed a general sense that these lesions have been increasing in incidence.

Materials and methods

We will present a small case series where cases were associated with CAPB containing toothpastes. The rise of cocamidopropyl betaine containing dentifrices began approximately in 2008 as a result of trying to eliminate sodium lauryl sulfate from oral healthcare products. However, it is not well-known that CAPB was recognized as 2004 allergen of the year by dermatologists.

Results

Simply changing the CAPB containing toothpaste resulted in resolution of the gingival/mucosal phenotype in multiple patients. However, we also eliminated other substances which have been associated with plasma cell gingivitis such as cinnamon or herbal ingredients making a definitive association impossible.

Conclusion

This abstract discusses another possible oral healthcare chemical that clinicians should be aware of in the diagnosis and potential management of oral plasma cell lesions. We also wish to seek out more evidence of whether the association is real or possibly due to the synergistic effect of multiple oral healthcare product chemicals.

Introduction

Oral squamous cell carcinoma (OSCC) shows a limited response to current systemic treatments, and this resistance could be associated with cancer stem cells (CSC). NFκB is an activated pathway in several malignancies, including head and neck cancer. Here we evaluated the role of NFκB inhibitor on the behavior of CSC derived from OSCC.

Material and methods

Emetine was used as an NFκB inhibitor. CSC presence was assessed by tumorspheres, and the emetine IC₅₀ was determined in this specific cell population. Also, the CSC was quantified by the enzymatic activity of aldehyde dehydrogenases (ALDH) using flow cytometry. Immunofluorescence staining for phosphorylated protein p65 was used to identify the NFκB levels of tumor cells. Finally, OSCC cells were sensitized with emetine for 24 hours followed by administration of cisplatin (IC₅₀).

Results

The IC₅₀ of emetine in CSC OSCC was 0.5μM. We then treated the OSCC cells with the emetine IC₅₀, which showed a significant reduction of the ALDH population, while the NFκB pathway was inhibited. Further, emetine sensitized OSCC cells to cisplatin, resulting in a reduction of the IC₅₀ from 3.9μM to 1.3μM for SCC9.

Conclusion

Our results suggested that CSCs play an important role in tumor resistance to chemotherapy and highlight the disruption of these cells by the NFκB inhibition as a promisor target therapy.

Introduction

Sebaceous carcinoma (SC) is an aggressive, rare cutaneous malignancy. It shows striking predilection for the skin of the eyelid, face, neck, and scalp, but may arise on any cutaneous site.. Extraorbital sites overall account for only approximately 25% of SCl, and the head and neck region is involved in 70% of cases. Involvement of the oral cavity in SC is rare. Hereby we present a case of cutaneous SC with direct extension and involvement of the oral mucosa.

Case presentation

A 25-year-old male presented with a painless ulcerated lesion on the left commissure area persisting for a duration of 3 months. The past medical history was unremarkable. Extra-oral examination showed a markedly ulcerated, exophytic, irregularly shaped mass of the left commissure extending to the labial and buccal mucosa. The lesion straddled the point of transition between mucosa and skin. The clinical differential diagnosis included squamous cell carcinoma, basal cell carcinoma, and salivary gland malignancy. On histopathological examination, a poorly differentiated malignant epithelial neoplasm with sebaceous differentiation was identified. Upon immunohistochemical testing, the neoplastic cells were strongly reactive for AE1/3, EMA, and CD15. In addition, CAM 5.2 and Ber-EP4 were also focally positive toward the periphery of the neoplastic islands. SOX10 was negative. The final diagnosis of sebaceous carcinoma was rendered. The patient was referred for oncologic treatment but lost to follow-up.

Conclusion

This case illustrates a rare case of SC with extension beyond its typical cutaneous boundaries into the oral mucosa. The presentation of SC in this manner underscores the complexity of diagnosing and managing such uncommon manifestations of skin cancers. This case reinforces that SC should be included in the differential diagnosis of lesions involving both skin and oral cavity and careful evaluation of both histopathology as well as immunohistochemistry is essential for accurate diagnosis.

Case Presentation

A 39-year-old female with unremarkable medical history presented with severe, generalized gingival enlargement of all quadrants. The gingival swelling was present for six years but only recently caused discomfort in addition to increasing tooth mobility. The patient denied any constitutional symptoms and was not taking any medications. She did not have any cutaneous, ocular, or genital lesions. She has had three miscarriages in the past and of note, the patient's symptoms worsen during pregnancy. On clinical examination all four quadrants demonstrated diffuse maxillary and mandibular buccal and lingual erythematous gingival enlargement associated with displacement of teeth and associated mobility. Radiographs showed generalized horizontal bone loss in both the maxilla and mandible. The clinical differential diagnosis of the severe gingival swelling included acute leukemia, lymphoma, inflammatory bowel disease, plasma cell gingivitis, and drug-induced gingival overgrowth. An incisional biopsy demonstrated a densely cellular proliferation of spindle cells arranged in a storiform pattern of short fascicles suggestive of a mesenchymal proliferation. The connective tissue stroma also showed areas of atypical metaplastic osteoid and chondroid deposition. Small islands of clear cells reminiscent of odontogenic islands were found encased within the osteoid deposits. There were also aggregates of granular cells noted. A comprehensive IHC panel included SATB2 (diffusely positive), CD56 (+ve), SSTR2A (focal +ve), CK19 (-ve but highlighted odontogenic epithelial nests), and the following stains were negative: AFB/ZN, TLE-1, MUC-4, ERG, CK7, CK20, Desmin, STAT-6, S-100, SOX-10, CD34, CD99, CD68, CD45, Kappa/Lambda (polytypic), and Ki67 (low). Molecular work-up for phosphaturic mesenchymal tumor was performed and CISH for fibroblast growth factor 23 (FGF23) was negative.

Conclusion

The features are most likely to represent multifocal peripheral ossifying/odontogenic fibromatosis supported by diffuse SATB2 expression which suggests origin from the periodontal ligament. Additionally, the findings may be in part related to an underlying undiagnosed genetic/hereditary etiology.

Introduction

Squamous Cell Carcinoma (SCC) is the most common malignant tumor in the oral cavity and has been reported to be more common in older men. It has been associated with smoking, smokeless tobacco and alcohol. Recent studies have posited an increase in incidence of oral SCC in younger populations. The objective of this study is to examine incidence of oral SCC in different age groups, stratified by sex and site.

Materials and Methods

Demographic information was collected by searching the Oral Pathology Laboratory, Inc/ NYPQ database from 2003-2023. Data was collected for all SCCs and separated into 5 year age brackets (<45, <40, <35 and <30) and by decade (2003-2010, 2011-2020 and 2021-2023) This was further subcategorized by location and by gender.

Results

The incidence of SCC decreased in the <40 and <45 yo age brackets from 2003-2023 by 1.1% and 1.54%, respectively. The incidence of SCC increased in the <30 and <35 yo age brackets from 2003-2023 by 0.7% and 0.4%, respectively. While the incidence of SCC amongst females decreased from 2003-2023, the incidence of SCC on the tongue in females in the <40, <35 and <30 yo age brackets increased from 2003-2023 by 17.1%, 20% and 33.3%, respectively. For men, the incidence of SCC varied over the years and across age brackets, with the highest incidence occurring in 2011-2020. The incidence of tongue SCC in men increased as age decreased. From 2003 to 2023, its incidence increased in all age groups except the <30 age group where it decreased by 16.9%.

Conclusion

This study shows a slight increasing trend in SCC in all patients <35. There's a marked increase in females with SCC of the tongue. Further demographic information is needed to better understand the etiology of these trends.