Intraoral CD30+ T-Cell Lymphoproliferative Disorder with Lymphomatoid Papulosis Type C Features Mimics Lymphoma Histopathologically and Immunohistochemically.

BACKGROUND: Previous studies have shown that at least a of intraoral eosinophilic ulcer is best classified as a CD30 + T-cell lymphoproliferative disorder (LPD), with histopathology reminiscent of lymphomatoid papulosis (LyP) of the skin. Microscopically, a mixed population of inflammatory cells, often including eosinophils and varying numbers of atypical lymphoid cells, frequently expressing CD30, is typical for LyP, whose clinicopathological spectrum includes type A, B, C, D, E, and LyP with DUSP22/IRF4 rearrangement. To date, about 27 intraoral LyP cases have been reported. Of them, 7 cases were diagnosed as LyP type C, which is frequently confused with anaplastic large cell lymphoma (ALCL) on histopathology. METHODS: A 60-year-old male was referred for a one-month history of a tongue ulcer. RESULTS: Microscopy showed numerous subepithelial atypical large lymphoid cells, which expressed CD4 (with partial loss of CD3, CD5, and CD7), CD8 (few cells), CD30 (about 50%, in non-diffuse pattern with size variability), TIA-1, and Ki-67 (85%), without staining for CD56, ALK, LMP1, and EBER1/2, concerning for a diagnosis of ALCL. However, after three weeks, the lesion completely healed. CONCLUSION: We present here a rare case of intraoral CD30+ T-cell LPD that we believe is the oral counterpart of cutaneous LyP type C.

Oral Carcinoma Arising Under Implant-Supported Prosthesis: Progression of Proliferative Verrucous Leukoplakia Initially Mimicking Lichen Planus.

Proliferative verrucous leukoplakia (PVL) is a non-homogenous type of oral leukoplakia, characterized by multifocal white plaques, propensity to recur after treatment, with strong tendency towards malignant transformation. Interestingly, some studies show that, at initial stages, PVL may resemble oral lichen planus (OLP), potentially leading to misdiagnosis. A 52-year-old woman, with a previous OLP diagnosis, was referred to our service for implant installation and follow-up of OLP lesions. After clinicopathological re-evaluation, a diagnosis of PVL (early stage) was made, and a maxillary full-arch implant-supported prosthesis supported by implants was installed. After 6 years of follow-up, the patient developed squamous cell carcinoma around the implants. The current case emphasizes that PVL patients with oral lesions suggesting peri-implantitis or peri-implant mucositis deserve a more meticulous investigation.

Intraoral primary syphilis mimicking lymphoproliferative disorder.

Syphilis can mimic, clinically and microscopically, many other diseases. By microscopy, typically syphilis presents with plasma cell infiltration, admixed with lymphocytes and macrophages, in lichenoid and/or perivascular/perineural distribution pattern. When exuberant, this inflammatory infiltrate can mimic a lymphoproliferative disorder (LPD), notably plasma cell neoplasia or lymphoma. To date, about 12 cases of secondary syphilis, all but one in extraoral location, suggesting initially a LPD, have been published. Here, to our knowledge, we report an unusual case of intraoral primary syphilis initially suggesting LPD, notably lymphoid hyperplasia (pseudolymphoma); however, mucosa-associated lymphoid tissue (MALT) lymphoma and follicular lymphoma could not be disregarded. Polyclonality of plasma cells on immunohistochemistry, in strict clinical correlation, was essential to arrive at the correct diagnosis.

Lingual Actinomycosis Clinically Simulating Nodular Median Rhomboid Glossitis: Literature Review and Report of Additional Case.

Actinomycosis is an uncommon, subacute to chronic, suppurative bacterial infection caused by Actinomyces Israelii. About 3% of all actinomycosis cases occur in the tongue, often affecting adult patients (mean age, 50 years). The clinical characteristics of actinomycosis can resemble malignant or benign tumors, and other infectious diseases. A 56-year-old woman was referred presenting an ulcerated lesion on the tongue 1 year ago. Intraoral examination revealed an edematous nodular lesion with an ulcerated surface, slightly symptomatic, on the midline dorsum of posterior tongue, suggesting nodular median rhomboid glossitis. Cytology smear was negative for fungus. After excisional biopsy, histopathological examination showed a chronic inflammatory infiltrate supported by a fibrovascular connective tissue stroma, and at the deepest part, broad basophilic areas surrounded by neutrophils, containing numerous filamentous bacilli, which were highlighted by Gram and Groccott-Gomori staining. The final diagnosis was lingual actinomycosis. Oral amoxicillin treatment (8/8 h for 2 weeks) was started, and after 1-month complete resolution was observed. Lingual actinomycosis is a rare lesion that must be recognized by dentists, because its early diagnosis and correct treatment reduce the possibility of a clinical complication that compromises the patient's quality of life. Noteworthy, when located on the midline dorsum of posterior tongue, actinomycosis can simulate nodular median rhomboid glossitis, expanding its spectrum of clinical differential diagnosis.

Non-Ulcerated and Ulcerated Necrotizing Sialometaplasia: Report of an Additional Case and Literature Review.

Necrotizing sialometaplasia (NS) is a rare, self-limiting, necrotizing inflammatory lesion, often involving the minor salivary glands of the palate. NS occurs often in men (60%) older than 45 years. Commonly, it presents as an ulcerative lesion, measuring 1 to 3 cm in diameter, with an evolution time of a few weeks or days, simulating malignancy. However, in some instances, the mucosal surface is intact, thus emphasizing the importance of considering NS in the differential diagnosis of oral ulcerative and non-ulcerative lesions. To date, 12 cases of non-ulcerated NS in the palate have been reported. Here, we report a 50-year-old male patient who presented a non-ulcerated, asymptomatic, nodular swelling 3 months ago, located on the hard palate, clinically suggesting salivary gland tumor or lymphoproliferative disorder. After biopsy, a diagnosis of non-ulcerated NS was established. After 4 weeks, the lesion evolved with complete resolution. After literature review, notably, unlike ulcerated NS, the non-ulcerated NS affected patients a decade younger (33 years vs. 45 years), with marked female predilection (83% vs. 40%) and not uncommon bilateral presentation (33% vs. 10%). NS should be considered in the differential diagnosis of ulcerative and non-ulcerative lesions affecting the palate.

Pseudocarcinomatous Squamous Hyperplasia After Surgical Management Of Pediatric Ameloblastoma: An Immunohistochemical Study.

Pseudocarcinomatous squamous hyperplasia (PSH) affecting the jaws is uncommon. The mandible is the most common location, usually as a complication associated with osteomyelitis, medication-related osteonecrosis or osteoradionecrosis. Herein, we report an 8-year-old male patient presenting a solid ameloblastoma, plexiform type, who was treated by segmental mandibulectomy with bone grafting. Three months later, on the underlying graft, a sinus tract was observed, and a small bone fragment was resected which was diagnosed as bone sequestration associated with PSH. After 7-year follow-up, the patient is well, without alterations. To our knowledge, for the first time, we report in detail the immunohistochemical features of PSH which, in addition to epithelial component profile and chronic inflammatory cells, revealed a diffuse infiltration by dendritic cells, suggesting that epithelial cell-immune cell interactions in PSH pathogenesis should also be considered.

Maxillary Parosteal Osteosarcoma: Additional case Report and Literature Review of Surface Osteosarcomas.

Introduction: Surface osteosarcomas represent around 3-6% of all osteosarcomas, which include the parosteal (low-grade), periosteal (intermediate-grade) and high-grade surface osteosarcomas. The classical location is the femur, followed by tibia and humerus. Parosteal osteosarcoma is the most common type of surface osteosarcoma. To date, 26 cases of parosteal osteosarcoma affecting the jaws have been reported, with most cases following an evolution time of several months to years, clinically favoring a benign osseous or fibro-osseous lesion. Methods: Here, we report a 39-year-old female who was referred presenting a maxillary tumoral mass 5 years ago, clinically diagnosed as osteoma. After clinical, imaginological and histopathological analysis, a diagnosis of parosteal osteosarcoma was made. Conclusion: Thus, parosteal osteosarcoma should also be considered in the differential diagnosis of benign-appearance, bone-forming nodular lesions affecting the jaws.

Lymphomatoid Papulosis "Type E" Affecting the Palate: A Detailed Case Report and Review of Literature.

ABSTRACT: Lymphomatoid papulosis (LyP) belongs to the spectrum of primary cutaneous CD30 + lymphoproliferative disorders, characterized by chronic, recurrent, self-healing papules, small nodules, or ulcers. The clinicopathological features of LyP can mimic overt lymphomas. To date, about 27 intraoral LyP cases have been reported. Of them, only 2 cases were diagnosed as angioinvasive LyP (type E). Herein, we report a 24-year-old Brazilian man who presented a large ulcerated lesion on the hard palate with rapid evolution. Remarkably, there was no involvement of the skin or other mucous membranes. Microscopy revealed a lymphoid infiltrate constituted by medium-sized to large atypical cells, with angiocentric and angiodestructive features. The atypical cells showed immunopositivity for CD3, CD8, CD30, CD56, granzyme B, perforin, and focally for MUM1/IRF4. Ki-67 highlighted almost all atypical lymphoid cells, whereas EBER1/2 was negative. After 2 months of follow-up, the lesion healed completely. Although rare, LyP type E should be included in the differential diagnosis of oral ulcers.

Macrophages CD163+ and Factor XIIIa+ Provide a First-Line Defence against Proliferative Verrucous Leukoplakia Antigens.

This study aimed to evaluate the density of the dendritic cells (DCs) and macrophages in oral leukoplakia (OL) and proliferative verrucous leukoplakia (PVL) by immunohistochemical analysis. We analysed paraffined tissue samples of PVL (n = 27), OL (n = 20), and inflammatory fibrous hyperplasia (n = 20) as the control group using the immunomarkers for DCs (CD1a, CD207, CD83, CD208 and CD123) and macrophages (CD68, CD163, FXIIIa and CD209). A quantitative analysis of positive cells in the epithelial and subepithelial areas was determined. Our results showed a reduction in CD208+ cells in the subepithelial area of the OL and PVL compared to the control. Additionally, we found a higher density of FXIIIa+ and CD163+ cells in the subepithelial area in PVL compared to the OL and control. Four-way MANOVA revealed a relationship between increased CD123+ cell density in the subepithelial area of "high-risk" samples regardless of disease. Macrophages provide the first line of defence against PVL antigens, suggesting a distinct pattern of innate immune system activation in PVL compared to OL, which may contribute to the complexity and the high rate of malignant transformation in the PVL.

Intraoral lipoma with degenerative changes mimicking atypical lipomatous tumor: an immunohistochemical study.

Lipomas are mesenchymal neoplasms relatively uncommon in the oral cavity. Lipomas can exhibit histopathological features mimicking atypical lipomatous tumors (ALT) or dysplastic lipoma (DL) in the presence of degenerative changes. Relevantly, immunohistochemistry assists in the correct diagnosis. Herein, we present the case of a 54-year-old male with a sessile nodule located on the dorsum of the tongue. The histopathological analysis showed a diffuse, non-circumscribed adipocytic proliferation constituted by cells of variable size containing cytoplasmic vacuoles and displaced nuclei, some resembling lipoblasts supported by fibrous connective tissue stroma. By immunohistochemistry, tumor cells were positive for vimentin, S100, FASN, CD10, and p16. Rb expression was intact. Moreover, CD34, p53, MDM2, and CDK4 were negative. After 2-year of follow-up, no alteration or recurrence was observed. In conclusion, MDM2, CDK4, p53, and Rb immunomarkers can be used reliably to differentiate benign lipoma with degenerative changes from ALT and DL.

Radicular cyst in primary dentition showing angiolymphoid hyperplasia with eosinophilia (ALHE)-like features: An immunohistochemical study

The microscopical diagnosis of radicular cyst (RC) is straightforward; nonetheless, in some cases with unusual histopathological features, strict clinicopathological correlation is necessary to achieve the correct diagnosis. We report a case of a 5-year-old girl was referred presenting extensive carious lesion in the tooth #55, associated with vestibular sinus tract. Medical history revealed allergic asthma diagnosis. After clinical and imagi-nological exams, the deciduous tooth was extracted. Microscopically, the soft tissue lesion attached to the root showed typical RC features; however, in the cystic capsule, lymphoid follicles and vascular networks (lined by epithelioid endothelial cells) surrounded by nu-merous eosinophils, were observed. Immunohistochemistry, through CD3, CD20, CD34 and alpha-smooth muscle actin antibodies, highlighted these findings. Moreover, CD1a and CD207 were negative. To the best of our knowledge, this is the first report of RC showing angiolymphoid hyperplasia with eosinophilia (ALHE)-like features. (AU)

High density of CD8 T cell and immune imbalance of T lymphocytes subsets are associated with proliferative verrucous leukoplakia.

Oral leukoplakia (OL) and proliferative verrucous leukoplakia (PVL) are oral potentially malignant disorders (OPMDs) that microscopically show no or varying degrees of dysplasia. Even sharing clinical and microscopic aspects, PVL shows a more aggressive clinical behaviour, with a malignant transformation rate greater than 40%. Inflammatory infiltrate associated with dysplastic lesions may favour malignant transformation of OPMDs. This study aimed to evaluate the density of T cells and cytokines in dysplastic lesions from OL and PVL patients. Additionally, we evaluated whether soluble products produced in vitro by dysplastic keratinocytes are capable of modulating apoptosis rates and Th phenotype (Th1, Th2, Th17 and Treg) of peripheral blood mononuclear cells. The density of CD3, CD4 and CD8 T cells was assessed by immunohistochemistry. Cytokines and chemokines profile from frozen tissue samples were analysed using the LUMINEX system. Apoptosis rates and Th phenotype modulation were evaluated by flow cytometry. Our results showed an increase in the number of CD8 T cell in the subepithelial region from PVL dysplastic lesions in relation to OL samples. PVL showed increased levels of IL-5 and a decrease in IL-1ß and IFN-γ levels compared to OL. Soluble products of PVL and oral carcinoma cell cultures were able to reduce apoptosis rate and promote an imbalance of Th1/Th2 and Th17/Treg. The high-subepithelial density of CD8 T cells and immune imbalance of T lymphocytes subsets probably play an important role in the pathogenesis of PVL and may explain its more aggressive behaviour in relation to OL.

Evaluation of tooth eruption rate of incisor teeth in rats with estrogen deficiency.

OBJECTIVES: To assess the influence of estrogen deficiency on tooth eruption rate (TER) and gene expression of estrogen receptor alpha and beta (ERα and ERß) in the odontogenic region of teeth with continuous formation in a rat model. MATERIALS AND METHODS: Ovariectomies (OVX; n = 25) and sham surgeries (SHAM; n = 25) were performed in female Wistar rats when animals were 25 days old. The TER of the lower incisors, both in impeded (hyperfunction condition) and unimpeded (trimmed incisal edge-hypofunction condition) conditions, was evaluated using standardized digital photographs acquired every 48-72 h for 3 weeks (35th-53rd day of life), using a camera coupled to a stereomicroscope. Quantitative real-time PCR was performed to evaluate the relative gene expression of ERα and ERß in the odontogenic region. RESULTS: The OVX group showed a significant reduction in TER when compared to the SHAM group, only in the impeded condition (p = 0.03). There was no statistically significant difference between the groups in ERα gene expression (p = 0.33). ERß showed a significantly higher gene expression in the OVX group (p ≤ 0.05). CONCLUSIONS: Estrogen deficiency decreases TER in teeth under impeded condition. Estrogen deficiency also increases ERß gene expression in the odontogenic region of teeth with continuous formation. CLINICAL RELEVANCE: Hormonal disturbances affecting estrogen levels can cause alterations in dental formation and teeth eruption.

Intraoral lipoma with degenerative changes mimicking atypical lipomatous tumor: an immunohistochemical study

ABSTRACT Lipomas are mesenchymal neoplasms relatively uncommon in the oral cavity. Lipomas can exhibit histopathological features mimicking atypical lipomatous tumors (ALT) or dysplastic lipoma (DL) in the presence of degenerative changes. Relevantly, immunohistochemistry assists in the correct diagnosis. Herein, we present the case of a 54-year-old male with a sessile nodule located on the dorsum of the tongue. The histopathological analysis showed a diffuse, non-circumscribed adipocytic proliferation constituted by cells of variable size containing cytoplasmic vacuoles and displaced nuclei, some resembling lipoblasts supported by fibrous connective tissue stroma. By immunohistochemistry, tumor cells were positive for vimentin, S100, FASN, CD10, and p16. Rb expression was intact. Moreover, CD34, p53, MDM2, and CDK4 were negative. After 2-year of follow-up, no alteration or recurrence was observed. In conclusion, MDM2, CDK4, p53, and Rb immunomarkers can be used reliably to differentiate benign lipoma with degenerative changes from ALT and DL.

Primary cutaneous CD8+ cytotoxic T-cell lymphoma of the face with intraoral involvement, resulting in facial nerve palsy after chemotherapy.

The primary cutaneous (PC) CD8+ T-cell lymphoproliferative disorders (LPDs) comprise clinically and histopathologically heterogeneous entities including mycosis fungoides, lymphomatoid papulosis, hydroa-vacciniforme-like LPD, subcutaneous panniculitis-like T-cell lymphoma (TCL), PC acral CD8+ TCL, PC CD8+ aggressive epidermotropic cytotoxic TCL, and PC peripheral TCL, not otherwise specified (PTCL-NOS). We describe a 33-year-old man who presented with progressive facial swelling and lower lip involvement 1 year ago. Microscopy revealed an atypical small to medium-sized lymphoid proliferation exhibiting perivascular accentuation, adnexotropism, and apoptotic cell debris, without surface epithelium involvement. The tumor cells were positive for CD3, CD8, granzyme B, perforin, MUM1/IRF4, and TCR-BF1. The Ki-67 labeling index was 48%. EBER1/2 was negative. Additional studies confirmed localized disease. The diagnosis favored PC-PTCL-NOS. Two months after completing chemotherapy, right-sided facial nerve palsy was diagnosed. CD8+ T-cell LPDs should be considered in the differential diagnosis when assessing facial swelling with intraoral involvement.