Küttner's tumor of the sub-mandibular gland associated with fibrosclerosis and follicular hyperplasia of regional lymph nodes: a case report

<p>Abstract</p> <p>Introduction</p> <p>Küttner's tumor is characterized through histology by peri-ductal fibrosis, dense lymphocytic infiltration with lymphoid follicles, loss of acini, and occasional marked sclerosis of the salivary gland. On occasion, Küttner's tumor can be difficult to distinguish from malignant neoplasm.</p> <p>Case presentation</p> <p>A 58-year-old Japanese man was referred to our hospital with a three-month history of a painless swollen mass in the right sub-mandibular region. Histological findings revealed both lymphoid follicles with reactive germinal centers and variously sized lymphoid follicle-like nodules without definitive germinal centers or mantle zones. B-cells of similar size and shape occupied the lymphoid follicle-like nodules and stained positive for B-cell lymphoma. These cells were detected in the polyclonal B-cells by flow cytometric analysis and tested negative for CD10. Unusual B-cell proliferation was observed, but as there was no definitive evidence of B-cell lymphoma, the lesion was diagnosed as Küttner's tumor.</p> <p>Conclusion</p> <p>We report on a rare case of Küttner's tumor associated with fibrosclerosis and atypical lymphoid hyperplasia in both the sub-mandibular gland and regional lymph nodes. Although more cases need to be investigated, our findings might be helpful to further studies seeking to clarify the etiology of idiopathic sclerosing lesions arising in the organs and regional lymph nodes.</p

IL-1 Generated by Oral Squamous Cell Carcinoma Stimulates Tumor-Induced and RANKL-Induced Osteoclastogenesis: A Possible Mechanism of Bone Resorption Induced by the Infiltration of Oral Squamous Cell Carcinoma

We previously observed a novel osteoclastogenesis system that is induced by oral squamous cell carcinoma (OSCC) cells, which target osteoclast precursor cells (OPC) without upregulation of the master transcriptional factor of osteoclastogenesis, NFATc1. Here, we analyzed inflammatory cytokines that were preferentially expressed in one of the osteoclastogenic OSCC cell lines, namely NEM, compared with the subclone that had lost its osteoclastogenic properties. Based on a gene expression microarray and a protein array analyses, IL-1, IL-6, IL-8, and CXCL1 were chosen as candidates responsible for tumor-induced osteoclastogenesis. From the results of the in vitro osteoclastogenesis assay using OPCs cultured with OSCC cells or their culture supernatants, IL-1 was selected as a stimulator of both OSCC-induced and RANKL-induced osteoclastogenesis. The IL-1 receptor antagonist significantly attenuated osteoclastogenesis induced by NEM cells. The stimulatory effects of IL-1 for OSCC-induced and RANKL-induced osteoclastogenesis were effectively attenuated with cannabidiol and denosumab, respectively. These results suggest that IL-1 secreted from OSCC cells stimulates not only tumor-induced osteoclastogenesis targeting OPCs but also physiological RANKL-induced osteoclastogenesis, and this may be the biological mechanism of bone resorption induced by the infiltration of OSCC. These results also suggest that IL-1 inhibitors are candidates for therapeutic agents against bone resorption induced by OSCC

Pseudotumor in the temporomandibular joint: A case report

Introduction: Neoplastic disease in the temporomandibular joint (TMJ) is a rare condition and is difficult to differentiate from temporomandibular disorders (TMD) based on symptoms and simple X-ray examinations. Potential differential diagnoses include synovial chondromatosis and pseudotumor, both of which are also relatively rare in the TMJ. Presentation of case: We report a case of pseudotumor of the TMJ that was difficult to differentiate from synovial chondromatosis in a 71-year-old woman with a chief complaint of pain in the left TMJ. MRI of the right TMJ initially led to diagnosis of synovial chondromatosis. Extirpation of the lesion was performed under general anesthesia. Histopathological findings of the resected specimen revealed inflammatory granulation tissue without cellular atypism. Discussion: The pathological findings for the resected specimen were compatible with pseudotumor of the TMJ. These findings were not supportive of synovial chondromatosis or other tumor diseases. Conclusion: This case illustrates the importance of careful examination of a mass lesion in the TMJ for differentiation from other TMJ-related diseases

Synovial chondromatosis of the right side temporomandibular joint extending to the middle cranial fossa: A case report with 7-year postoperative follow up and expression of a biomarker of cell proliferative activity

Introduction: Synovial chondromatosis of the temporomandibular joint (TMJ) with cranial extension is rare. Here, we report 7-year follow-up of a case with immunohistochemical examination of cell proliferative activity. Presentation of case: The patient was a 72-year-old man. Severe bone resorption of the glenoid fossa was apparent on CT images. Pathological findings by biopsy led to diagnosis of synovial chondromatosis of the right side TMJ. Extirpation of the tumor was performed via temporopreauricular incision under general anesthesia. PCNA expression was examined by immunohistochemical analysis. The lesion had penetrated into the middle cranial fossa, but the cranial dura mater was intact. Expression of PCNA was confirmed. Discussion: The PCNA expression suggested that growth activity caused expansion of the lesion to the skull base. Conclusion: We were able to follow up this case for a long period without recurrence postoperatively

Metastasis of lower gingival squamous cell carcinoma to buccinator lymph node: case report and review of the literature

Abstract Background Metastasis of oral cancer to the buccinator lymph nodes (BN) is uncommon. The antegrade lymphatic flow in patients with normal anatomy and physiology makes metastasis of lower gingival cancer to BN unlikely. Case presentation A 67-year-old woman presented with a 46 × 25-mm tumor on her lower gingiva, along with metastatic foci in BN and cervical lymph nodes. After neoadjuvant chemotherapy, she underwent radical resection of the primary tumor and BN, along with neck dissection. Following surgery, she received adjuvant chemoradiotherapy. Two years after treatment, there has been no evidence of tumor recurrence or metastasis. Conclusion This is the first report of lower gingival squamous cell carcinoma with metastasis to BN. Metastasis to BN from lower gingival cancer is very rare but should be considered in patients with locally advanced tumors or tumors that metastasize to the submandibular node

Diagnosis and treatment of oral focal mucinosis: a case series

Abstract Background Oral focal mucinosis, the oral counterpart of cutaneous focal mucinosis, is a rare disease. As it has no characteristic clinical or radiological features, diagnosis is established by histopathological and immunohistological examination. We present three cases of oral focal mucinosis occurring in the retromolar (which is extremely rare) and gingival regions. Case presentation Case 1 involved a 26-year-old Japanese man with radiolucency in the right retromolar region on panoramic radiograph and computed tomography; no obvious protrusion was observed in the region. This finding was clinically diagnosed as a tumor of the retromolar region. Case 2 involved a 60-year-old Japanese woman. A tumor-like mass of tissue was identified on the buccal gingiva at the maxillary right canine and first premolar region. The lesion measured 7 × 6 mm and exhibited elastic hardness and healthy-colored mucosa. The lesion was diagnosed as an epulis. Case 3 involved a 47-year-old Japanese woman. A tumor-like mass of tissue was identified on the buccal gingiva at the maxillary right canine and first premolar region. The lesion measured 10 × 10 mm and exhibited elastic hardness and redness of the surface mucosa. This lesion was also diagnosed as an epulis. Resection was performed in all three cases, and the lesions were histopathologically diagnosed as oral focal mucinosis. Postoperative courses were uneventful and, thus far, there have been no recurrences. Conclusions Although it is difficult to diagnose oral focal mucinosis based on clinical symptoms and imaging findings, the disease should be considered a possibility when diagnosing benign oral tumors. We believe that an emphasis on histopathologic study is essential to confirm the clinical suspicion

A Novel, Tumor-Induced Osteoclastogenesis Pathway Insensitive to Denosumab but Interfered by Cannabidiol

Bone metabolism is strictly regulated, and impaired regulation caused by hormonal imbalances induces systemic bone loss. Local bone loss caused by tumor invasion into bone is suggested to be induced by the generation of cytokines, which affect bone metabolism, by tumor cells. The major cause of systemic and local bone losses is excess bone resorption by osteoclasts, which differentiate from macrophages by receptor activator of nuclear factor kappa-B ligand (RANKL) or tumor necrosis factor-alpha (TNF-alpha). We previously found a novel pathway for tumor-induced osteoclastogenesis targeting osteoclast precursor cells (OPCs). Tumor-induced osteoclastogenesis was resistant to RANKL and TNF-alpha inhibitors. In the present study, we confirmed that exosomes derived from oral squamous cell carcinoma (OSCC) cells induced osteoclasts from OPCs. We also showed that the depletion of exosomes from culture supernatants of OSCC cells partially interfered with osteoclastogenesis, and cannabidiol, an innoxious cannabinoid without psychotropic effects, almost completely suppressed tumor-induced osteoclastogenesis. Osteoclastogenesis and its interference by cannabidiol were independent of the expression of nuclear factor of T cell c1 (NFATc1). These results show that osteoclastogenesis induced by OSCC cells targeting OPCs is a novel osteoclastogenic pathway independent of NFATc1 expression that is partially caused by tumor-derived exosomes and suppressed by cannabidiol

Epithelioid cell granuloma with caseating necrosis possibly caused by periapical periodontitis: a case report

Abstract Background Epithelioid cell granuloma with caseating necrosis is a typical pathological finding in tuberculosis. While specific inflammation, including that related to tuberculosis, can induce caseating granuloma formation, there have been very few reports on the induction of caseating granuloma by non-specific inflammation. Chronic periapical periodontitis is usually related to bacterial biofilm formation as well as fungal or viral infection in the periapical lesion. However, it is difficult to eliminate these extraradicular pathogenic microbes by normal endodontic therapy alone, and more invasive surgical removal is almost always required. Case presentation Here we describe the case of a 30-year-old Japanese woman who had suffered from dull pain related to periapical periodontitis for approximately 10 years. Although the causal tooth had been previously extracted at the Department of Oral Surgery of another hospital in 2015, inflammation of the surrounding tissue had not abated. She was referred to our hospital in May 2016 and underwent surgical debridement via an intra/extraoral approach under general anesthesia. A caseating granuloma accompanied by a small amount of fungi was histopathologically confirmed in the excised specimen. Her inflammation has not been exacerbated since the operation. Conclusions This is the first report in which non-specific inflammation is shown to induce caseating granuloma arising in the jaw. Our report also highlights the importance of sufficient root canal treatment during the first stage of the procedure

Malignant transformation of oral lichen planus: a retrospective study of 565 Japanese patients

Abstract Background Oral lichen planus (OLP) is a chronic inflammatory oral mucosa disease that is recognized as an oral potentially malignant disorder. However, the potentially malignant nature of OLP remains unclear. Methods We designed this study to examine the demographic and clinical characteristics of patients with OLP and evaluate the associated malignant transformation rate. A total of 565 patients with a clinical and histopathological diagnosis of OLP who presented at our department between 2001 and 2017 were retrospectively studied. Patients who had clinical and histopathological features of oral lichenoid lesions (OLLs) classified as oral lichenoid contact lesions, oral lichenoid drug reactions and oral lichenoid lesions of graft-versus-host disease were excluded. Results The study population included 123 men and 442 women aged 21–93 years (mean ± standard deviation, 60.5 ± 11.8). The 565 patients were followed up for a duration of 55.9 ± 45.3 months, during which 4 (0.7%) patients developed squamous cell carcinoma (SCC). In three of these 4 patients who developed SCC, the clinical type of OLP was the red type. Conclusions Our results suggested that OLP was associated with a low risk of malignant transformation. We recommend regular follow-up for OLP patients and clear differentiation of oral epithelial dysplasia and OLLs to enable early detection of malignant transformation. Further investigation of the clinical risk factors associated with malignant transformation is necessary