A case report of angiosarcoma originating from the tongue.

Key Clinical Message: Angiosarcoma is a rare malignant disease with an extremely poor prognosis, showing rapid progression of the local tumor and/or distant metastases. Although multidisciplinary approach including systemic chemotherapy and radiation therapy is ideal for this disease, surgical resection have a role in disease control and should be performed as soon as possible. Abstract: Angiosarcomas originating from the tongue are rare and have extremely malignant features, leading to a poor prognosis. Herein, we report the case of a patient with angiosarcoma arising from the tongue who was successfully treated surgically. A 71-year-old man was diagnosed with a mass on the right side of his tongue and visited the Department of Oral and Maxillofacial Surgery at our hospital. The patient was referred to our department for further examination and treatment after a biopsy of the right edge of the tongue. An irregularly raised tumor 50 mm in length was noted on the right lingual border. The preoperative diagnosis was a primary angiosarcoma of the tongue (clinical stage, T3N2bM0, Stage IV). As his tumor had been growing rapidly, he emergently underwent partial right-sided tongue resection and right neck dissection without reconstructive surgery. The histopathological diagnosis was pT3N0. Postoperatively, the patient showed no signs of recurrence or metastasis during the 1-year follow-up. As for angiosarcomas, surgical resection is the only curative treatment, and surgery should be performed as soon as possible after the final diagnosis.

A case of complete remission of Hodgkin lymphoma confirmed histopathologically by neck dissection.

BACKGROUND: Hodgkin lymphoma (HL) is diagnosed definitively by biopsy, and treatment is based on stage. Owing to the nature of the disease, post-treatment efficacy is determined mainly by fluorodeoxyglucose-positron emission tomography/computed tomography, and the efficacy of treatment is not confirmed by histopathology. We report a case of tongue cancer after treatment for HL, in which a post-treatment lymph node with complete remission was histopathologically confirmed by neck dissection. CASE: The patient was a 74-year-old man who was referred to our hospital for cancer on the right side of his tongue. He had previously undergone chemotherapy for HL involving the right side of his neck and achieved complete remission. Because he had cT3N2cM0 tongue cancer, glossectomy and bilateral neck dissection were performed. Surprisingly, histopathological examination revealed that there was neither metastatic lymph nodes nor lymphoma cells in his right neck. Moreover, there was no lymphatic structure in his remnant lymph nodes. CONCLUSION: This was a rare case in which complete remission of HL was confirmed by histopathological analysis. The absence of lymph node structure and lymphatic flow led to contralateral neck lymph node metastases of tongue cancer.

Intramuscular Myxoma of the Intrinsic Muscles of the Tongue: A Case Report with Literature Review.

Myxoma is a benign tumor of mesenchymal origin. It frequently occurs in the muscles of the hip and extremities; however, it rarely occurs in the head and neck region. This report describes the second case of an intramuscular myxoma of the tongue. A 23-year-old woman was referred to our institution for the diagnosis and treatment of a left tongue lesion. T2-weighted magnetic resonance imaging revealed an 8 × 6-mm mass in the tongue. Based on a clinical diagnosis of a tongue tumor, excisional biopsy was performed under general anesthesia. The histopathological diagnosis was an intramuscular myxoma. The postoperative course was uneventful, and there was no evidence of tumor recurrence 3 years after surgery.

Diagnostic Accuracy of High-Grade Intraepithelial Papillary Capillary Loops by Narrow Band Imaging for Early Detection of Oral Malignancy: A Cross-Sectional Clinicopathological Imaging Study.

This study aimed to clarify the advantages and disadvantages of conventional visual inspection (CVI), endoscopic white light imaging (WLI), and narrow-band imaging (NBI) and to examine the diagnostic accuracy of intraepithelial papillary capillary loops (IPCL) for the detection of oral squamous cell carcinoma (OSCC). This cross-sectional study included 60 participants with oral mucosal diseases suspected of having oral potentially malignant disorders (OPMDs) or OSCC. The patients underwent CVI, WLI, NBI, and incisional biopsy. Images were evaluated to assess the lesion size, color, texture, and IPCL. Oral lichen planus (OLP) and oral leukoplakia lesions were observed in larger areas with NBI than with WLI; 75.0% were associated with low-grade (Type 0-II) IPCL. Various types of oral leukoplakia were seen; however, all OSCC cases showed high-grade (Type III-IV) IPCL. The diagnostic accuracy of high-grade IPCL for OSCC showed a sensitivity, specificity, positive predictive value, negative predictive value, and accuracy of 100%, 80.9%, 59.1%, 100%, and 85.0%, respectively. A non-homogeneous lesion with high-grade IPCL strongly suggested malignancy. Overall, our results indicate that WLI and NBI are powerful tools for detecting precancerous and cancerous lesions using IPCL. However, NBI is influenced by mucosal thickness; therefore, image interpretation is important for accurate diagnosis.

Osteosarcoma of the Mandible in an Elderly Patient.

Osteosarcoma is a malignant tumor in which the cancerous cells produce an osteoid matrix or mineralized bone. Jaw bones are affected in 6% of all osteosarcomas and are the fourth most common site of origin. Surgical treatment of osteosarcoma in elderly patients is rarely reported. Here, we report successful treatment of osteosarcoma arising in the mandible of a 90-year-old man. The patient was referred to our institution for diagnosis and treatment of an oral lesion. Intraoral examination revealed that a hard mass measuring 35 × 27 mm was located on the floor of the oral cavity, attached to the bone, and its growth displaced the tongue posteriorly. Therefore, he experienced difficulty in speech and swallowing. Biopsy of the mandibular mass was suspicious for chondrosarcoma. Preoperative examination did not detect critical risks for general anesthesia or surgery. Based on a clinical diagnosis of a malignant bone tumor of the mandible, segmental mandibular resection with reconstruction using a titanium plate was performed. Surgical site infection occurred on postoperative day 12, which was resolved by drainage, local irrigation, and administration of antibiotics. There was no delirium or cardiovascular or pulmonary complications. Surgery resolved the patient's difficulties in speech and swallowing. There was no evidence of tumor recurrence or metastasis 4 years after surgery. This case showed that it was not necessary to exclude surgical treatment merely because the patient was 90 years old. Indications for surgery should be determined individually to improve the patient's quality of life.

Tophaceous pseudogout of the temporomandibular joint extending into the cranium: a case report with literature review.

Pseudogout is a disease characterized by calcium pyrophosphate crystal deposition. Involvement of the temporomandibular joint (TMJ) is rare. We herein report a case of tophaceous pseudogout of the TMJ with cranial extension. An 83-year-old woman was referred to our institution for treatment of right TMJ pain. The patient's medical and family histories were unremarkable. Magnetic resonance imaging showed a mass of about 35 mm in diameter compressing the bottom of the right temporal lobe of the brain. Based on a clinical diagnosis of a right TMJ tumour, biopsy was performed under general anaesthesia. The histopathological diagnosis was pseudogout. Considering the risk of surgically induced brain damage, the patient's advanced age and her relatively good quality of life, the treatment plan simply involved the observation of the lesion. Fourteen months after biopsy, the patient's activities of daily living remained unchanged and she had no TMJ pain.

Epigallocatechin gallate stimulated histamine production and downregulated histamine H1 receptor in oral cancer cell lines expressing histidine decarboxylase.

OBJECTIVES: Increased histamine production and the overexpression of receptors (H1R∼H4R) has been reported in several tumors. The effects of TGFß1 and epigallocatechin gallate (EGCG) on histamine synthesizing enzymes (HDCs), and the histamine transporter systems and receptors were investigated in this study. METHODS: Four oral cancer cell lines (HSC2, HSC3, HSC4, and SAS) were treated with or without TGFß1 or EGCG for 24 h. The expression levels of HDC, SLC22A3, H1R∼H4R, and TAS2R14 were investigated by Western blotting. Histamine concentrations were determined using the enzyme immune assay. Bitter taste receptor (TAS2R14 and TAS2R39) mRNAs were investigated by RT-PCR. RESULTS: Varying expression levels of HDC, SLC22A3, H1R∼H4R, and TAS2R14 were observed in the four cell lines, where histamine concentrations were found to be ∼500 fmol/ml in cell culture media and induced 2-2.5 times higher amounts of histamine following EGCG treatment. TGFß1 increased HDC expression in three cell lines, SLC22A3 expression in three cell lines, H1R expression in two cell lines, H2R expression in three cell lines, H3R expression in three cell lines, and H4R expression in three cell lines. EGCG decreased HDC expression in all four cell lines, SLC22A3 expression in three expression, H1R expression in all four cell lines, H2R expression in two cell lines, H3R expression in three cell lines, and H4R expression in two cell lines. CONCLUSIONS: EGCG upregulated histamine production and decreased the expression level of H1R in the oral cancer cell lines. It might prove useful for cancer therapy during histamine regulation.

Clinical Diagnostic Imaging Study of Osteoradionecrosis of the Jaw: A Retrospective Study.

Radiation therapy (RT) plays a significant role in the management of head and neck malignancies. This study aimed to review the clinical symptoms and various imaging findings of osteoradionecrosis (ORN) and provide a clinical perspective on the development of ORN. The retrospective cohort was composed of 57 sites in 54 patients who had a history of RT and suspected ORN and 48 sites in 45 patients who were confirmed to have ORN. Image analyses included computed tomography (CT), magnetic resonance imaging (MRI), positron emission tomography (PET)/CT, bone scintigraphy, and single-photon emission CT (SPECT). The irradiated tissue was damaged by RT, and the extent of damage was correlated with clinical symptoms. The bone marrow showed sclerotic changes and the devitalized bone showed bone resorption after invasive stimulation. Chronic trismus and pathological fracture are considered severe conditions, typically occurring in the last stage of ORN. Furthermore, neurological symptoms were an important sign of tumor recurrence, since diagnostic imaging was difficult. The possible treatment options vary depending on the stage of ORN. We speculate that bone sclerosis reactions and bone resorption are sequential reactions that seem to be protective measures of the bone to radiation injury.

A case report of interdigitating dendritic cell sarcoma originating from the oropharynx.

Interdigitating dendritic cell sarcoma is an extremely rare tumor and typically originates from lymph nodes. Here, we report a patient with tumor originated from the oropharynx who received successful surgical treatment.

Evaluation and comparison of oral function after resection of cancer of the upper gingiva in patients who underwent reconstruction surgery versus those treated with a prosthesis.

BACKGROUND: We retrospectively analyzed the articulation, mastication, and swallowing function of patients who underwent reconstruction or used a prosthesis after resection of the upper gingiva. METHODS: This study included patients who underwent resection of cancer of the upper gingiva from January 2014 to December 2018. Articulatory function was evaluated with Hirose's conversational function evaluation criteria. Mastication function was evaluated with the Yamamoto's occlusion table. Swallowing function was assessed with the MTF (Method of intake, Time, Food) score. RESULTS: The mean articulatory function score was 8 points in the Reconstruction Surgery Group (RSG) and 8.8 points in the Prosthesis Group (PG). The mean mastication function score was 2.8 points in the RSG and 3.3 points in the PG. The mean swallowing function score was M3T4F4 in the RSG and M4T4F4.3 in the PG. CONCLUSIONS: The prosthesis depends on the remaining occlusal support area. Our study suggest that prosthesis is better indication when there is more than one occlusal support area.

Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion.

BACKGROUND: Angiosarcoma occurs very rarely in the oral cavity, and the epithelioid type is even rarer. Here, we report a rare case involving an elderly man with a primary epithelioid angiosarcoma that originated from the mandibular gingiva and resembled a dentigerous cyst on radiographs. CASE PRESENTATION: A 66-year-old Japanese man visited our hospital with a chief complaint of gingival swelling in right mandibular third molar region. A panoramic radiograph showed bone resorption around the crown of right mandibular third molar, which was impacted. Incisional biopsy confirmed a diagnosis of epithelioid angiosarcoma. The lesion exhibited aggressive proliferation after biopsy resulting in uncontrolled bleeding and difficulty in closing the mouth. Mandibular segmental resection including the tumor was performed without reconstruction. Because of the aggressive preoperative course of the tumor, the patient received adjuvant chemotherapy. There were no signs of recurrence during a 2-year follow-up period. CONCLUSIONS: A review of the literature yielded only four reported cases of epithelioid angiosarcoma in the jaw region, with the lesions occurring in the maxilla in three cases. To our knowledge, this is the second case of primary epithelioid angiosarcoma in the mandible.

Radiation Esophagitis in a Patient with Oral Carcinoma and Bone Metastasis.

Radiation esophagitis requiring endoscopic evaluation occurs quite rarely, affecting <1% of patients undergoing radiation treatment. Acute radiation esophagitis develops within 3 weeks of radiation therapy. We describe herein a case of radiation esophagitis in a patient with oral carcinoma with multiple general bone metastases. Cisplatin, fluorouracil, and cetuximab were given for 3 cycles. Radiation therapy (30 Gy) to the thoracic vertebrae and lumbar vertebrae was prescribed to prevent worsening of bone metastases and relieve pain. Neutropenia was also observed due to chemotherapy. After the end of radiation therapy, the patient experienced chest pain, heartburn, and dysphagia. Upper gastrointestinal endoscopy revealed severe radiation esophagitis of endoscopic Fukui Acute Radiation Esophagitis grade 4. Oral food was discontinued and an intravenous proton-pump inhibitor was administered. After 3 weeks, upper gastrointestinal endoscopy showed improvement of radiation esophagitis, with scars. The symptoms of chest pain, heartburn, and dysphagia had also disappeared. This is the first case to be reported of acute radiation esophagitis in a patient with oral carcinoma with bone metastasis who experienced dramatic improvement of endoscopic findings. Neutropenia appears to be associated with more severe acute radiation esophagitis.

Effects of butyric acid, a bacterial metabolite, on the migration of ameloblastoma mediated by laminin 332.

Ameloblastoma is a benign tumor that develops in the jawbone. Occasionally, however, it may become malignant and metastasize to other tissues. Although it has been suggested that various cytokines and several adhesion factors may play a role in its malignant transformation, the details have not been elucidated. In this context, it has been reported that butyric acid produced by periodontopathic bacteria causes progression of malignant tumors occurring in the mouth via podoplanin. However, the influence of butyric acid on ameloblastoma has not been clarified. In the present study, therefore, the expression of various cytokines and adhesion factors in ameloblastoma upon stimulation with butyric acid or cytokines was investigated using real-time reverse-transcription polymerase chain reaction. Three cell lines (HAM1, HAM2 and HAM3) established from the same ameloblastoma were used in the experiments. It was found that the expression of mRNAs for epidermal growth factor (EGF) and transforming growth factor beta 1 (TGFß1) was increased in HAM2 and HAM3, respectively, upon stimulation with butyric acid. In addition, stimulation with EGF and TGFß1 led to an increase in the expression of laminin ß-3 mRNA in the respective cell lines. These results suggest that butyric acid may be involved in ameloblastoma exacerbation through the expression of laminin 332 (LM332) via EGF and TGFß1 produced by ameloblastoma itself.

Hypopituitarism manifesting after invasive dental treatment in a patient with carcinoma of the tongue: a case report.

BACKGROUND: The symptoms of hypopituitarism are not usually discussed in the clinical setting of oral surgery. CASE PRESENTATION: We herein report a case of hypopituitarism that became evident after biopsy and extraction of several teeth in a 68-year-old man with tongue cancer. Three days after biopsy, the patient developed nausea and vomiting, and his serum sodium had fallen to 124 mEq/L. His serum cortisol concentration was low. Although the plasma concentration of adrenocorticotropic hormone (ACTH) was within the normal range, ACTH stimulation testing showed a lack of cortisol response. Given these findings, we suspected secondary adrenal insufficiency. To investigate the cause of secondary adrenal insufficiency, MRI of the head was performed, which revealed pituitary gland atrophy. The results of pituitary anterior lobe hormone-stimulation tests were compatible with hypopituitarism. Thirty days after biopsy, partial tongue resection was successfully performed under general anesthesia with perioperative hydrocortisone supplementation. CONCLUSIONS: We must be aware of various signs of hypopituitarism when we perform invasive dental treatment.

Oral diverticulum: a case report with histopathological findings.

Oral diverticulum is rarely encountered, and the histopathological findings are unclear. We herein report a case of an oral diverticulum and describe its histopathological findings. A buccal mucosal outpouching was observed in a 66-year-old man. Food debris was seen within the outpouching. Under the clinical diagnosis of a diverticulum-like lesion, excision of the outpouching was performed through an intraoral incision to stop food impaction. Histopathological examination revealed that the excised lesion was a true diverticular structure composed of a keratinising, squamous epithelial lining with a surrounding fibrous connective tissue wall, with inflammatory cell infiltration and haemorrhage. The histopathological diagnosis was a diverticular pouch of the buccal region. Clinicians should be aware of this lesion to ensure correct diagnosis and treatment.

A pilot study of the clinical evidence for the methodology for prevention of oral mucositis during cancer chemotherapy by measuring salivary excretion of 5-fluorouracil.

Objective to re-examine measures to prevent oral mucositis caused by drugs in head and neck cancer patients during cancer treatment by measuring salivary excretion of 5-fluorouracil. Saliva, blood, and urine were simultaneously collected from oral cancer patients and breast cancer patient at the point in time of before, during, and after the administration of 5-FU, then the 5-FU levels of the samples were quantitatively analysed using LC-MS/MS. In all patients, the 5-FU levels in saliva and serum peaked at 30 min to 3 h after the start of 5-FU treatment, and high levels were maintained throughout the administration of the drug. With regard to urinary 5-FU levels, they remained high from 3 to 120 h after the start of 5-FU treatment. After the completion of 5-FU treatment, even though it not appeared in the patients' serum and urine promptly, 5-FU was detected in saliva at 12 h after the completion of 5-FU treatment in one oral cancer patient and at 48 h after the completion of 5-FU treatment in the breast cancer patient. It was suggested that the level of hydration after the completion of chemotherapy may be involved in the differences in 5-FU excretion.

Primordial Odontogenic Tumor: A case report with histopathological analyses.

Primordial odontogenic tumor (POT) is a benign mixed epithelial and mesenchymal odontogenic tumor included into the current World Health Organization (WHO) classification of Head and Neck tumours in 2017. As far as the authors have confirmed, only eight cases of this tumor have been reported so far. This paper reports a case of POT that occurred in the right mandible of a 5-year-old patient. Panoramic radiograph showed a well-defined homogeneous radiolucency displacing the unerupted second deciduous molar to the deep part of the mandible. Histopathologically, the tumor was composed of cell-rich mesenchymal tissue with myxoid areas, surrounded by columnar epithelium and non-keratinized cuboidal epithelium in the outer layers. The histopathological diagnosis was POT. The expression patterns of cytokeratins (CK) 14, 18, 19, vimentin and CD34 suggested that the grade of differentiation of the POT was approximately equivalent to that of normal primary tooth germ tissues in cap stage to late bell stage.