Clear cell odontogenic carcinoma (CCOC) was reclassified from a benign tumor to a malignant tumor by the WHO classification in 2005 because of its aggressive nature and potential for local recurrence and distant metastases. It is a rare epithelial tumor of odontogenic origin. Only 82 well-documented cases have been reported to date to the best of our knowledge. Gardner syndrome is familial adenomatous polyposis (FAP), characterized by osteoma, soft tissue tumor, and colorectal polyposis. We report a case of CCOC arising in the upper jaw of a patient with with Gardner syndrome. A 34-year-old woman with Gardner syndrome was referred to our clinic because of rapid swelling of the left side of the maxilla. Malignancy was suspected on biopsy. She had a history of two operations at the same region. Computed tomography showed a bone defect in the upper jaw, with invasion to the surrounding soft tissue. We diagnosed CCOC of the left side of the upper jaw and performed tumor resection and prophylactic functional neck dissection. There was no recurrence or metastasis after a follow-up period of 4 years 8 months. Histologically, the tumor showed a biphasic pattern characterized by cords and nests of clear cells scattered with islands of cells with eosinophilic cytoplasm. We finally diagnosed CCOC. We discuss this rare case of CCOC with Gardner syndrome and review the literature.
Peripheral ameloblastoma is an uncommon variant of ameloblastoma occurring in the extraosseous region, which accounts for only 1 % to 5 % of all types of ameloblastomas. The most frequent sites of peripheral ameloblastoma are the upper and lower gingiva, but the anteromedial part of the mandibular ramus is not a common site of this tumor. We report a case of peripheral ameloblastoma arising in the anteromedial part of the right mandibular ramus of a 59-year-old woman who noticed a painless mass during swallowing. Computed tomographic and magnetic resonance images showed a well-circumscribed mass in the anteromedial part of the mandibular ramus. We performed an excisional biopsy with a rim of surrounding normal tissue under general anesthesia. Histopathologically, the mass was diagnosed as a plexiform type peripheral ameloblastoma. There was no evidence of recurrence at 45 months of postoperative follow-up.
Dedifferentiated carcinoma is defined as the abrupt transformation of a low-grade, well-differentiated tumor into a tumor of high malignancy. Because the transformed cells are highly aggressive, recurrence and metastasis frequently occur. We report a case of dedifferentiated carcinoma in adenoid cystic carcinoma of the submandibular gland with multiple metastases to the cervical lymph nodes and distant metastasis to the ileum. A 73-year-old woman was referred to our hospital because of submandibular gland carcinoma with multiple lymph node metastases. In another clinic, adenocarcinoma was pathologically diagnosed on lymph node biopsy. A surgically resected specimen showed that the tumor was composed of both a cribriform pattern of conventional low-grade adenoid cystic carcinoma and a high-grade dedifferentiated carcinoma. The lesion was thus a dedifferentiated carcinoma. Lymph node metastases contained dedifferentiated components. One year and a half after the operation, the tumor metastasized to the small intestine (ileum) . The tumor in the ileum was also pathologically diagnosed as a high-grade dedifferentiated carcinoma.
We report a case of alveolar soft-part sarcoma（ ASPS） arising in the mid-dorsum of the tongue in a 30-year-old woman. She had been aware of a slow-growing, painless mass of this region for 10 years and was referred to our department because of an episode of oral bleeding. Physical examination revealed an elastic hard mass arising in the mid-dorsum of the tongue, measuring 30 × 27 mm in diameter. Tumor excision was performed under general anesthesia. The histopathological diagnosis was ASPS because of the presence of alveolar structures composed of large polygonal cells with an acidophilic fine granular cytoplasm and PASpositive diastase-resistant crystals. There has been no recurrence or metastasis for 18 months.
We report a case of allergic contact stomatitis extensively affecting the oral mucosa that was caused by eating grated Japanese radish for several months. A 39-year-old woman was referred to our hospital because of an eating disorder due to stomatitis. She received supplementary liquid treatment after hospitalization. The symptoms improved, and she was discharged. However, she was readmitted because stomatitis recurred extensively after eating grated Japanese radish. Contact stomatitis or oral allergy syndrome was suspected. We therefore performed blood tests, patch testing, and provocative tests. The results of a skin patch test was positive, and stomatitis developed after several hours on provocative testing. We advised her to completely avoid eating grated Japanese radish. She was discharged after stomatitis had improved. Subsequently, stomatitis has not recurred because she has avoided eating grated Japanese radish.
Idiopathic hemarthrosis is frequently associated with osteoarthritis of the knee, but rarely develops in the temporomandibular joint (TMJ) region. We describe a patient with a hematoma in the TMJ in whom bone resorption was noted in the mandibular condyle. The patient was a 76-year-old man who visited our hospital because of pain in the left TMJ region. At presentation, the mouth opening was 38 mm, and tenderness was noted in the left TMJ region. A high-density area in the posterior region of the left mandibular condyle was noted on magnetic resonance imaging (MRI). Splint therapy was performed. When MRI was performed during the observation period, a tumorous lesion was noted in the posterior region of the left mandibular condyle, and the lesion slowly enlarged. Since bone resorption was noted in the posterior region of the left mandibular condyle on computed tomography, tumorectomy was performed under general anesthesia. The tumor was pathologically diagnosed as hematoma. The hematoma slowly enlarged over the course of about 2 years, and the histopathological findings suggested that it was a chronic expanding hematoma (CEH). As of about 1 year after surgery, the course has been favorable, without recurrence.
Adenosquamous cell carcinoma (ASC) is a rare tumor containing components of both adenocarcinoma and squamous cell carcinoma. It is often associated with lymph node and distant metastases, and is known to be among the most highly malignant tumors. There is no well-established treatment strategy for ASC yet because of the limited number of cases reported; therefore, evidence, particularly for the efficacy of non-surgical treatment, is being sought. We report the clinical features of two patients with ASC. Patient 1 was a 70-year-old man with ASC of the floor of the mouth (left side). The patient underwent by surgery, with a favorable postsurgical outcome, although he died of another disease 3 years after surgery. Petient 2 was a 48-year-old man with ASC of the left upper jaw associated with intracranial involvement. Although surgery was not performed, a complete response was obtained by radiotherapy (66 Gy) and two courses of chemotherapy (docetaxel at 60 mg/m2 on day 1, cisplatin at 60 mg/m2 on day 1, and 5-fluorouracil at 600 mg/m2 on days 1-5). However, the cancer recurred 10 months after the completion of chemoradiotherapy, and the patient died 18 months later. Surgery is currently recommended as the treatment of choice for ASC. Our experience with these cases suggests that survival may be prolonged by surgery combined with chemoradiotherapy.
Extranodal NK/T cell lymphoma, nasal type is a rare malignant lymphoma, especially in the oral cavity. It is characterized by a necrotizing ulcer, often strongly associated with Epstein-Barr virus. We describe a case of extranodal NK/T cell lymphoma, nasal type found because of antiphlogistic-resistant swelling in the palate. A 73-year-old man was referred to our hospital because of pain and swelling in the left side of the hard palate. Computed tomography showed no evidence of bone destruction, and the symptoms did not change after antibiotic therapy. Therefore, a biopsy was performed, and immunohistochemical analyses revealed positivity for CD56, TIA-1, EBER on in situ hybridization, CD45RO (UCHL1), and CD3 and negativity for CD79a and CD20 (L26). Four courses of DeVIC (CBDCA, VP-16, IFO, DEX) were administered to the patient. There has been no sign of recurrence after chemotherapy. This case demonstrates that antiphlogistic-resistant swelling with no inflammation in the oral cavity can be a clinical sign of extranodal NK/T cell lymphoma, nasal type.
Metastatic tumors of the temporomandibular joint are rare, although breast cancer frequently metastasizes to various bones. We report two cases of breast cancer that metastasized to the temporomandibular joint during treatment with bisphosphonates (BPs). Patient 1: A 49-year-old woman was referred to our hospital in 2006 because of a swelling of the left preauricular region. She had received a mastectomy in 2002 for a diagnosis of left breast cancer, and had been receiving BPs to treat multiple bone metastases. Computed tomography showed a soft tissue mass surrounding the left condylar head with bone resorption． Patient 2: A 53-year-old woman was referred to our hospital in 2012 because of a click of the right temporomandibular joint and trismus. She had received a mastectomy in 2009 for a diagnosis of left breast cancer and had been receiving BPs since 2011 because of metastases to the humerus and ischium. Computed tomography showed a soft tissue mass surrounding the right condylar head as well as the ribs. Both patients died of multiple metastases. We need to consider metastasis to the temporomandibular joint in patients with breast cancer who present with symptoms in this region.
We report the rare case of a 47-year-old man who received a penetrating stab wound caused by the branch of a tree that entered the floor of the mouth from the submental region. He climbed a tree and was pruning the branches. He fell down from the branch to the ground, and a branch penetrated his oral cavity. He was urgently transported to our hospital by an ambulance. He was alert, but could not speak because of the branch. His general condition was good, and there was a little bleeding. Emergency computed tomography was performed. On the same day, we performed an emergency operation under general anesthesia. We removed the branch, and the stab wound was sutured and drained. The branch was 17 cm in length and 3 cm in diameter. There was no damage to other structures including the salivary ducts. The postoperative course was good, and he was discharged 14 days after surgery. Preoperative computed tomography was effective for defining the positional relationship between a wooden foreign body and important organs.