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1/26. Fine needle aspiration cytology of an intraductal papilloma originating in a sublingual gland. A case report.

    BACKGROUND: Intraductal papilloma of the salivary gland is a very rare tumor; only eight cases have been reported to date. Only histopathology has been used to investigate this tumor to our knowledge. There has not been any reported case of this tumor in a sublingual gland. CASE: A 3-cm oral floor mass with a lanulalike lesion in a 72-year-old female was aspirated. The cytologic features included various cellular smears containing compact clusters of papillary, radial, palisading, trabecular, tubular and buddinglike projected patterns; monomorphic columnar cells with oval-to-spindle nuclei basally located; abundant, finely vacuolar cytoplasm; indistinct nucleoli; and no squamous differentiated cells. A sublingual glandectomy was performed. Cytology, histology, immunohistochemistry and electron microscopy were performed with standard methods. The intraductal papilloma seems to have arisen within an excretory duct and differentiated into acinar cells. CONCLUSION: The cytologic findings of intraductal papilloma are unique and may allow its specific diagnosis on fine needle aspiration.
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2/26. Immunohistochemical and ultrastructural study of a papillary cystadenocarcinoma arising from the sublingual gland.

    Immunohistochemical and ultrastructural findings in a rare case of papillary cystadenocarcinoma arising from the left sublingual gland of a 55-year-old Japanese man are reported. Histologically, the tumor tissue was found to be composed of various-sized cystic cavities in which papillary epithelial projections with thin fibrovascular cores were observed. The papillary projections consisted of a single layer to several layers of high columnar epithelial cells. Invasion to the surrounding fibrous tissue and into the lymphatics was observed, thus suggesting an aggressive potential in the present case. The possibility of the involvement of myoepithelial cells could be excluded based on the immunohistochemical and ultrastructural findings. The immunohistochemical and ultrastructural findings also suggested that this type of salivary gland tumor, at least the present case, may arise from striated or excretory ducts. There was positive immunostaining for tumor markers CA19-9 and CA125. However, the biological role of these carbohydrate antigens in salivary gland tumors is unclear at present. Further investigations are, therefore, called for to solve this issue.
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3/26. Intraductal papillary tumors of the major salivary glands: case reports of benign and malignant variants.

    Intraductal papilloma is an extremely rare benign salivary gland tumor that occurs most commonly in the minor salivary glands. To our knowledge, a malignant counterpart of intraductal papilloma has not been described previously. We report one case each of benign and malignant intraductal papillary tumors. The benign tumor occurred in the sublingual gland and was a typical example of intraductal papilloma, with the exception that we found no previously published reports of this type of tumor in this location. The other patient had a left parotid gland tumor that was architecturally similar to the intraductal papilloma, with the addition of cytologic atypia, intraductal extension, microinvasion, and lymph node metastases. This tumor was diagnosed as intraductal papillary adenocarcinoma with an invasive component. Both patients were alive and well without evidence of recurrence 2 years and 6 months (case 1) and 6 years (case 2) after surgery. Immunohistochemical examination revealed that the tumor cells resembled duct luminal cells in both cases. The 2 tumors had different immunoreactivities for carcinoembryonic antigen, p53, and Ki-67. The malignant counterpart of intraductal papilloma should be considered in the differential diagnosis of salivary gland tumors with a predominantly papillary structure, even though this tumor is extremely rare.
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4/26. carcinosarcoma of the submandibular and sublingual salivary glands. A case report and review of the literature.

    carcinosarcoma of the salivary gland is a rare lesion, estimated at 0.2% of malignant salivary gland tumors [1, 2]. The present article describes a case occurring in a 54-year-old man with a submandibular mass in the right neck. Histologically, this lesion was a carcinosarcoma which involves the submandibular and the sublingual glands. Its stromal component was a chondrosarcoma and the epithelial one was an undifferentiated carcinoma in the whole tumor, with areas of adenoid cystic carcinoma in the sublingual gland. To our knowledge, we have not found any previously reported carcinosarcoma involving the sublingual gland.
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5/26. Acinic cell carcinoma of the sublingual gland accompanied by bone formation.

    A rare case of acinic cell carcinoma of the sublingual gland accompanied by bone formation is reported. The patient is a 79-year-old male who was referred to Yokohama Minami Kyosai Hospital with sublingual swelling. A tumor mass, 20 x 10 mm in diameter, was detected on the right side of the floor of the mouth. Computed tomography (CT) revealed a mass lesion with calcification in the sublingual gland. The patient underwent total sialadenectomy of the sublingual gland with conservation of the lingual nerve. Histologically, the lesion showed amylase-positive atypical cells with thyroid gland-like arrangement, and mature bone tissue in the stroma. Based on these findings, the tumor was diagnosed as acinic cell carcinoma accompanied by bone formation. Postoperative recovery was uneventful, and two years after surgery, there are no signs of distant metastases or recurrence.
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6/26. Solitary fibrous tumor with malignant potential arising in sublingual gland.

    A rare case is described of a solitary fibrous tumor (SFT) with malignant potential arising in the sublingual gland. A 59-year-old man presented with a 4-month history of a slowly enlarging painless mass in the center of the floor of the mouth. The tumor was a well-demarcated, firm mass with a multicystic lesion. The tumor exhibited highly cellular areas of spindle cells with patternless architecture alternating with hypocellular areas. The tumor cells were positive for CD34 and bcl-2 as well as vimentin, and negative for epithelial, myogenic, neurogenic and histiocytic markers. The tumor cells formed multiple satellite nodules around dilated ducts in the multicystic lesion, indicating infiltrative growth. In addition, areas exhibiting higher cellularity with increased mitoses were noticed in the satellite nodules, although cellular atypia was not obvious. These findings led to a final diagnosis of SFT with malignant potential. There has been no recurrence or metastasis for 27 months after the surgery. Solitary fibrous tumor of the salivary gland must be differentiated from various spindle cell neoplasms including myogenic, peripheral nerve sheath, fibroblastic and fibro-histiocytic spindle cell neoplasms, hemangiopericytoma and myoepithelioma. In addition to characteristic morphological features, an immunohistochemical positivity for CD34 and bcl-2 may aid in the diagnosis of SFT.
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7/26. Oncocytic tumor in myoepithelioma arising from the grossopalatine gland.

    Oncocytoma or oncocytic change in salivary glands normally occurs in old patients and mostly in the parotid gland, but those arising from the grossopalatine gland in young patients are extremely rare. The present case shows that oncocytic ductal structures were observed in myoepithelioma, consisting of spindle, plasmacytoid or epithelioid cells. The oncocytic tumor contained large amounts of eosinophilic granular cytoplasm and small nuclei.
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8/26. Basal cell adenoma of the sublingual gland.

    Salivary gland tumors constitute about 3% to 4% of all head and neck neoplasms. Approximately 80% originate in the parotid gland, and they rarely present in the sublingual gland; however, a disproportionately large majority of sublingual gland tumors are malignant. Basal cell adenoma is a benign epithelial salivary gland tumor that appears to have unique histologic characteristics, different from those of mixed tumors, and has a predilection for development in the parotid and minor salivary glands. No case has ever been reported as arising from the sublingual gland in the otolaryngology literature. We report here a case of a middle-aged woman with basal cell adenoma of the sublingual gland. The clinical presentation, pathological features, differential diagnosis, and treatment options for this relatively rare tumor are discussed.
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9/26. diagnosis of intra-oral MALT lymphoma using seminested polymerase chain reaction.

    diagnosis of mucosa-associated lymphoid tissue (MALT) lymphoma based on histological examination alone is difficult. We report three patients with histologically suspected MALT lymphoma who developed lymphoproliferative lesions of the sublingual gland. Seminested polymerase chain reaction (PCR) analysis applied to formalin-fixed and paraffin-embedded specimens showed clonal rearrangement of immunoglobulin heavy chain genes in two patients and a polyclonal characteristic in one. The clinical findings and Southern blot analysis confirmed the accuracy of the diagnosis. The molecular method described can be applied routinely to processed specimens to obtain helpful information for the diagnosis of low-grade malignancies of lymphoproliferative disorders, such as MALT lymphoma.
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10/26. A basal cell adenoma of the sublingual gland.

    To the knowledge of the authors, only one case of a basal cell adenoma of the sublingual gland has ever been reported. We report a second case of a basal cell adenoma arising from the sublingual gland in this paper.
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