Sialadenoma papilliferum in a young patient: a case report and review of the literature.

Sialadenoma papilliferum is a rare exophytic tumor of salivary gland origin, accounting for less than 1% of minor salivary gland tumors. It usually occurs in males older than 50 years as a painless papillary intraoral lesion. An 18-year-old male patient presented with an exophytic intraoral lesion present for approximately 12 years. The tumor was excised with a clinical diagnosis of infected hemangioma. However, histopathological diagnosis was consistent with sialadenoma papilliferum. The patient was followed up at regular intervals and no evidence of recurrence was noted. Our patient, aged 18 years, with tumor lasting for 12 years becomes the youngest case of intraoral sialadenoma papilliferum yet reported. This case highlights the importance of keeping sialadenoma papilliferum as a differential diagnosis of an intraoral exophytic proliferative lesion even in a young patient.     

A rare case of sialadenoma papilliferum with epithelial dysplasia and carcinoma in situ.

Sialadenoma papilliferum is a rare benign tumor of salivary glands that predominantly affects intraoral minor salivary glands. It is characterized by a biphasic growth pattern of exophytic squamous component and endophytic glandular component. Malignant transformation of sialadenoma papilliferum has only been recently recognized. The present article reports a rare case of sialadenoma papilliferum with epithelial dysplasia and carcinoma-in-situ in the exophytic component.     

Sialadenoma papilliferum of the parotid gland: Case report and review of literature

Sialadenoma papilliferum is a rare benign salivary gland tumor. We present an unusual case of sialadenoma papilliferum of the parotid gland, discuss clinical presentation, diagnostic challenges, and review of the literature. A 65‐year‐old male smoker presented with a large, exophytic, fungating, painless mass in the tail parotid for 8 years. The tumor developed an exophytic component 2 years before presentation. The patient subsequently underwent superficial parotidectomy with facial nerve preservation. Initial pathological analysis suggested a variant of Warthin's tumor. The pathology underwent extensive internal and external review. Final diagnosis was consistent with sialadenoma papilliferum. We present the fourth reported case of sialadenoma papilliferum within the parotid gland and only the second that has presented with breach of the overlying skin. The clinical presentation may mimic an advanced parotid malignancy. However, facial nerve preservation and good surgical outcomes can be obtained. © 2011 Wiley Periodicals, Inc. Head Neck, 2013     

A Spectrum of Basaloid Morphology in a Subset of EBV-Associated “Lymphoepithelial Carcinomas” of Major Salivary Glands

Nasopharyngeal carcinomas of the undifferentiated or lymphoepithelial type are most commonly seen in South East Asians. Identical tumors have also been described at a variety of other sites including lung, skin and salivary gland and have been referred to by a number of names including lymphoepithelial carcinoma (LEC). LECs of major salivary gland are extremely rare. They are particularly common amongst the Inuit populations of the arctic region including Greenland (Denmark), Canada and Alaska, as well as South East Asians. Within the Inuit group, this tumor represents the majority of all salivary gland carcinomas. Amongst primary LEC of major salivary gland, most cases reported in the literature have represented typical nasopharynx-like tumors. Variants of Epstein–Barr Virus (EBV) associated LEC have not been described previously, to the best of our knowledge. In this report, we describe 4 EBV-associated major salivary gland LECs with prominent basaloid morphology, which represent 22 % of a cohort of 18 salivary LECs from an Inuit population in Greenland. The features described in these cases raise a differential diagnosis of other basaloid tumors, particularly in light of the salivary gland location. A basaloid variant of LEC in major salivary gland should be recognized, especially in highly prone populations, to avoid misdiagnosis of other more common salivary tumors.     

Primary lymphoepithelioma-like carcinoma of minor salivary gland: a case report with immunohistochemical and in situ hybridization studies

BACKGROUND: Lymphoepithelioma-like carcinomas (LEC) of salivary glands represent rare epithelial malignancies, with most cases affecting the parotid gland. To our knowledge, there was only one LEC arising from the minor salivary gland described in the English-language literature. METHODS: We report the second LEC of the minor salivary gland in the buccal area of a 50-year-old Taiwanese woman, who underwent surgical resection and adjuvant radiotherapy and remained alive and well after 120 months of follow-up. Histologically, irregular tumor nests of undifferentiated epithelial cells with syncytial cell boundaries were found embedded within rich lymphoplasmacytic stroma. The tumor cells also showed strong c-KIT expression and evidence of Epstein-Barr virus (EBV) infection. CONCLUSIONS: Our case suggests potential pathogenic implications of both c-KIT and EBV in LEC of the minor salivary gland that can be cured by the combination of surgery and radiotherapy and has a very favorable long-term prognosis.     

Basal cell adenocarcinoma of the oral minor salivary glands: review of the literature and presentation of two cases.

Basal cell adenocarcinoma (BCA) is an unusual salivary gland malignancy that very rarely affects the minor glands. Here we present 2 cases of BCA arising in the minor salivary glands of the left cheek and junction of the hard and soft palate, respectively. The patients were both women aged 66 and 42 years. The tumors assumed a tubular-trabecular and a solid-membranous pattern, respectively. Both tumors showed immunopositivity for S-100, Bcl-2, and cytokeratin 7. One tumor was also immunopositive for epithelial membrane antigen and carcinoembryonic antigen, and the other tumor was reactive with p53 and vimentin. Both tumors were surgically removed. A comprehensive literature review revealed only 21 previously reported cases of BCA of oral minor salivary glands. This is an exceptionally rare salivary gland tumor, which, despite its low-grade behavior, demands complete surgical removal with adequate margins. Immunohistochemical studies may complement a thorough histopathologic analysis in discriminating BCA from other salivary gland tumors.     

Synchronous benign and malignant salivary gland tumors in ipsilateral glands: a report of two cases and a review of literature

BACKGROUND: Ipsilateral salivary gland tumors of different histologic types are rare and make up less than 0.3% of all salivary gland neoplasms. Only nine cases of synchronous benign and malignant ipsilateral parotid gland tumors have been described in the literature. METHODS: Two additional cases of synchronous benign and malignant neoplasms in the parotid gland are reported and discussed with a review of literature. RESULTS: Our first case describes a pleomorphic adenoma and a salivary duct carcinoma, an entity not previously reported in the literature. The second case documents the most common benign and malignant ipsilateral parotid gland neoplasm reported in this case series, a Warthin's tumor and a mucoepidermoid carcinoma. CONCLUSIONS: Synchronous salivary gland tumors exhibiting both benign and malignant components are uncommonly observed, with only nine cases published to date. We describe two additional cases of a synchronous benign and malignant ipsilateral parotid gland tumor.     

Plasmacytoid Myoepithelioma of the Palate: Case Report

Myoepitheliomas are extremely rare benign neoplasms of salivary glands. They represent 1–1.5% of all salivary gland tumors. They occur, principally, in the parotid gland and infrequently in minor salivary glands. There are distinct histological and immunohistochemical characteristics of the tumor which aid in the diagnosis. Myoepithelioma of the palate is uncommon and only a limited number of cases has been reported in the English literature. Herein, a case of plasmacytoid type of myoepithelioma of the palate in an adult female is presented along with review of the literature.     

Primary acinic cell carcinoma of mandible,report of a case and literature review

Introduction and importanceAcinic cell carcinoma (ACC) is a rare low-grade salivary gland malignancy that accounts for approximately 17% of all salivary gland malignancies. The most common site affected by ACC is the parotid gland followed by the submandibular glands, minor salivary glands, and sublingual glands. Also, it could hardly be observed in unusual sites such as the jaw bones.Case presentationThis case is an example of a central acinic cell carcinoma in a 73-year-old man who came up with a painless gradual swelling for 15 months. Based on clinico-radio-pathologic findings, the diagnosis of a solid variant Intraosseous Acinic Cell Carcinoma was established. Subsequently, the patient underwent hemimandibulectomy and modified radical neck dissection, followed by postoperative radiotherapy. Within a six-month follow-up period, no evidence of residual tumor was found.Clinical discussionCentral salivary gland carcinoma is a rare entity and intraosseous ACC is more scarcely observed. Based on our findings, a total of 17 cases of primary intraosseous ACC have been reported so far. Etiology and clinical presentations of this tumor are still vague due to its rarity.ConclusionDentists and oral surgeons must be aware of such a rare malignant lesion when encountering a radiolucent lesion within the jaws. The early diagnosis and a complete surgical excision to achieve tumor-free surgical margins and a long-term follow-up could result in significantly improved survival rates.     

Papillary Cystadenoma of Minor Salivary Glands: Report of 11 Cases and Review of the English Literature

Papillary cystadenoma is a rare, benign salivary gland tumor which is well-circumscribed, containing cystic cavities with intraluminal papillary projections. Only 19 cases arising within minor salivary glands (MnSG) from the oral cavity sites have been reported in the English literature (PubMed 1958–2014). We report 11 new cases of MnSG papillary cystadenomas in conjunction with a review of the literature. Demographic information, clinical and histologic features, treatment and prognosis are compiled and discussed for all 30 cases reported in the English literature.     

Clear Cell Carcinoma of Palatal Minor Salivary Gland Harboring a Novel EWSR1-ATF1 Fusion Gene: Report of a Case and Review of the Literature

Clear cell carcinoma (CCC) is a rare low-grade malignant salivary gland carcinoma. EWSR1-ATF1 fusion has been characterized as a consistent finding in CCC, with breakpoints described between EWSR1 exon 11 and ATF1 exon 3. So far, over 100 cases of CCC harboring EWSR1 rearrangement arising from salivary gland of the oral cavity have been reported. Although EWSR1 involvement in these cases was confirmed by EWSR1 break-apart FISH indicating the translocation, sequence analysis for EWSR1-ATF1 fusion type has been reported only in three cases of CCC so far. Herein, we report a CCC case with novel EWSR1-ATF1 fusion (EWSR1 exon 15 and ATF1 exon 5) arising in minor salivary gland and review the role of the chimeric variants in some malignancies with EWSR1-ATF1 rearrangement. Current tumor was composed of the small nests of clear tumor cells and hyalized fibrous stroma. Immunohistochemically, the tumor was positive for AE1/AE3, CK5/6 and p63, negative for S100, Melan-A, SMA and CD10. After 8 months of follow-up, there are no evidence of recurrence.     

Papillary cystadenoma of a minor salivary gland: report of a case involving cytological analysis and review of the literature.

A 91-year-old man presented with an asymptomatic swelling in the roof of his mouth. Clinically the lesion was nontender and appeared cystic. A CT scan showed a soft tissue swelling of his palate with no bony involvement. An orthopantogram and blood tests were noncontributory. A fine-needle aspiration biopsy was suggestive of a minor salivary gland neoplasm. An excision biopsy showed papillary cystadenoma of the minor salivary gland, with numbers of psammoma bodies. A stepwise approach is demonstrated in the diagnosis of this rare minor salivary gland neoplasm. To the authors' best knowledge, this is the 12th case of this tumor seen in the palate. Furthermore, no previous case has included a fine-needle aspiration biopsy in the work-up, the cytological features of which will be described.     

Tongue base minor salivary gland tumor: report of a case with mucoepidermoid and acinous cell components

A case of a large (5.5 cm), multilobulated, mucosa-covered, minor salivary gland tumor of the base of the tongue that displayed morphologic features of both a mucoepidermoid carcinoma and an acinous cell tumor is described. Generally, each of these salivary gland tumors occurs only rarely at this site. A tumor with both these elements has not previously been reported.     

Adenocarcinoma of the salivary gland metastatic to the pituitary gland: case report

OBJECTIVE AND IMPORTANCE: A case of metastasis to the pituitary gland from a ductal adenocarcinoma of the salivary gland is presented. Metastasis to this site is rare, and a salivary gland source has never previously been described. CLINICAL PRESENTATION: This patient presented with hypopituitarism, including diabetes insipidus. INTERVENTION: A craniotomy was performed to alleviate visual loss. The histological features of the sellar tumor were identical to those of a tumor removed from the parotid gland 18 months earlier. CONCLUSION: Although intrasellar tumors originating from embryonic rests of salivary gland tissue have been reported, metastasis from a malignant neoplasm arising within a true salivary gland is also possible and should not be excluded from consideration for patients in whom a salivary gland-like tumor is discovered in the sella turcica.     

Adenomatoid hyperplasia of the minor salivary glands on the buccal mucosa: A rare case report

INTRODUCTIONAdenomatoid hyperplasia of the minor salivary glands is a hyperplastic oral lesion which may be seen on minor salivary gland bearing areas on all oral mucosa, especially on soft and hard palate. This study reports a rare case of buccal adenomatoid hyperplasia of the minor salivary glands and discusses the clinical significance.PRESENTATION OF CASE48 year old male patient presented with a complaint of a swelling on his left cheek. Clinical examination revealed a bluish mass on the buccal mucosa. A provisional diagnosis of salivary gland neoplasm was made and the lesion was excised under local anesthesia. The histological diagnosis was adenomatoid hyperplasia of the minor salivary glands.DISCUSSIONBuccal localization of the adenomatoid hyperplasia of the minor salivary glands is quite uncommon in the literature. There are two cases in the English literature for our knowledge. In the clinical examination, the nodular and protuberated appearance of the lesion resembles buccal minor salivary gland tumors and vascular lesions. Histological analysis is fundamental to achieve correct diagnosis.CONCLUSIONThe differential diagnosis of buccal nodular, exophytic and colored mucosal lesions should include adenomatoid hyperplasia of the minor salivary glands.     

Osteoclast-type giant cell neoplasm of salivary gland. A microdissection-based comparative genotyping assay and literature review: extraskeletal "giant cell tumor of bone" or osteoclast-type giant cell "carcinoma"?

Primary salivary gland tumors resembling giant cell tumor of bone are very rare and have unsettled histogenesis. Both mesenchymal and epithelial origins have been suggested. We review 14 cases in the English-language literature and report another case, the first of which to be studied by microdissection-based microsatellite analysis. One-half of the tumors have been associated with a carcinoma, usually salivary duct carcinoma and carcinoma ex pleomorphic adenoma. Significant differences between this tumor and giant cell tumor of bone were observed. Unlike giant cell tumor of bone, in which the nuclei of the mononuclear and giant cells are similar, those of salivary gland show obvious differences between the nuclei of mononuclear cells and osteoclastic giant cells. In addition and in contrast to giant cell tumor of bone, the mononuclear cells of giant cell tumor of salivary gland express epithelial markers (epithelial membrane antigen, EMA; carcinoembryonic antigen, CEA) and androgen receptor. Genotypically, the microsatellite pattern of the giant cell component is more akin to the carcinomatous component and does not resemble giant cell tumor of bone. Biologically, giant cell tumor of salivary gland tends to be more aggressive than giant cell tumor of bone. We conclude that giant cell tumor of salivary gland is an unusual carcinoma that is not related to giant cell tumor of bone.     

Unique presentation of adenoid cystic carcinoma in postcricoid region: a case report and review of the literature

Malignant tumors of minor salivary glands are uncommon, representing only 2–4% of all head and neck malignancies. In the larynx, minor salivary gland tumors rarely occur and constitute less than 1% of laryngeal tumors. Most of the minor salivary gland tumors arise in the subglottis; however, they can also occur in the supraglottis in the false vocal cords, aryepiglottic folds, and caudal aspect of the epiglottis. The most common type of malignant minor salivary gland tumor is adenoid cystic carcinoma. In the last 20 years, only 40 cases of adenoid cystic carcinoma arising in variable locations of the larynx have been reported in the English language literature. We present a case of adenoid cystic carcinoma of the larynx arising in the inter-arytenoid and postcricoid region in a 54-year-old woman. To our knowledge, this is the first case of adenoid cystic carcinoma arising in this particular region of the larynx.     

Sialolipoma in Minor Salivary Gland: Case Report and Review of the Literature

Sialolipoma is a rare benign neoplasm characterized by a well-circumscribed mass composed of neoplastic mature adipose tissue and non-neoplastic salivary gland elements. A 72-year-old woman presented with a painless swelling located in the hard palate, which had been identified 15 days earlier. Microscopically, the tumor was well-circumscribed consisting of lobular proliferation of the lipomatous tissue with thin fibrous tissue septa containing clustered salivary gland elements. Both the glandular and adipose components were found in almost equal proportion. No atypia in the adipose tissue was observed. The definitive diagnosis was sialolipoma. The patient showed no signs of recurrence 8 months after surgical excision. Including the present case, 35 cases of sialolipoma have been reported in the English literature. Of these 35 cases, 16 cases were located in minor salivary glands. Gender was identified in 14 of these cases with 4 males (28.5%) and 10 females (71.5%). The age distribution was from 27 to 84 years (mean, 61.6 years) and the tumor size ranged from 0.9 to 4 cm (mean, 1.7 cm). The most frequently reported clinical presentation was of a painless swelling (56.3%).     

Distinctive Pattern of Glial Fibrillary Acidic Protein Immunoreactivity Useful in Distinguishing Fragmented Pleomorphic Adenoma, Canalicular Adenoma and Polymorphous Low Grade Adenocarcinoma of Minor Salivary Glands

Objectives Immunohistochemistry (IHC) can be helpful in the diagnosis of minor salivary gland neoplasms including those that have been incisionally biopsied or fragmented during surgery that do not contain key diagnostic features on hematoxylin and eosin sections. IHC has been used as an adjunct to distinguish among many salivary gland neoplasms using both qualitative and quantitative methods. The objective of this study was to determine whether a distinctive immunoreactivity staining pattern to GFAP can be consistently observed among three selected minor salivary gland neoplasms and thus serve as a diagnostic adjunctive procedure. Study Design Glial fibrillary acidic protein (GFAP) reactivity was examined among 78 minor salivary gland neoplasms: 27 canalicular adenomas (CAA), 21 pleomorphic adenomas (PA) and 30 polymorphous low grade adenocarcinomas (PLGA). Each case was evaluated by two oral and maxillofacial pathologists (OMP) blinded to the diagnosis. Consensus was reached on the pattern of GFAP reactivity among the neoplastic cells and on the similarities and differences among the cases. Results Ninety-six percent (96%) of CAAs demonstrated a distinctive linear immunoreactive pattern among cells in proximity to connective tissue interface. All (100%) PAs demonstrated diffuse immunopositivity within tumor cells. All (100%) PLGAs showed little or no intralesional reactivity and no peripheral linear immunoreactivity. Additional challenge cases were examined by outside OMPs to demonstrate the utility of these findings. Conclusions This study demonstrates that the pattern of GFAP immunoreactivity may be an adjunct to diagnosis among PA, CAA and PLGA. The pattern of distinctly linear GFAP immunoreactivity at the tumor/connective tissue interface in CAA has not been reported previously. This distinctive feature may permit the pathologist to differentiate among CAA, PA and PLGA when an incisional biopsy and/or fragmentation cause key diagnostic features to be absent. Because each of these neoplasms requires a different treatment approach, this can be of major significance.     

Metastatic hepatocellular carcinoma to the parotid gland: Case report and review of the literature

INTRODUCTIONHepatocellular carcinoma, the most frequent primary hepatic tumor, metastasizes in more than 50% of cases. However, parotid gland metastatic HCCs are very uncommon. We report a patient in whom the finding of a left parotid mass revealed metastatic HCC.PRESENTATION OF CASEA thirty-six-year-old male presented with a round palpable left neck mass that persisted for 3 months. He had received right hemihepatectomy for hepatocellular carcinoma (HCC). Preoperative evaluation revealed a benign tumor of the parotid gland. We performed superficial parotidectomy. Metastatic hepatocellular carcinoma of the parotid gland was diagnosed.DISCUSSIONAlthough HCC metastases to the oral cavity have been reported, to date, only 4 cases HCC metastasis to the parotid gland have been reported. Although clinicians and cytopathologists alike both agree that salivary gland fine needle aspiration biopies (FNABs) are highly useful and safe diagnostic alternatives to biopsies and resections, we believe that in specific clinical situations, awareness of potential diagnostic pitfalls in salivary gland FNAB is a necessary part of the microscopic interpretations of these lesions.CONCLUSIONAlthough rare, since HCC can metastasize to the parotid gland, high suspicion should be maintained in a patient presenting with a parotid mass with a history of HCC. In addition, since potential diagnostic pitfalls in salivary gland fine-needle aspiration (FNA) biopsies exist, incisional or excisional biopsy may be necessary for definite diagnosis of metastatic HCC to the parotid gland.