Sialadenoma Papilliferum Research Articles

Intraductal Papillary Mucinous Neoplasms of Minor Salivary Glands With AKT1 p.Glu17Lys Mutation.

The spectrum of low-grade intraductal papillary proliferations of the salivary glands is heterogenous, and reproducible morphologic diagnostic criteria have not yet been established. Recognized types are sialadenoma papilliferum, inverted ductal papilloma, and intraductal papilloma, but some lesions have been possibly included in the morphologic spectrum of cystadenoma or low-grade intraductal carcinomas. We herein present detailed morphologic, immunophenotypic, and genotypic features of 3 minor salivary gland neoplasms affecting 2 men (aged 65 and 71 y) and 1 woman (aged 78 y). They ranged in size from 1 to 2.5 cm. All tumors showed atypical papillary intraductal growth that presented either as uninodular/unicystic lesions (intraductal papilloma-like; n=2) or as a discontinuous growth along the ductal system in a manner similar to pancreatic intraductal papillary mucinous neoplasm (n=1). Variable cytologic and architectural atypia was observed, ranging from bland intraductal papilloma-like features, to areas mimicking atypical ductal hyperplasia and low-grade ductal carcinoma in situ of the breast. Amplicon-based massive parallel sequencing revealed an identical AKT1 p.Glu17Lys mutation in all 3 cases, but absence of concurring mutations in other genes of the RAS or PI3K pathway. This small series represents the first genetic study on salivary intraductal papillary neoplasms. Our cases showed significant variation in the degree of cytologic and architectural atypia, which overlaps with intraductal papillomas at the one end and with low-grade intraductal carcinoma at the other end of the spectrum, suggesting a disease continuum. As the full biological and morphologic characteristics of these ductal papillary lesions remain to be defined, the noncommitted term "intraductal papillary neoplasms" might be more appropriate. Our novel genetic findings mirror similar activating mutations of AKT1 and other PI3K pathway members in intraductal papillary lesions of the breast and anogenital glands.

Rare occurrence of sialadenoma papilliferum at lower lip

Sialadenoma papilliferum (SP) is a rare intraoral neoplasm that superficially resembles any other papilloma. It mainly originates from the excretory ducts of minor salivary glands (MSGs). Despite the MSGs being widely distributed from the lips and nose up to the tonsils and bronchi, the handful number of SPs reported so far predominantly affected the palate; while many other locations remain relatively secluded. In here, we describe the clinical and pathological features of one such SP located at lower lip, a never been reported site in English literature.

Sialadenoma papilliferum in the buccal mucosa detected on 18F-fluorodeoxyglucose-positron emission tomography

An incidental discovery of an accumulation of 18F-fluorodeoxyglucose (FDG) in the oral cavity of patients with malignant tumours on FDG positron emission tomography (PET) can pose a problem regarding the differential diagnosis of metastatic lesions. Large accumulations can often be found even when tumours in the salivary or thyroid gland are benign, so FDG-PET is limited in its ability to differentiate between benign and malignant disease. This report describes a rare case of sialadenoma papilliferum in the buccal mucosa that was discovered incidentally on FDG-PET in a patient with multiple metastases to bone after an operation for rectal cancer.

Sialadenoma papilliferum – ein seltener benigner endobronchialer Tumor

HISTORY AND PRESENTATION AT ADMISSION: An 82-year-old male patient presented with a 3 week history of exercise-induced dyspnea, productive cough and left sided chest pain. Computertomography of the chest revealed an occluding endobronchial tumor in the left main bronchus with enlarged mediastinal lymph nodes, mediastinal shift and post-stenotic peribronchitis. The tumor was removed completely with an optical forceps in rigid bronchoscopy. Histologically an endobronchial sialadenoma papilliferum was diagnosed. Benign tumors of the lower airways are rare. They cannot be distinguished reliably from malignant tumors by their endoscopic and radiologic appearance. Sialadenoma papilliferum is an extremely rare benign salivary gland tumor which is characterized by coexisting glandular and (pseudo) papillar formations. It occurs mainly in the oral cavity. The relapse rate is 10-15%. In single cases a malignant transformation may appear.

Intraductal Papilloma: Atypical Presentation

Ductal papillomas have unique papillary features arising from the salivary gland duct system. They comprise three rare benign adenomas, namely, inverted ductal papilloma, sialadenoma papilliferum, and intraductal papilloma. Intraductal papilloma is an extremely rare benign salivary gland tumor that occurs most commonly in the minor salivary glands. Here, we are presenting a case report of intraductal papilloma in an 18-year-old patient.

Sialadenoma Papilliferum of the Hard Palate: A Case Report

Sialadenoma papilliferum (SP), which forms a group of ductal papillomas with inverted ductal papilloma and intraductal papilloma, is a rare benign papillary salivary gland tumor usually discovered as a whitecolored exophytic mass in the oral and maxillofacial region. Since the first 2 cases of SP in the parotid gland and the palate were reported in 1969, only 48 cases of the tumor have been reported in the Englishlanguage literature. The clinical appearances of the tumor in the oral cavity are similar to papilloma. However, the tumor consists of dilated salivary duct-like structures with papillary and exophytic proliferation of doubleor multiple-layered epithelium, as well as a concomitant overgrowth of surface squamous epithelium that exhibits verrucous acanthotic hyperplasia. Microscopic studies of SP were performed to better understand the cell origin of the tumor, and the findings suggested the lesion to originate from the intercalated duct cells. However, other studies reported that the tumor cells appeared to exhibit characteristic features of various cell types of the salivary gland duct apparatus. Furthermore, immunohistochemical analyses of SP showed the cell origin of the tumor to be the excretory ducts. However, the cell origin of the umor is still unclear. This report presents a new case of SP of the hard alate occurring in a 62-year-old man and includes a iscussion of the cell origin of the tumor.

Sialadenoma Papilliferum: Clinical Misdiagnosis with a Histological Decree

Sialadenoma papilliferum is a rare salivary gland tumor clinically resembling papilloma originating probably from the excretory duct. It is characterized by a biphasic growth pattern of exophytic squamous component and endophytic glandular component. We report a rare case of sialadenoma papilliferum in the floor of the mouth with epithelial dysplasia with pertinent review of literature. The present case highlights the importance of keeping sialadenoma papilliferum as a differential diagnosis of exophytic papilliferous oral lesions and the need to explore the etiology and malignant potential of the tumor.

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.

Sialadenoma papilliferum: a case report and immunohistochemical study review

Sialadenoma papilliferum (SP) is a rare benign neoplasm that normally arises from the minor salivary glands, particularly in the palate. SP is normally encountered in older men with an exophytic papillary surface growth. In the present study, an SP of the hard palate of a 69-year-old woman was examined immunohistochemically. Myoepithelial cell markers, such as S-100, smooth muscle actin and vimentin, were observed in the basal or luminal layer of tumor cells, indicating that myoepithelial cells participate in the pathogenesis of SP. In addition, cytokeratin 7 was also strongly detected in the tumor cells, suggesting that excretory ductal epithelial cells have a role in its histogenesis. A review of the literature of immunohistochemical studies on SP showed that the expression and co-expression of cytokeratins and myoepithelial cell markers have been reported in tumor cells. These results suggested that excretory duct cells and myoepithelial cells participate in the pathogenesis of SP.

Bronchial Sialadenoma Papilliferum: A Very Rare Cause of Hemoptysis

Purpose: This case is only the third case of the Sialadenoma papilliferum of the bronchial system. This is a extremely rare tumor of the bronchial system. This report highlights once again the histo-pathological difficulties of diagnosing such a rare tumor. Patients and methods: A 53 year old woman with a 3 weeks history of a productive cough associated with hemoptysis presented to the Community hospital Stuttgart (Teaching Hospital of the University of Tubingen). A thoracic CT revealed a solid mass in the right lower lobe with 10 mm diameter. In the community hospital a bronchoscopic biopsy was suspicious for an adenocarcinoma of the lung. Result: The patient was transferred to our institution for thoracic surgery and a right lower bilobectomy with semicircular intrapericardial vessel resection and total nodal resection was performed. By immune-histochemical analysis , the removed tumor (size 10 mm) revealed to be a benign adenoma from the seromucosal bronchial glands, which is a very rare benign tumor of the Sialadenoma papilliferum type. All of the removed lymph nodes were analyzed and showed no signs of malignancy. Conclusion: At present there have been reported only two cases of the pulmonary Sialadenoma papilliferum in the literature. This case report represents the first case of pulmonary Sialadenoma papilliferum in Germany and western Europe. The biologic behavior of this tumor still remains unknown.

Sialadenoma papilliferum of the bronchus: Rare tracheobronchial tumor of salivary gland type

Presented herein is the second case of sialadenoma papilliferum of the bronchus in a 75-year-old man. The bronchial tumor had an exophytic papillary lesion protruding into the lumen of the ostial region of the right superior lobe bronchus on chest CT and bronchoscopy. Grossly, the resected tumor was pedunculated, 5 x 3 x 3 mm in size and consisted of both an exophytic papillary lesion protruding into the bronchial lumen and a glandular component in the bronchial submucosa. Histologically, the papillary structures were lined by ciliated or non-ciliated columnar epithelium and metaplastic non-keratinizing stratified squamous epithelium. The submucosal glandular components were well circumscribed beneath and in continuity with the papillary lesion. The glandular components of cysts and duct-like structures were lined by a double-layer epithelium composed of luminal cells and basal cells. Sialadenoma papilliferum is a rarely recognized salivary gland tumor, mostly occurring at intraoral mucosal sites including the hard and soft palate and buccal mucosa. There have been sporadic cases of sialadenoma papilliferum of the esophagus, nasopharynx and nasal cavity. To the authors' knowledge only one case of sialadenoma papilliferum of the bronchus has been reported previously.

Mucoepidermoid Carcinoma Arising in a Background of Sialadenoma Papilliferum: A Case Report

Sialadenoma papilliferum is a rare tumor, primarily of minor salivary gland origin, first described by Abrams and Finck (Cancer 24:1057–63, 1969). It is both an exophytic and endophytic papillary lesion histologically resembling syringocystadenoma papilliferum of sweat gland. The tumor is considered benign although rare recurrent cases have been reported. Three cases of malignant transformation of sialadenoma papilliferum have been described in the literature. We report a high grade mucoepidermoid carcinoma arising in a background of sialadenoma papilliferum, at the base of the tongue, an unusual location for minor salivary gland neoplasms. Eleven months after excision and nodal dissection, there is no evidence of recurrence or metastasis.

Intraoral intraductal papilloma: a case report

Ductal papillomas have unique papillary features arising from the salivary gland duct system. It comprises three rare benign adenomas, namely, inverted ductal papilloma, sialadenoma papilliferum and intraductal papilloma. Here the first case of intraductal papilloma developed in the minor salivary gland of the vestibule of the oral cavity in a 71-year-old Chinese female living in a nursing home is described. This case is worthy of clinical investigation as it presents as an intraoral swelling and is mistakenly regarded as the result of a periapical pathosis. It also emphasises that a nurse or an oral hygienist who is usually the first-line oral carer of the residents of a nursing home, should be trained to perform the daily dental check and request a dentist's services when necessary.

Sialadenoma papilliferum of nasal cavity: case report

We report a rare case of sialadenoma papilliferum. A case report of sialadenoma papilliferum is presented. A 67-year-old woman presented with recurrent epistaxis. She was found to have an exophytic, well circumscribed mass on the nasal septum mucosa. The lesion was completely excised. A diagnosis of sialadenoma papilliferum was made based on the characteristic histological pattern. Follow up showed no evidence of recurrence. Subsequently, the patient remained well without complaint of epistaxis. To our knowledge, this is the first report in the world literature of sialadenoma papilliferum of the nasal cavity. This case indicates that this rare tumour can present with epistaxis, and can be resolved by means of total excision.

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 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 CASE OF SIALADENOMA PAPILLIFERUM OF THE ESOPHAGUS

Sialadenoma papilliferum of the esophagus is an extremely rare benign tumor that derives from the submucosal gland duct of the minor salivary gland. A 45‐year‐old man underwent upper gastrointestinal series. A 13 mm diameter esophageal polyp was suspected in the mid esophagus. Endoscopy examination revealed a pedunculated polyp arising from the posterior wall of the esophagus. The head of the polyp was covered with exudate and was slightly nodular but the pedicle had normal mucosa. We performed endoscopic mucosal resection (EMR) and the histological examination of the polyp showed that it was compatible with sialadenoma papilliferum of the esophagus.

Sialadenoma papilliferum of the buccal mucosa: A case report of a rare localization and review of the literature

AbstractSialadenoma papilliferum is a rare, benign salivary gland tumor which most commonly arises in the palate. It is an exophytic papillary proliferation that involves mucosal surface or epidermis and salivary duct epithelium. Approximately 11% of sialadenoma papilliferum cases were reported in the buccal mucosa. The sialadenoma papilliferum is often misinterpreted as squamous papilloma by surgeons.In this case, sialadenoma papilliferum located in the buccal mucosa of a 64-year-old man is reported. The mass was asymptomatic, slow-growing, movable and was clinically similar to the lipoma. A surgical excision was performed and after 22 months there has been no recurrence.

Intraductal Papilloma of Minor Salivary Glands : Case Report

The authors present a case of intraductal papilloma. It is the rarest among ductal papillomas, which is a group of salivary gland neoplasms that comprise three different histological sub-types : intraductal papilloma, inverted duct papilloma, and sialadenoma papilliferum. Intraductal papilloma is localized mainly in the ducts of minor salivary glands but it is rare in the parotid gland, the submandibular gland, and the sublingual gland. Patients' mean age is about 50-60 years. The growth is slow and asymptomatic. The overlying mucosa seems to be normal. Microscopically, the intraductal papilloma shows fibrovascular papillae surrounded by columnar or cuboidal epithelial cells to form a mass protruding into the duct lumen to create a cyst-like cavity ; usually mitoses are absent, but sometimes low grade cellular atypia is seen in a few areas. Preoperatory diagnosis is difficult, and only histopathologic examination is definitive. Fine needle aspiration biopsy is the best method for preoperative diagnosis, even if sometimes its results are deceptive. Excision is the resolutive therapy, and no recurrences after surgical excision have been reported. The purpose of this paper is to emphasize the importance of differential diagnosis of various ductal papillomas with this single case of intraductal papilloma.

Sialadenoma papilliferum: immunohistochemical study

Sialadenoma papilliferum (SP) is a rare benign tumour of salivary gland origin, which has been included among the ductal papillomas in the latest classification of tumours by the World Health Organisation. Two SP from the minor salivary gland of the palate of middle age patients were presented and studied by immunohistochemical. Our results showed presence of cytokeratins (CKs) 13, 14, 7, 8, 19 and absence of vimentin and smooth muscle actin. This immunoprofile is similar to the excretory duct of salivary gland.