Ductal papillomas of salivary gland origin: A report of 19 cases and a review of the literature.

The term ductal papilloma is used to identify a group of 3 rare benign papillary salivary gland tumors known as inverted ductal papilloma, sialadenoma papilliferum, and intraductal papilloma. They represent adenomas with unique papillary features and arise from the salivary gland duct system. We describe the clinical and microscopic features of 19 new cases of ductal papillomas and combine them with 116 cases previously published in the English-language medical literature. These 19 cases were composed of 13 inverted ductal papillomas, 3 sialadenoma papilliferums, and 3 intraductal papillomas. Collectively, these 19 ductal papillomas occurred most commonly in the sixth to eighth decade of life, with an average patient age of 54 years. Men predominated 1.37:1. The sialadenoma papilliferums presented as papillary lesions clinically with all 3 misinterpreted as squamous papillomas by the surgeons. The inverted ductal papillomas and intraductal papillomas appeared as submucosal nodules. The lip and the palate were the most common locations for inverted ductal papilloma and sialadenoma papilliferum, respectively. The sites for the 3 intraductal papillomas were the parotid papilla of the Stensen's duct, the upper lip, and the buccal mucosa. With light microscopy, inverted ductal papillomas appeared to arise from the excretory ducts near the mucosal surface, whereas intraductal papillomas appeared to arise from the excretory ducts at a deeper level. Sialadenoma papilliferum had a more complex histology, with a biphasic growth pattern of exophytic papillary and endophytic components. All 19 cases were treated by surgical excision. Follow-up information was available in only 2 cases, both inverted ductal papillomas. Neither case recurred during a 20-year period. Sialadenoma papilliferum, with a literature-derived recurrence rate of 10% to 15%, seems to assert a more significant biological behavior than inverted ductal papilloma and intraductal papilloma. Our findings justify the separation of these 3 entities based on clinical and histologic parameters. They are tumors of adulthood and, in addition, have in common their relationship to the excretory duct system, their anatomical distribution, and their general behavior.     

Relative frequency of intra-oral minor salivary gland tumors: a study of 380 cases from northern California and comparison to reports from other parts of the world

BACKGROUND: The relative frequency of individual intra-oral minor salivary gland tumors (IMSGT) is not well documented in the literature. The aim of this study was to determine the relative frequency and distribution of IMSGT in an oral pathology biopsy service and to compare the data with similar studies from different parts of the world. METHODS: Files from the Pacific Oral and Maxillofacial Pathology Laboratory of the University of the Pacific, San Francisco, California served as a source of material for this study. Files were systematically searched for all cases of IMSGT during a 20-year period. Tumors were classified according to the 2005 WHO classification of salivary gland tumors. RESULTS: IMSGT were identified in 380 (0.4%) cases of 92 860 accessed. This is the largest series of IMSGT from one source reported in recent years. Of the 380 tumors, 224 (59%) were benign and 156 (41%) were malignant. Of the benign tumors, pleomorphic adenoma (PA) was the most common (39.2%), followed by cystadenoma (6.3%), canalicular adenoma (6.1%), ductal papillomas (4.4%), basal cell adenoma (1.6%), and myoepithelioma (1.3%). Of the malignant tumors, mucoepidermoid carcinoma was the most common (21.8%), followed by polymorphous low-grade adenocarcinoma (7.1%), adenoid cystic carcinoma (6.3%), adenocarcinoma, not otherwise specified (NOS; 2.1%), acinic cell carcinoma (1.6%), clear cell carcinoma, NOS (1.0%), and carcinoma ex PA (0.5%). CONCLUSIONS: Studies related to the relative frequency of individual IMSGTs from different parts of the world are difficult to compare because many studies are outdated, the number of cases is small, the list of tumors is limited, and new entities are not included. To determine the true relative frequency, more studies should be conducted, on a large number of cases from one source, by experienced pathologists in the field of salivary gland tumors.     

Inverted ductal papilloma arising from the buccal minor salivary gland: A case report and immunohistochemical study

Oral inverted ductal papilloma is a rare, benign epithelial tumor that exhibits an endophytic growth pattern and is found almost exclusively in the minor salivary glands. We report on a case of inverted ductal papilloma in the buccal mucosa. We also performed an immunohistochemical study. The tumor cells were positive for cytokeratin and epithelial membrane antigen, while negative for calponin, S-100 protein, α-SMA, vimentin, and desmin. This result indicated that the lesion arises from the excretory duct near the oral mucosal surface but not the myoepithelial cells. In addition, Ki-67 labeling index of 3.96% indicated the low level of proliferation.     

Oral inverted ductal papilloma associated with condyloma acuminata and HPV in an HIV+ patient

Oral inverted ductal papillomas are rare benign tumours of minor salivary glands. A case that appeared in a middle-aged HIV+ woman simultaneous to two condyloma acuminata on the lips is described. The presence of human papilloma virus (HPV) 11 DNA was demonstrated in all the samples using polymerase chain reaction. HPV could play an important role in the aetiology and pathogenesis of these lesions in this patient.     

4种涎腺肿瘤及人胚颌下腺免疫组化对比研究

目的：探讨涎腺肿瘤细胞的形态分化、组织发生及其异质性。方法：采用光镜、免疫组化技术对８例腺淋巴瘤、３例嗜酸性腺瘤、１２例多形性腺瘤及２例上皮－肌上皮癌进行研究,同时观察了５６例胚胎颌下腺组织作为对照。结果：多形性腺癌中导管样结构系肿瘤性闰管结构和肿瘤性分泌管结构;腺淋巴瘤和嗜酸性腺瘤中导管结构系肿瘤性分泌管结构;上皮－肌上皮癌中导管结构系肿瘤性闰管结构;涎腺肿瘤组织结构及抗原表达复杂多样,与人胚涎腺发生过程中结构及抗原表达有相似之处。结论：涎腺肿瘤的复杂性系致瘤因素作用于不同干细胞或同一干细胞不同分化阶段所致。     

Warthin瘤合并T淋巴母细胞淋巴瘤1例

Warthin瘤是一种良性唾液腺肿瘤,由导管上皮和淋巴样间质组成,迄今为止,有关Warthin瘤内淋巴成分恶变的报道罕见,且多恶变为B细胞淋巴瘤,合并T细胞淋巴瘤更尤为少见。本文报道1例Warthin瘤合并T淋巴母细胞淋巴瘤,旨在强调要对Warthin瘤内的淋巴组织进行仔细的光学显微镜评估,以识别隐匿性淋巴瘤的存在,减少误诊及漏诊并及时治疗。     

Immunolocalization of α2, α5, and α6 integrin subunits in salivary tissue and adenomas of the parotid gland

The localization of the integrin subunits α₂, α₅, α₆ was studied immunohistochemically in samples of normal salivary gland and in a series of 8 pleomorphic adenomas, 5 Warthin's tumors, and 2 basal cell adenomas. In normal salivary tissue, acinar and ductal cells expressed α₂ and α₆ chains at the basal cell pole facing the basement membrane. α₂ also localized at sites of cell-cell contact. No staining of the epithelial component was seen with α₅. The polarized expression of α₂ and α₆, subunits was retained in salivary adenomas. These subunits were present at the basal cell pole of solid nests, tubules and ducts of pleomorphic adenomas, as well as of the basal layer of the epithelium of Warthin's tumor, and of the trabecular structures of basal cell adenomas. The α₅ subunit was consistently expressed only by cells embedded in the myxoid or chondroid matrix of pleomorphic adenomas. We conclude that the pattern of a integrin subunit expression in salivary adenomas may be related to the "epithelial" or "mesenchymal" phenotype of the neoplastic cells.     

Localization of lysozyme mRNA in the labial salivary glands by in situ hybridization in Sjögren's syndrome

Abstract – In this study, lysozyme mRNA in labial salivary glands has been localized with in situ hybridization technique using 35S-labeled hen lysozyme cDNA (cDNALZM) as a hybridization probe in normals and in patients with Sjögren's syndrome. 35S-DNALZM:mRNA hybrids were detected only in acinar serous cells, although lysozyme was identified in ductal cells using immunohistochemical techniques. Our results suggest that the serous acinar cells are the only site of lysozyme synthesis in small salivary glands. The presence of lysozyme in ductal cells may be a result of reabsorption from the saliva or concentration from the blood or surrounding tissues.     

Incidental Langerhans cell histiocytosis of the parotid gland resembling marginal zone B-cell lymphoma: a case report

Langerhans cell histiocytosis (LCH) manifesting as a parotid gland mass is an extremely rare clinical presentation. We report a case of LCH involving the bilateral parotid glands in an 81-year-old Japanese female. Pathologically, the lesion was characterized by numerous lymphoid follicles, dense lymphoplasmacytoid infiltrate and cystic dilatation of the parotid gland duct in addition to nodular and diffuse proliferation of LCs. Moreover, both LCs and small T-lymphocytes invaded the ductal epithelium forming a lymphoepithelial lesion-like morphology. The present case indicates that LCHs should be added to the different diagnosis for marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue type involving the salivary glands.     

Immunohistochemical studies of organic anion transporters and urate transporter 1 expression in human salivary gland

Abstract

Background. Various substances including uric acid, organic acids and drugs are transported by organic anion transporters (OATs) in the kidney. In addition, a member of the OAT family, urate transporter 1 (URAT1), is involved in the reabsorption of uric acid from the renal tubule. Benzbromarone inhibits URAT1 to block uric acid reabsorption. Methods. Our group previously observed higher salivary uric acid levels than serum levels in patients taking benzbromarone, and reported the possible existence of URAT1-like uric acid excretion mechanism in the salivary gland. The purpose of this study was to elucidate the uric acid excretion mechanism in salivary gland tissues using rabbit anti-human OAT1-4 and URAT1 polyclonal antibodies with EnVision^? system. Results. In the salivary gland, OAT1 was expressed in ductal cells. OAT2 was found in both ductal cells and serous acinar cells and weak expression was also observed in several nuclei. OAT3 expression was observed in serous acinar cells and nuclei and OAT4 was expressed only in ductal cells. URAT1 expression was observed in the cytoplasm of ductal cells and strong punctuate staining was seen in part of the supra-nuclear cytoplasm. The number of cells expressing URAT1 was smaller compared with OATs. In the kidney, however, OAT1-4 and URAT1 were strongly expressed on proximal renal tubules. Conclusions. The present study confirmed the existence of OAT1-4 and URAT1 in the salivary gland. These results may support the previous speculation that benzbromarone inhibits URAT1 to block uric acid reabsorption in the salivary gland, resulting in higher salivary uric acid levels than serum levels.     

涎腺导管癌临床病理和免疫组化研究

目的　探讨涎腺导管癌 ( SDC)的临床病理特点。方法　对 5例 SDC患者的临床和病理资料进行回顾性分析 ;用免疫组化方法检测细胞角蛋白 ( CK) ,平滑肌肌动蛋白 ( SMA)在 SDC中的表达 ,并与腺样囊性癌对照。结果　 1例术后 2年死于全身广泛转移 ,2例带瘤生存 2～ 5年 ,2例失访。5例 SDC镜下呈典型导管癌表现 ,侵犯神经、血管壁和周围的软组织。 2例曾误诊为腺样囊性癌。免疫组化 CK阳性 ,SMA阴性 ,示肿瘤细胞为导管上皮。结论　涎腺导管癌是一种高侵袭性的涎腺癌 ,易发生转移 ,预后差。彻底手术联合放疗和 (或 )化疗的综合治疗有助于延长生存期。     

Variation in the response to ductal obstruction of feline submandibular and sublingual salivary glands and the importance of the innervation

A variable response following ductal ligation of feline salivary glands corresponds to the human condition but contrasts with a predictable atrophy in obstructed salivary glands of rodents popularly used as a model for human salivary problems. The present investigation is concerned with a possible reason for the variable response, namely the preservation of the innervation. Ducts of feline submandibular and sublingual salivary glands were ligated with or without the inclusion of the chorda tympani. Inclusion led to a delayed initial response followed by progressive atrophy until the parenchyma was extremely atrophic, whereas avoidance of the chorda led to the variable response in which variable numbers of acini of a similar form to normal persisted. The results establish the atrophic effect of inclusion of the chorda tympani in ductal ligation and indicate the caution that should be exercised in the extrapolation of the rodent model to the human condition.     

Immunohistochemical study of the distribution of endogenous biotin and biotin-binding enzymes in ductal structures of salivary gland tumours

To clarify the pathologic value of endogenous biotin in the salivary gland, we examined in a series of neoplasms of the salivary gland by immunohistochemical staining the distribution of endogenous biotin and of biotin-binding enzymes, namely, acetyl CoA carboxylase (AC), which is a cytosolic enzyme, and pyruvate carboxylase (PC), which is a mitochondrial enzyme. In pleomorphic adenoma, we found biotin and PC in ductal epithelial elements, while AC was found mainly in myoepithelial elements. Carcinoma ex pleomorphic adenoma, adenocarcinoma and mucoepidermoid carcinoma were frequently immunopositive for biotin, PC and AC, while adenoid cystic carcinoma was rarely immunopositive for biotin, PC or AC. These results indicate that endogenous biotin might be associated with the mitochondrial enzyme, which is present at high levels in ductal cells of the salivary gland. However, the neoplastic cells in adenoid cystic carcinoma seemed to have an unusual expression of biotin and related enzymes.     

P63蛋白在人胚涎腺发育中的表达及意义

目的探讨P63蛋白在涎腺上皮干细胞发育中的作用及干细胞存在的可能位置。方法收集不同发育阶段的合法流产人胚24例、正常涎腺组织标本10例,对切片行常规HE染色和免疫组织化学SP法染色。结果组织切片见涎腺发育从早期上皮芽增生分支形成树枝状上皮条索,然后形成管腔,最终分支末端细胞增生膨大分化为导管、肌上皮和腺泡细胞。P63的表达从胚胎早期上皮芽中的广泛阳性表达到阳性细胞逐渐减少,呈现区域化散在分布特征,正常成人涎腺中阳性表达细胞仅少量存在于闰管、分泌管、排泄管管壁基底侧。结论P63在维系涎腺上皮性干细胞的生存和分化状态中可能具有重要作用,正常成人涎腺上皮性干细胞仅少量存在于导管管壁基底侧。     

颌下腺导管内涎石取出术的疗效分析

目的研究颌下腺导管内涎石取出术的远期疗效和腺体功能的恢复情况。方法对颌下腺导管内涎石取出术的患者进行术后随访2~15年,观察患者手术的远期疗效和腺体分泌功能的恢复情况。结果单纯取石术后,患者的近期疗效和腺体分泌功能恢复情况较好,而远期疗效和分泌功能呈下降趋势;腺体感染和结石的复发是影响的关键因素,患者的年龄也有一定影响。结论在颌下腺导管内结石的治疗中,早期取出结石术后,合理使用抗生素,辅助使用催涎剂,有效阻断结石形成前的一系列病理过程,控制炎症的发生、发展,以及采用颌下腺区域的局部按摩,对颌下腺导管内涎石的治疗能取得较好的疗效。     

Nucleolar organizing regions (NORs) in pleomorphic adenomas of the salivary glands

The number of nucleolar organizing regions (NORs) can be used to reveal the degree of cell activity or metabolism in histopathology specimens. Forty-one cases of pleomorphic adenomas of the salivary glands were studied with a silver nitrate colloid staining method to demonstrate the NORs in the cells of the solid and duct pattern areas and in chondroid areas. Acinic and intercalated ducts cells of the surrounding normal salivary glands were also examined. The data was analyzed statistically and it was found that the cells in the solid/ductal areas had a higher cellular activity than the chondroid cells. This difference is probably directly related with the possibility of malignant transformation and the incidence of the different types of malignant pleomorphic adenomas. Furthermore, it is concluded that the number of NORs in pleomorphic adenomas to define malignancy should be done discriminating between the different histologic patterns because of their different metabolic activities.     

涎腺黏液腺癌1例

黏液腺癌是一种少见的上皮性恶性肿瘤,好发于阑尾、胰腺、乳腺等部位,发生于涎腺者罕见。本文报道1例发生在涎腺的黏液腺癌,并对相关文献进行复习。     

Ultrastructural localization of microliths in salivary glands of cat

Although microliths occur in normal human salivary glands and may be an aetiological factor of sialadenitis, little is known of their natural history. In an attempt to remedy this, we investigated a large archival collection of normal and experimental feline parotid, submandibular and sublingual salivary glands. In submandibular and sublingual glands, microliths were detected ultrastructurally in: all types of acinar secretory cells; myoepithelial cells; ductal cells; lumina; intercellular spaces; basement membrane; stroma; macrophages; multinuclear giant cells; and neutrophils. Microliths were not detected ultrastructurally in parotid glands. Microliths appear to form in acinar cells during autophagy and in stagnant secretory material in lumina. Microliths appear to be removed by secretion in the saliva, discharge from cells laterally and basally, and engulfment by macrophages. There appears to be a turnover of microliths, which possibly is upset by secretory inactivity with a resulting accumulation that leads to localized obstruction and sialadenitis.     

Ultrastructural and immunohistochemical observations of a true malignant mixed tumor (carcinosarcoma) of the tongue

True malignant mixed tumor (TMMT) of salivary glands, with both carcinomatous and sarcomatous components, is exceedingly rare. We offer a case of TMMT in a 79-yr-old man, which may represent the first report example of this unusual neoplasm arising in the tongue. The carcinomatous component was mainly of solid basaloid carcinoma with focal glandular differentiation, while the sarcomatous component was composed of pleomorphic elements such as chondrosarcoma, myxosarcoma and fibrosarcoma. Carcinoma cells at the periphery of solid nests occasionally merged into these sarcomatous elements. Immunohistochemically, basaloid carcinoma cells showed positive reaction for both low molecular weight cytokeratin and S-100 protein, whereas carcinoma cells lining ductal spaces were positive for a wide spectrum of keratin and EMA. The sarcomatous elements revealed the presence of vimentin and S-100 protein. Ultrastructurally, basal lamina-like material and/or mucoid precipitates often accumulated separating the tumor cells from each other singly or into a few cell group. Some sarcomatous cells assumed the myoepithelial features, such as the presence of microfilament bundles with dense bodies and pinocytotic vesicles along the cell periphery. These findings may indicate that TMMT shares a common histogenesis with pleomorphic adenoma.