Inverted ductal papilloma of minor salivary gland: case report with immunohistochemical study and literature review

Inverted ductal papilloma (IDP) is a type of ductal papilloma arising in ducts of minor salivary glands. Very few cases, and no cases in Japan, have been reported. Reported herein is a case of IDP with a review of the literature. The patient was a 49-year-old man presenting with a lump in the right buccal mucosa of the premolar area of the mandible. The tumor was excised en bloc after a biopsy diagnosis of IDP. On the surface of the covering epithelium, an opening was seen to be filled with mucinous material. On cut surface the opening led to the tumor cavity. The major portion of the tumor parenchyma was made up of papillary proliferation of basaloid squamous cells. Some crypts, microcysts, and mucous cells were seen. There were no findings suggestive of a malignant tumor. The patient's postoperative course was uneventful and there has been no recurrence after 1 year's follow up. Immunohistochemical analysis of the present case supports the hypothesis that IDP originates from squamous metaplasia and proliferation of minor salivary gland duct cells.     

Polymorphous low-grade adenocarcinoma of the salivary glands

Six cases of polymorphous low-grade adenocarcinoma (terminal duct adenocarcinoma) of the minor salivary glands are presented. In all but one there was a history of a painless intra-oral mass of fairly long duration. The histopathological appearances were characterized by cytological uniformity in a variety of morphological patterns, including tubular, solid, fascicular and cribriform areas. At a cellular level, the tumours possessed regular, often vesicular nuclei and generally eosinophilic cytoplasm. Five of the patients are still alive, although one had recurrent disease 16 years after her original operation; none died of their tumour. These findings are compared with those of six salivary adenoid cystic carcinomas, a neoplasm with many similar histological features, but with a much worse prognosis. The microscopic differences were mainly cytological and, to a lesser extent, morphological. The immunohistochemical reactions of the two tumours were not sufficiently dissimilar to be of practical value. Polymorphous low-grade adenocarcinoma has only rarely been reported in Britain, but we believe it deserves wider recognition as a distinct clinicopathological entity and, in particular, separation from adenoid cystic carcinoma.     

Polymorphous low-grade adenocarcinoma of submandibular gland origin

A case of polymorphous low-grade adenocarcinoma (PLGA) in the submandibular gland is reported. A 72 year old woman presented with a 5 year history of a gradually expanding tumor in the submandibular region. The surgical specimen revealed a relatively well demarcated tumor, 35 × 35 × 20 mm in size. Macroscopically, necrosis and hemorrhage were not seen in the solid tumor. Histologically, the tumor growth pattern was variable, composed of tubular, papillary, solid, trabecular and cribriform structures. Immunohistochemically, some tumor cells were positive for epithelial membrane antigen (EMA), S-100 protein, keratin, and carcino-embryonic antigen (CEA). Electron microscopically, prominent microvilli projected into the luminal spaces, and basal lamina and hemidesmosomes were seen in the tumor cells adjacent to the connective tissues. The submandibular gland is an extremely rare location for PLGA. To the authors' knowledge, this is the first case of its kind reported in the English literature.     

Oral and salivary gland angiosarcoma: a clinicopathologic study of 29 cases.

Angiosarcomas of the oral and salivary gland area are extremely rare, mostly presented as case reports. We wanted to study the clinicopathologic features of a series of oral and salivary gland angiosarcomas. Cases coded as "angiosarcoma" were retrieved from the Oral and Maxillofacial Pathology Department of the Armed Forces Institute of Pathology. Patient folders and pathology were reviewed and recorded; immunohistochemistry and follow-up were obtained. Inclusion required oral or salivary gland location, vasoformative growth, cytologic atypia, mitoses, and vascular markers. Skin, bone, and subcutaneous angiosarcomas were excluded. Primary and secondary (metastatic) oral angiosarcomas were included. The 22 primary angiosarcomas involved tongue (n = 9), parotid (n = 4), lip (n = 4), submandibular gland (n = 3), and 1 each of soft and hard palate. The 7 secondary angiosarcomas involved the gingiva (n = 4) and parotid gland (n = 3). Overall, patient ages ranged from 6-90 years (mean, 55 years). There were 15 males and 14 females. Symptoms included a mass with recent enlargement and bleeding. Tumor sizes ranged from 0.8-7.0 cm (mean, 2.6 cm). Histologically, all tumors were vasoformative; 86% had solid and 17% had distinctive papillary areas. Eight (28%) were classified as the epithelioid subtype. Immunohistochemical stains showed that the tumor cells were positive for Factor VIIIrag in 19/21, CD31 in 16/19, CD34 in 7/12, and Ulex in 1/1. Primary tumors were classified as low grade (n = 7, in all locations except salivary gland), intermediate (n = 7), and high grade (n = 8); all secondary tumors were high grade. Follow-up was available on 14/22 primary and 7/7 secondary angiosarcomas. Of primary tumors, two tongue angiosarcoma patients died at 1 and 9 years, but 4 were alive without disease over a mean of 7.3 years (range, 1-13 years). Four primary salivary gland angiosarcoma patients were alive without disease over a mean of 5.8 years (range, 1-14 years), and 1 had only a late (15 years) metastasis and death (at 20 years). Three primary lip angiosarcoma patients were without disease over a mean of 14.3 years (range, 13-16 years). Of secondary tumors, three salivary gland angiosarcoma patients died within 1 year, and all four secondary gingival angiosarcoma patients died of disease within 3 years. Assessing follow-up of primary oral and salivary gland angiosarcoma patients by grade, 5 patients with high-grade tumors had no evidence of disease over a mean of 7.6 years (range, 1-16 years), 3 patients with intermediate-grade tumors had no evidence of disease over a mean of 12.7 years (range, 11-14 years), 2 patients with intermediate-grade tumors died of disease at 9 and 20 years, 3 patients with low-grade tumors had no evidence of disease over a mean of 6.3 years (range, 1-14 years), and 1 patient with low-grade tumor died of disease at 1 year. Primary oral and salivary gland angiosarcomas, albeit rare, mostly involve the tongue, parotid gland, and lip of adults, often with relatively good outcome. Although the most common angiosarcoma morphology in this area is spindled vasoformative and solid, almost one third of oral and salivary gland angiosarcomas are the rare epithelioid angiosarcoma variant. Most gingival and few parotid angiosarcomas appear to be metastases from other locations, with many patients succumbing to death within 3 years. Despite predominantly high- or intermediate-grade morphology, patients with primary angiosarcoma of the tongue, salivary gland, and lip have a better prognosis than do patients with primary cutaneous or deep soft tissue angiosarcoma, including those patients with secondary oral and salivary gland involvement.     

涎腺恶性多形性腺瘤161例临床病理分析

目的分析涎腺恶性多形性腺瘤（malignant pleomorphic adenoma,MPA）的临床病理特征。方法对经病理复片诊断为MPA的161例临床病理材料进行回顾性分析。结果161例MPA中,152例为原发性肿瘤、7例为局部复发性肿瘤、2例为肿瘤颈淋巴结转移灶。152例原发MPA中发生于腮腺85例、腭部39例、颌下腺21例、颊部3例、唇部2例、磨牙后区1例、上颌骨1例;男性95例,女性57例,男女比为1．7：1;发病年龄为27～92岁,平均59岁。恶性成分的组织学类型包括肌上皮癌62例、非特异性腺癌59例、腺样囊性癌8例、黏液表皮样癌7例、导管癌3例、腺鳞癌3例、上皮-肌上皮癌3例、癌肉瘤3例、未分化癌2例、腺泡细胞癌1例、黏液腺癌1例。组织学高、中、低度恶性的分别为45、69、38例。同一肿瘤中恶性成分≤50％的为21例,〉50％的为131例。侵袭性、微侵袭性、非侵袭性癌分别为106、10、33例。侵袭性癌、组织学分级与肿瘤发生颈淋巴结转移之间有显著相关性（P值均〈0．01）。结论MPA多见于中老年男性,好发于腮腺;临床表现多为无痛渐大性肿块,以近期生长迅速为特征;组织学表现为常同时具有良性多形性腺瘤成分和恶性肿瘤成分,侵袭性癌多见,恶性成分常多于良性成分,恶性成分以肌上皮癌和非特异性腺癌为多见;侵袭性癌、组织学分级高度恶性者易发生颈淋巴结转移。     

Sarcomatoid salivary duct carcinoma of minor salivary gland: a rare case

A 35-year-old female presented with swelling in the soft palate. Fine needle aspiration cytology (FNAC) revealed pleomorphic adenoma, and on histopathological examination, it was diagnosed as carcinosarcoma/salivary duct carcinoma in the minor salivary gland, which was confirmed by immunohistochemical stains. We report this case for its rarity.     

Basal cell adenocarcinoma arising from the minor salivary gland in the soft palate: a case report

Basal cell adenocarcinomas (BCACs) of the oral minor salivary gland are very rare neoplasms. We report on an 86-year-old woman with BCAC arising from the minor salivary gland in the soft palate. Histologically, the tumor was located in the submucosa and showed microinvasion into the adjacent soft tissue without encapsulation. It contained tiny tumor islands with solid and tubular patterns, as well as myxoid stroma. The neoplastic cells were basaloid cells and were composed of large pale cells and small dark cells. They were positive for alpha-smooth muscle actin, cytokeratin 14, and vimentin in the periphery of the tumor island, showing a myoepithelial differentiation. The myxoid stroma was positive for alcian blue and colloidal iron. Apical membranes of the neoplastic cells were positive for MUC1 and CEA. The present case is the 14th documented case of oral BCAC (the fifth case of palatal BCAC).     

Cytology of low‐grade cribriform cystadenocarcinoma in salivary glands: Cytological and immunohistochemical distinctions from other salivary gland neoplasms

Low‐grade cribriform cystadenocarcinoma of the parotid gland is rare malignancy that is classified as a variant of cystadenocarcinoma. In routine cytologic slides from fine‐needle aspiration of a parotid gland, we found several pseudopapillary clusters comprising mucus‐producing cells. They included a few tumor cells having three‐dimensional nuclear atypia and slight hyperchromatism, although most of the tumor cells showed bland nuclei. Our initial cytological diagnosis was: “Indeterminate. Uncertain whether cystadenocarcinoma or cystadenoma.” The subsequent histological diagnosis was low‐grade cribriform cystadenocarcinoma. Immunohistochemical staining showed diffuse and strong reactivity for S‐100; tumor nests that were rimmed by p63⁺ cells, which suggests intraductal proliferation. Here, we report cytomorphological findings of this case, and discuss cytological and immunohistochemical distinctions between low‐grade cribriform cystadenocarcinoma and other salivary gland tumors, including a review of the literature. Diagn. Cytopathol. 2016;44:241–245. © 2015 Wiley Periodicals, Inc.     

胚胎发育不良性神经上皮瘤与皮质发育不良

目的观察胚胎发育不良性神经上皮瘤（DNT）的病理形态学及免疫组织化学特点,探讨其与皮质发育不良之间的关系以及组织来源。方法应用光镜和免疫组织化学EnVision法对14例DNT进行观察分析,并对患者进行长期随访。结果肿瘤位于颞叶的有11例,镜下由神经元和神经胶质成分混合构成,9例可见“特异的胶质神经元结构”。1例为简单型,8例为复杂型,5例为非特异型。11例标本充足的病例10例伴有皮质发育不良改变,表现为分子层和（或）白质内异位神经元数量增多（7例）,分子层内可见成行的外颗粒细胞层残留（4例）,脑皮质的结构异常（10例）,以及出现异常形态的神经细胞。免疫组织化学染色少突胶质样细胞（OLC）均呈现Olig2的免疫反应性,部分OLC表达nestin、微管相关蛋白2、神经丝蛋白、胶质纤维酸性蛋白,但神经元核抗原呈阴性。癫痫控制结果Ⅰ级12例,Ⅱ级2例,无肿瘤复发。结论DNT与皮质发育不良关系密切,应用免疫组织化学染色有助于DNT和皮质发育不良的诊断。     

Basal cell adenocarcinoma of minor salivary glands

Basal cell adenocarcinoma of minor salivary glands is a relatively rare slow-growing tumor with an infiltrating growth pattern. The infiltrating growth pattern and likelihood of vascular and perineural involvement distinguishes basal cell adenocarcinoma from basal cell adenoma. Other diagnostic considerations include adenoid cystic carcinoma and basaloid squamous carcinoma. Basal cell adenocarcinomas show strong immunoreactivity to cytokeratin 7 and variable myoepithelial staining with S100. It is necessary to differentiate basal cell adenocarcinoma from other basaloid cell tumors of the minor salivary glands because of the prognosis and potential differences in treatment, particularly adenoid cystic adenocarcinoma and basaloid squamous carcinoma. Surgical excision with a wide margin to ensure complete removal has been suggested as the primary treatment for basal cell adenocarcinoma. Radiotherapy has been proposed for lesions in the minor salivary glands because of the higher likelihood of vascular and neural invasion and for those that are diffusely infiltrative.     

Myoepithelial cells in salivary gland tumors. An immunohistochemical study

Normal salivary glands and 55 salivary gland tumors were examined by immunostaining (immunoperoxidase [IMP] and immunofluorescence [IMF]) to identify myoepithelial cells (MCs) and speculate on their role in the histogenesis of the tumors. The classic (C) MCs of normal salivary glands stained by IMP with antibodies to cytokeratin and S100 protein and stained by IMF with the same antibodies and with antibodies to vimentin and actin. Modified (M) MCs of pleomorphic adenomas stained positively by IMP and IMF with all of the preceding antibodies. In many mucoepidermoid carcinomas, adenoid cystic carcinomas, and basal cell adenomas, variable numbers of CMCs and MMCs stained positively by IMP with anti-cytokeratin and anti-S100 protein antibodies. No MCs were detected in adenolymphomas or acinic cell carcinomas. We believe that MCs play a major role in the histogenesis of pleomorphic adenomas and may also be important in many mucoepidermoid carcinomas, adenoid cystic carcinomas, and basal cell adenomas.     

Sialoblastoma: a clinicopathologic and immunohistochemical study of 7 cases

Sialoblastoma is a rare congenital or perinatal salivary tumor that varies in histologic features and biologic potential. Seven cases from the files of the Armed Forces Institute of Pathology are presented. These tumors occurred in 4 males and 3 females with ages ranging from prenatal to 6 months at the time of discovery. Five lesions originated from the parotid gland; 2 lesions were from the submandibular gland. All lesions presented as nodular to multinodular swellings and ranged in size from 2.0 to 7.0 cm. The principal sign or symptom was rapid growth. Two histologic patterns with differing behavior predominated: (1) a favorable pattern had semiencapsulation of cytologically benign basaloid tumor cells with intervening stroma; and (2) an unfavorable histology of anaplastic basaloid tumor cells, minimal stroma, and broad pushing to infiltrative periphery. Four and three tumors had favorable and unfavorable growth patterns, respectively. One unfavorable lesion had vascular invasion, and another demonstrated perineural invasion. All 3 tumors with unfavorable histology recurred. Tumor cells in 3 cases were immunohistochemically reactive for keratin, S-100, smooth muscle actin, and calponin to varying degrees. All 3 tumors were reactive for p63. alpha-Fetoprotein was expressed in 2 unfavorable tumors. Ki67 was expressed at 3% in a favorable tumor and 40% and 80% in the 2 unfavorable lesions. Treatment involved surgical excision. One patient received adjuvant chemotherapy. Two sialoblastomas resulted in recurrences within a year and another developed a recurrence after 4 years. One sialoblastoma developed lung metastasis within 1 month of the original biopsy. Although a clinical correlation is suggested by a favorable/unfavorable histologic grading system the biologic behavior is nonetheless considered unpredictable.     

Cribriform adenocarcinoma of minor salivary gland: A report of two cases with an emphasis on cytology

Cribriform adenocarcinoma of minor salivary gland (CAMSG) is a recently characterized low grade salivary gland malignancy that most commonly presents as a mass in the base of the tongue, frequently with regional lymph node metastasis. Given its relative rarity and overlapping cytomorphology, CAMSG may be confused with polymorphous low grade adenocarcinoma (PLGA) in minor salivary gland sites and papillary thyroid carcinoma (PTC) in cervical metastasis, in both fine‐needle aspiration and excisional specimens. As there are no cytology reports in the literature, we present two new cases of CAMSG and describe the aspiration cytology of the tumor taken from bench top aspirates, compare it with the histomorphology, and discuss the features that may help one avoid misdiagnosis of PTC in the setting of cervical lymph node metastasis. We found that like PTC, aspirates of CAMSG contain polymorphic fragments of epithelial cells arranged in monolayer sheets, papillary fronds and tips, and occasional cribriform configurations, and metachromatic stromal fragments, which may be misinterpreted as colloid. A background of myxoid/mucoid material also reminiscent of colloid was prominent. Differentiation from PLGA is more difficult based strictly on cytology. A review of the most current literature in relation to the molecular and immunohistochemical profiles, therapeutic options, and prognosis is also presented. It is critical for pathologists and clinicians to be aware of this tumor when presented with patients having a cervical lymph node mass in the absence of a primary tumor. Diagn. Cytopathol. 2014;42:1085–1090. © 2014 Wiley Periodicals, Inc.     

Immunological features of minor salivary gland saliva

Concentrations of IgA, IgG, IgM, and IgA subclasses were measured in 138 pairs of parotid gland saliva (PS) and labial gland saliva (LS), using an enzyme-linked immunosorbent assay (ELISA) technique in which levels of Ig were quantitated using affinity-purified anti-heavy chain reagents for capture and development. Both PS and LS were collected simultaneously during sour lemon drop stimulation. As previously observed, IgA was the dominant immunoglobulin in both salivary fluids, the concentrations of which were highly correlated within the subjects studied. The mean proportion of IgA1 to total IgA was slightly higher in LS (0.66), compared with PS (0.60). Little IgM was usually detected in either secretion. In contrast, LS had IgG concentrations (mean, 8.1 µg/ml) which were significantly higher (P<0.001) than those found in parotid saliva (mean, 0.3 µg/ml). Over 30% of the subjects had mean LS IgG levels above 10 µg/ml. The mean percentage of LS IgG to IgA was 20% in the 138 samples tested. Gel filtration of pairs of PS and LS from four individuals revealed IgM, IgA, and IgG to elute in positions commensurate with pentameric IgM, secretory IgA, and monomeric IgG. Little or no monomeric IgA could be detected. These results suggest that, in addition to IgA, the IgG isotype may also be important in antibody-mediated phenomena which occur in oral microenvironments bathed by minor salivary gland secretions.     

涎腺腺样囊性癌的免疫组织化学及免疫电镜观察

采用抗平滑肌的肌动蛋白（ａｃｔｉｎ）、肌球蛋白（ｍｙｏｓｉｎ）、Ｓ—１００蛋白和胶质纤维酸性蛋白（ＧＦＡＰ）对４例涎腺腺样囊性癌进行免疫组化和免疫电镜研究。结果发现，该癌中肿瘤性肌上皮细胞对抗ａｃｔｉｎ、ｍｙｏｓｉｎ和Ｓ—１００蛋白反应阳性，对抗ＧＦＡＰ反应阴性。这些细胞衬里在囊样腔隙周边、小导管外周或散在于上皮团块中；肿瘤性腺上皮细胞对上述抗体反应阴性。讨论了肿瘤性肌上皮细胞的分化趋向和免疫特性。     

Ductal papilloma of the minor salivary gland

A case of ductal papilloma occurring in a minor salivary gland is reported. To date, only a few cases of ductal papillomas have been reported; these comprise mainly inverted ductal papilloma, intraductal papilloma and sialoadenoma papilliferum. However, it may be that these neoplasms form a spectrum of neoplastic changes seen in minor salivary gland duct epithelium.