Juvenile sclerosing polycystic adenosis cytologically mimicking Warthin tumor

Sclerosing polycystic adenosis (SPA) is a rare salivary gland disease. Histologically it resembles a low‐grade ductal carcinoma in situ or sclerosing adenosis of the breast, characterized by lobular proliferation of ducts with apocrine cellular features surrounded by fibrosclerotic stroma. Although SPA is typically benign, recurrence is not uncommon, and cases with a malignant component have been documented. Thus, complete excision is desirable but preoperative diagnosis is challenging. A 12‐year‐old boy presented with a painless mass in the right neck. We identified a well‐demarcated mass in the right parotid region measuring approximately 2 cm using cervical echography and magnetic resonance (MR) imaging. Fine‐needle aspiration (FNA) revealed two cell types. There were loosely cohesive clusters of polymorphic epithelioid cells with irregular nuclei and abundant vacuolated cytoplasm containing zymogen granules. Some of these cells were binuclear. The other cell types represented normal ductal cells. The original cytological diagnosis was Warthin tumor. Right parotidectomy was performed. Histologically, we observed proliferation of ducts with granular, vacuolated, zymogen granules, and apocrine‐like features in the cytoplasm with hyalinizing sclerotic stroma and some binuclear cells. Four years after parotidectomy, there has been no recurrence or malignant transformation.Cytological diagnosis of SPA is challenging on FNA specimens since SPA is a very rare entity of the salivary gland that can mimic other salivary gland neoplasms. A mixture of apocrine‐like cells and sebaceous‐like cells, nuclear pleomorphism, and zymogen granules can help to diagnose this rare lesion during the initial cytological diagnosis.     

Pure intraductal carcinoma of the parotid gland: Cytologic findings on FNA sample. Report of one case

One case of intraductal carcinoma of the parotid gland in a 67‐year‐old male patient is here introduced. The patient, who had a one‐year history of a parotid mass, had undergone ultrasound and MRI examination that disclosed a 13x4x3 mm well delimited nodular mass of the accessory lobe of his left parotid gland. Ultrasound‐guided Fine Needle Aspiration (FNA) had been performed by the clinician. The obtained smears showed widespread cellular necrosis in which cellular clusters with moderate and focally severe atypias displayed papillary and cribriform architecture and were admixed with sheets of epithelial cells with less striking nuclear atypias, squamous, or apocrine metaplasia. Histopathological examination disclosed a pure intraductal carcinoma of the parotid gland with classical morphology, which was radically excised. The differential cytological diagnosis of pure intraductal carcinoma of salivary glands may be difficult and comprises mucoepidermoid carcinoma as well as “in situ” carcinomas developping in the context of sclerosing polycystic adenosis, mammary analogue secretory carcinoma (MASC) of the salivary glands and cystic variants of salivary adenocarcinoma NOS (formerly called cystadenocarcinomas).     

Sclerosing polycystic adenosis: A rare tumor misdiagnosed as retention cyst on fine needle aspiration cytology

Sclerosing polycystic adenosis (SPA) is a rare benign neoplasm of the salivary gland which resembles the fibrocystic disease of the breast clinically as well as morphologically. This entity has varied morphological presentation on fine needle aspiration. Only a few case reports and occasional case series are found in the literature describing its cytology. Here we are presenting a case of SPA in the parotid gland in a 13‐year‐old male patient who presented with a slow growing infra‐auricular mass since one year without any other symptoms. Radiological investigation suggested a possibility of matted level II cervical lymph nodes. Fine needle aspiration of the same showed only crystalloids in a dirty background. Hence, a possibility of retention cyst was given and excision was advised. On histopathological examination, the mass was diagnosed as Sclerosing polycystic adenosis of the parotid gland. Since SPA is a rare entity with diverse morphological features, it is more likely to be misdiagnosed on cytology. Recognition of this benign entity is important since the differential diagnosis includes other more common benign and malignant salivary gland neoplasm particularly mucoepidermoid carcinoma and other tumors with cystic and oncocytic features. Repeat aspiration from multiple sites should be considered in cystic lesions of the salivary gland so that its varied cytological components can be yielded to help with the diagnosis. Diagn. Cytopathol. 2017;45:640–644. © 2017 Wiley Periodicals, Inc.     

Cystic lesions of the salivary glands: cytologic features in fine-needle aspiration biopsies

A variety of neoplastic and nonneoplastic lesions of the salivary glands have a predominantly cystic architecture. Fine-needle aspirates of these lesions yield watery or mucoid material, frequently of low cellularity. Such aspirates may be obtained from mucus retention cysts, lymphoepithelial cysts, cystadenomas, Warthin's tumors, cystic pleomorphic adenomas, low-grade mucoepidermoid carcinomas, cystadenocarcinomas, and examples of polycystic disease of the parotid gland. The cellular component within the fluid obtained from these lesions may be exceedingly scant or absent, making cytologic diagnosis difficult and, at times, impossible. We studied a series of 56 cystic lesions of the salivary glands, including 38 Warthin's tumors, 6 benign cysts, 2 lymphoepithelial cysts, 5 low-grade mucoepidermoid carcinomas, 1 cystic pleomorphic adenoma, 2 cystadenomas, and 2 cystadenocarcinomas. Careful attention to the cellular elements present often allowed definitive cytologic diagnosis, with an overall accuracy rate of 84%. The presence of atypical squamous metaplasia in oncocytic lesions was a significant cause of false-positive diagnoses of carcinoma (4 cases, 7%). Aspirates of low-grade mucoepidermoid carcinoma may contain no epithelial cells and result in false-negative diagnoses (1 case, 2%).     

Selected problems in fine needle aspiration of head and neck masses.

A wide variety of masses in the head and neck, including those in the major salivary glands, can be approached by fine needle aspiration. In many instances, a correct definitive diagnosis con be rendered after examination of smears or cell block material. However, several significant but uncommon areas can lead to diagnostic difficulties, with the potential for clinically important diagnostic errors. Many of these occur in salivary gland lesions. The most frequent problems involve variations in the expected cytology of pleomorphic adenoma. Then, there are several benign-malignant "look-alike" pairs of lesions. The first of these is related to small-cell epithelial neoplasms of low nuclear grade; the most frequent problem is between basal cell adenomas and adenoid cystic carcinoma, particularly the solid (anaplastic) type. The next area contrasts mucoepidermoid carcinoma with its cytologic mimic, benign salivary gland duct obstruction. The final difficulty in salivary gland aspiration contrasts large-cell epithelial lesions of low nuclear grade: oncocytic proliferations and acinic cell carcinoma. The clinical implications of cytologically benign squamous cell-containing cyst aspirates from the lateral neck will be discussed. Finally, a brief consideration of methodological optimization for thyroid aspirations will be offered.     

Fine-needle aspiration biopsy of cystic pleomorphic adenoma with adnexa-like differentiation mimicking mucoepidermoid carcinoma: a case report

Although pleomorphic adenomas (PAs) usually can be diagnosed very accurately with fine-needle aspiration biopsy (FNAB), even this common salivary gland neoplasm can be diagnostically challenging and cause pitfalls in cytodiagnosis. In particular, the presence of either cystic changes or squamous, mucinous, or sebaceous metaplasia can lead to a false positive diagnosis of mucoepidermoid carcinoma (MEC). Here, we present a case of a 70-yr-old man with an asymptomatic left deep lobe parotid mass for which CT-guided FNAB was performed. The FNAB cytology revealed cohesive clusters of squamous epithelial cells, sebaceous cells, oncocytes, macrophages, and rare myoepithelial cells. Characteristic metachromatic fibrillar chondromyxoid stroma, which usually is seen in PAs, was not seen in the aspirate. Although cytodiagnosis of PAs was suggested based on the presence of other cellular components, resection was recommended. The subsequent parotidectomy specimen revealed an encapsulated cystic PA with mixed appendageal differentiation including areas of squamous, mucinous, sebaceous, and oncocytic metaplasia. Chondromyxoid stroma was only focally present. Presence of squamous, mucinous, and/or sebaceous metaplasia, especially in the absence of chondromyxoid stroma, presents the potential for misinterpretation of the FNAB as indicative of malignancy in general and MEC in particular.     

Fluorescence in situ hybridization for detection of MAML2 rearrangements in oncocytic mucoepidermoid carcinomas: utility as a diagnostic test

Oncocytic mucoepidermoid carcinoma poses diagnostic challenge because of its histologic overlap with other oncocytic salivary gland lesions, including Warthin tumor. Although the prognostic value of the t(11;19) MECT1-MAML2 fusion gene has been established in mucoepidermoid carcinoma, its diagnostic use in discriminating oncocytic mucoepidermoid carcinoma from histologic mimics is unexplored. We evaluated the translocation status in 14 cases of oncocytic mucoepidermoid carcinoma using a MAML2-11q21 break-apart probe spanning the entire chromosome region of the MAML2 gene and correlated these findings with clinicopathologic parameters including age, sex, stage, predominant growth pattern, grade, and p63 immunostaining pattern. All oncocytic mucoepidermoid carcinomas were parotid tumors with a mean patient age of 54.6 years (range, 9-85) and a female to male ratio of 5:2. Grade distribution was as follows: low grade, 9; intermediate grade, 2; and high grade, 3. The histologic patterns observed were as follows: solid, 4; cystic, 8 (of these, 5 had Warthin-like lymphoid stroma); and mixed, 2. Solid oncocytic mucoepidermoid carcinomas showed a diffuse p63 staining pattern, whereas cystic oncocytic mucoepidermoid carcinomas showed staining of the outer layer of intermediate cells ranging from a bilayer to areas of complex multilayering and plaque-like proliferation. Ten (71%) of the 14 cases showed a MAML2 rearrangement by fluorescence in situ hybridization. No correlation was seen between rearrangement status and histologic grade, growth pattern, or p63 staining pattern. However, we demonstrate that the presence of MAML2 rearrangement can be used as supportive evidence to distinguish oncocytic mucoepidermoid carcinoma from other oncocytic lesions.     

Synchronous ipsilateral Warthin tumor encased by a separate mucoepidermoid carcinoma of the parotid gland: A case report and review of the literature

Fine‐needle aspiration (FNA) is a cost effective and low morbidity procedure in the initial assessment of salivary gland tumors. However, cytological assessment of ipsilateral synchronous tumors (which make up less than 0.3% of all salivary gland neoplasms) may pose diagnostic challenges. Therefore, a wholesome approach, including FNA with clinical and radiological correlation, is of utmost importance. Here, we report a unique case of Warthin tumor encased by a separate high‐grade mucoepidermoid carcinoma that was first diagnosed on FNA. Another striking feature seen was the presence of chronic sialadenitis in the surrounding nonneoplastic salivary gland. The presence of two different neoplasms in the background of chronic sialadenitis raises the question of a possible causal relationship. Traditionally, there has been diagnostic difficulty when dealing with synchronous tumors of the salivary gland and the background of chronic sialadenitis may further complicate the diagnosis. FNA is very helpful and can give important cues to the diagnosis. Diagn. Cytopathol. 2010. © 2009 Wiley‐Liss, Inc.     

Epithelial tumors of the lacrimal glands: a clinicopathologic study.

We report the clinicopathologic features of epithelial tumors of the lacrimal gland apparatus, which are rare and therefore represent a major challenge for diagnosis and treatment. Histologic material from 22 lesions was studied by light microscopy, histochemistry, and immunohistochemistry. A comparison with major and minor salivary gland tumors was performed to analyze the relative distribution of these tumors and to establish whether salivary glands and lacrimal gland tumors are similar or different in their pathologic appearance and clinical behavior. There were three benign pleomorphic adenomas and 19 malignant tumors. The gender distribution was equal. The ages of the patients ranged from 10 to 73 years (mean age, 46 years). Among the malignant tumors, adenoid cystic carcinoma was the most common (nine cases), followed by mucoepidermoid carcinoma (three cases). There were two cases each of malignant mixed tumor and adenocarcinoma. All mucoepidermoid carcinomas and the adenocarcinomas were histologically high grade. There also was one case each of salivary duct carcinoma, spindle cell carcinoma, and oncocytic adenocarcinoma. Of 14 patients in whom clinical follow-up was available, seven had distant metastases and four died of their disease. The only case occurring in a child was an adenoid cystic carcinoma that recurred locally after 14 years. The clinical and pathologic features of lacrimal gland tumors resemble those lesions that arise in the intraoral minor salivary glands. The greater relative proportion of malignant cases in this series probably reflects a selection bias.     

Tigroid background in cytology of hyalinizing clear cell carcinoma of the salivary gland

Hyalinizing clear cell carcinoma (HCCC) is a rare and low grade salivary gland carcinoma with a unique recurrent EWSR1 gene translocation. HCCC most commonly arises from intraoral minor salivary glands and its cytological features have not been well evaluated. We report a 28‐year‐old man with palatal HCCC and emphasize the “tigroid background” and metachromatic matrix observed in scrape cytology. This “tigroid background,” usually associated with glycogen‐rich clear cell tumors, is an important cytological feature of HCCC. Awareness of this feature can do great help in pathological research and diagnosis. Diagn. Cytopathol. 2016;44:338–341. © 2015 Wiley Periodicals, Inc.     

Nestin is a wide‐spectrum abluminal cell marker of salivary gland tumors

Nestin is an intermediate filament that was first identified in neural progenitor cells. It is expressed in various cell types in the nervous system as well as in other systems. In the present study, we investigated nestin expression in non‐neoplastic salivary gland tissue and in salivary gland tumors. In non‐neoplastic salivary glands, nestin expression was observed in only a few abluminal cells. In contrast, diffuse nestin staining was observed in the abluminal cells of pleomorphic adenoma (11 of 11 cases), basal cell adenoma (7 of 7 cases), and epithelial‐myoepithelial carcinoma (2 of 2 cases). The stromal cells in basal cell adenoma also expressed nestin. In adenoid cystic carcinoma (6 of 7 cases) and polymorphous low‐grade adenocarcinoma (3 of 3 cases), nestin positive cells were observed focally. Nestin was not detected in Warthin tumor (6 cases), classical acinic cell carcinoma (2 cases), mucoepidermoid carcinoma (5 cases), or salivary duct carcinoma (4 cases). Because the nestin expression pattern in each histological salivary gland tumor type is unique, nestin could be a very useful abluminal cell marker for the diagnosis of salivary gland tumors.     

Hautadnex- vs. Speicheldrüsentumoren

Despite major discrepancies in basic microscopic anatomy, remarkable similarities are manifest within the wide spectrum of cutaneous adnexal and salivary gland tumors. In this study salivary gland and adnexal tumors were identified and investigated with respect to similarities in histology, terminology and pathogenesis. Histological similarities of certain types of salivary gland tumors relate to eccrine, apocrine and rarely sebaceous (but not trichofollicular) types of adnexal tumors. The most striking similarity was found with salivary gland pleomorphic adenoma and cutaneous mixed tumor. Multistep carcinogenesis starting with intraductal carcinoma, identified in carcinoma ex pleomorphic adenoma is identical to that found in cutaneous carcinoma ex spiradenoma. Further histological and terminological similarities are shown for mucinous and mucoepidermoid carcinoma, for lymphadenoma and lymphoepithelial carcinoma, for sebaceous adenoma and carcinoma, for adenoid-cystic carcinoma, as well as for salivary gland basal cell adenoma versus cutaneous spiradenoma. Manifest diagnostic problems related to histologically similar salivary gland and adnexal tumors are rare and are topographically limited to the parotid and oral areas.     

Oncocytic variant of mucoepidermoid carcinoma of submandibular gland: an unusual clinical and morphological entity

In this case report we describe a rare tumor--Oncocytic variant of Mucoepidermoid carcinoma of the submandibular salivary gland with a review of the literature. Oncocytic metaplasia in salivary glands is a benign change that is associated with increasing age and also seen in a few salivary gland neoplasms', which include oncocytoma, Warthin's tumor, and the rare, oncocytic carcinoma. Oncocytic differentiation in mucoepidermoid carcinoma (MEC) is uncommon. Only twelve well-documented cases of oncocytic MEC have been reported previously all of which occurred in the parotid gland. To the best of our knowledge this is the first case of oncocytic mucoepidermoid carcinoma involving the submandibular salivary gland. The recognition of this entity is important, since most of the other primary oncocytic lesions of the salivary gland are benign.     

Oncocytic lipoadenoma of the parotid gland: Cytological findings and differential diagnosis on fine‐needle aspiration

Oncocytic lipoadenoma is a rare benign neoplastic lesion of salivary gland. To the best of our knowledge, the detailed cytomorphological findings were described only in two cases. We are reporting a 56‐year‐old woman who presented with 1‐year history of right parotid gland mass. Cytologic examination revealed tight clusters of bland looking oncocytic ductal cells with few aggregates of mature adipose tissue in a lipoid background and a benign oncocytic tumor of parotid gland was rendered. Histologically, a tumor with islands of oncocytic epithelial cells admixed with abundant mature adipose tissue was identified. Oncocytic lipoadenoma despite its rarity should be considered in the differential diagnosis of salivary gland fine‐needle aspirations containing oncocytes especially those which are accompanied by mature adipose tissue and lipoid background. Diagn. Cytopathol. 2015;43:72–74. © 2014 Wiley Periodicals, Inc.     

Salivary gland-type neoplasm of the lung

Salivary gland-type tumours of the lung, which include adenoid cystic carcinoma and mucoepidermoid carcinoma, are rare entities. Histologically, they show morphologies that are similar to those of salivary gland origin. For classification purposes, the terminology applied to tumours of salivary gland origin is also applied to their pulmonary counterparts. Of these types of tumours, adenoid cystic carcinoma is the most common followed by mucoepidermoid carcinoma. Epithelial-myoepithelial carcinoma and pleomorphic adenoma are uncommon. The possibility of metastasis from the salivary glands is important to exclude through careful examination of patients' medical history because low-grade salivary cancers can metastasize after long latencies. A gene rearrangement analysis is helpful in making a definitive diagnosis, particularly in cases with an unusual morphology. Molecular-based classification may be useful to understand the nature of morphologically varied tumours. The immunohistochemical profile of the tumours is also a valuable tool for an accurate diagnosis in difficult cases.     

Selected topics in salivary gland tumour pathology

The complexity of salivary tumours is largely due to the participation of neoplastic myoepithelial cells which exhibit remarkable phenotypic and secretory abilities. The proportions and morphologic appearances of ductal and myoepithelial cells, type of tumour matrix and relation of tumour cells with extracellular matrix is what defines entities such as mixed tumours, myoepitheliomas, adenoid cystic carcinomas, epithelial/myoepithelial carcinoma, and basal cell adenocarcinoma. Other lesions such as polymorphous low-grade adenocarcinoma, acinic cell carcinoma, and hyalinizing clear cell carcinoma show little or no myoepithelial differentiation. Understanding this so-called morphogenetic concept is quite important in the diagnosis of these tumours. Pathologic grading of salivary gland tumours is generally implied in the diagnosis; however, tumour grade provides significant prognostic information in mucoepidermoid carcinoma, carcinoma ex-pleomorphic adenoma, and adenoid cystic carcinoma.     

Cribriform adenocarcinoma of the minor salivary gland arising in the tonsil with metastasis to a cervical lymph node: A case report with description of fine needle aspiration cytology

Cribriform adenocarcinoma of minor salivary gland (CAMSG) is a rare tumor of the head and neck. We report a case of a 70‐year‐old female who presented with a 4–5‐month history of a left neck mass. CT scan of the neck showed a left neck mass just inferior to the angle of the mandible and left tonsillar prominence. Fine‐needle aspiration (FNA) of the neck mass showed epithelial groups with focal cribriform architecture. The cells had round to oval nuclei and fine chromatin. The background contained scattered lymphocytes. A preliminary diagnosis of low grade adenocarcinoma with cribriform features metastatic to a lymph node was made. Subsequent biopsy of the tonsil mass showed a tumor with a combination of tubular, solid, and papillary architecture containing round to ovoid nuclei with very fine chromatin, consistent with cribriform adenocarcinoma of the minor salivary gland. Cribriform adenocarcinoma of minor salivary gland has a documented tendency to metastasize to cervical lymph nodes. Since this neoplasm can cytologically and histologically resemble other neoplasms of the head and neck, including polymorphous low‐grade adenocarcinoma (PLGA), papillary thyroid carcinoma (PTC), and occasionally adenoid cystic carcinoma, being aware of and familiar with the cytologic features of CAMSG on FNA smears is important for patient management. Diagn. Cytopathol. 2017;45:468–471. © 2017 Wiley Periodicals, Inc.     

Aspiration biopsy of mammary analogue secretory carcinoma of accessory parotid gland: Another diagnostic dilemma in matrix‐containing tumors of the salivary glands

Mammary analogue secretory carcinoma (MASC) is a newly described rare salivary gland tumor, which shares morphologic features with acinic cell carcinoma, low‐grade cystadenocarcinoma, and secretory carcinoma of the breast. This is the first reported case of MASC of an accessory parotid gland detected by aspiration biopsy with radiologic and histologic correlation in a 34‐year‐old patient. Sonographically‐guided aspiration biopsy showed cytologic features mimicking those of low‐grade mucoepidermoid carcinoma, including sheets of bland epithelial cells, dissociated histiocytoid cells with intracytoplasmic mucinous material, and spindle cells lying in a web‐like matrix. Histologic sections showed a circumscribed tumor with microcystic spaces lined by bland uniform epithelial cells and containing secretory material. The tumor cells expressed mammaglobin and BRST‐2. The cytologic features, differential diagnosis, and pitfalls are discussed. The pathologic stage was pT1N0. The patient showed no evidence of disease at 1 year follow‐up. Diagn. Cytopathol. 2014;42:49–53. © 2012 Wiley Periodicals, Inc.