A Rare Primary Neuroendocrine Tumor (Typical Carcinoid) of the Sublingual Gland

Yamagata, Kenji; Ohki, Kohji; Uchida, Fumihiko; Kanno, Naomi; Hasegawa, Shogo; Yanagawa, Toru; Bukawa, Hiroki

doi:10.1155/2016/7462690

Cited by 4 publications

(7 citation statements)

References 11 publications

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“…The overwhelming majority are high-grade with the small cell type out numbering large cell carcinomas by a factor of about 5:1 [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16][17][18]. Well and moderately-differentiated NECs are exceedingly rare with < 10 cases described in the literature [19][20][21][22]. This includes an interesting example of a hereditary well-differentiated neuroendocrine neoplasm of the salivary glands with unique morphology and association with sensorineural hearing loss and enamel hypoplasia that was described in a family from the Isle of Man [21].…”

Section: Introductionmentioning

confidence: 99%

Proceedings of the NASHNP Companion Meeting, March 18th, 2018, Vancouver, BC, Canada: Salivary Neuroendocrine Carcinoma—An Overview of a Rare Disease with an Emphasis on Determining Tumor Origin

Chernock

Duncavage

2018

Head and Neck Pathol

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Salivary neuroendocrine carcinomas are rare and the overwhelming majority is high-grade. The parotid gland is the most commonly involved site followed by the submandibular gland. Most arise de novo but rare examples occurring as a high-grade transformation of another type of salivary gland neoplasm exist. There is significant morphologic and immunophenotypic overlap with neuroendocrine carcinomas of other sites, especially the skin. Like cutaneous neuroendocrine (or Merkel cell) carcinomas, approximately three-fourths are cytokeratin 20 positive. Cytokeratin 20 positive salivary neuroendocrine carcinomas are often referred to as being of the 'Merkel cell type' since most other non-cutaneous neuroendocrine carcinomas are cytokeratin 20 negative. Salivary neuroendocrine carcinomas may be challenging to separate from Merkel cell carcinomas of the head and neck on pathologic grounds because the latter often metastasize to the parotid gland. Clinical history is often relied upon to separate primary salivary tumors from cutaneous metastases but may not be helpful in all cases. Here we review the clinical, pathologic and molecular features of salivary neuroendocrine carcinomas focusing on high-grade major salivary gland tumors. The difficulty in separating salivary tumors from metastatic Merkel cell carcinoma will be highlighted.

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Section: Introductionmentioning

confidence: 99%

Proceedings of the NASHNP Companion Meeting, March 18th, 2018, Vancouver, BC, Canada: Salivary Neuroendocrine Carcinoma—An Overview of a Rare Disease with an Emphasis on Determining Tumor Origin

Chernock

Duncavage

2018

Head and Neck Pathol

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“…One published case of a sublingual gland "typical carcinoid tumor" in a 62-year-old woman looks remarkably similar to what we are calling palisading adenocarcinoma. 11 This reported case had strong CD56 staining but was synaptophysin negative, and published images demonstrate very little chromogranin (which could represent mast cells). This case was from Japan.…”

Section: Discussionmentioning

confidence: 70%

“…This reported case had strong CD56 staining but was synaptophysin negative, and published images demonstrate very little chromogranin (which could represent mast cells). This case was from Japan 11 . Rarely, salivary gland carcinomas have geographical associations.…”

Section: Discussionmentioning

confidence: 84%

“…Indeed, 3 of the cases presented here were originally diagnosed as paraganglioma, and as previously noted, a probable case was previously reported as a sublingual low-grade neuroendocrine tumor. 11 The prominent palisading, pseudorosette formation, stromal hyalinization, and frequent degenerative, bizarre nuclear atypia are all reminiscent of a tumor with genuine neuroendocrine differentiation, and the strong CD56 positivity seen in all cases could be used as immunohistochemical support for that hypothesis. In contrast, true low-grade neuroendocrine neoplasms are exceedingly rare in the major salivary glands.…”

Section: Discussionmentioning

confidence: 97%

“…This case was from Japan. 11 Rarely, salivary gland carcinomas have geographical associations. For example, lymphoepithelial carcinoma has a much higher incidence in certain ethnic groups (Arctic Inuits, patients from southern China) but this is almost certainly due to endemic EBV infection.…”

Section: Discussionmentioning

confidence: 99%

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Palisading Adenocarcinoma

Bishop

Weinreb²,

Vliet

et al. 2023

American Journal of Surgical Pathology

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Adenocarcinoma, not otherwise specified (NOS) is a heterogenous group of salivary gland tumors that likely contains distinct tumors that have not yet been characterized. Indeed, in recent years, cases previously diagnosed as adenocarcinoma, NOS have been recategorized into novel tumor designations such as secretory carcinoma, microsecretory adenocarcinoma, and sclerosing microcystic adenocarcinoma. We sought to describe a distinctive, hitherto-undescribed salivary gland tumor encountered in the authors’ practices. Cases were pulled from the surgical pathology archives of the authors’ institutions. Histologic, immunohistochemical, and clinical findings were tabulated, and targeted next-generation sequencing was performed on all cases. Nine cases were identified, arising in 8 women and 1 man ranging from 45 to 74 years (mean, 56.7 y). Seven tumors (78%) arose in the sublingual gland, while 2 (22%) arose in the submandibular gland. The cases shared a distinctive morphologic appearance. They were biphasic, with ducts scattered among a predominant polygonal cell with round nuclei, prominent nucleoli, and pale eosinophilic cytoplasm. These cells were arranged as trabeculae and palisaded as pseudorosettes around hyalinized stroma and vessels, resembling a neuroendocrine tumor. Four of the cases were well-circumscribed, while the remaining 5 showed infiltrative growth including perineural invasion in 2 (22%) and lymphovascular invasion in 1 (11%). Mitotic rates were low (mean, 2.2/10 HPFs); necrosis was absent. By immunohistochemistry, the predominant cell type was strongly positive for CD56 (9 of 9) and variably positive for pan-cytokeratin (AE1/AE3) (7 of 9) with patchy S100 (4 of 9), but negative for synaptophysin (0 of 9) and chromogranin (0 of 9), while the ducts were strongly positive for pan-cytokeratin (AE1/AE3) (9 of 9) and CK5/6 (7 of 7). Next-generation sequencing did not reveal any fusions or obvious driver mutations. All cases were resected surgically, with external beam radiation also done in 1 case. Follow-up was available in 8 cases; there were no metastases or recurrences after 4 to 160 months (mean, 53.1 mo). A dual population of scattered ducts with a predominance of CD56-positive neuroendocrine-like cells characterizes a unique salivary gland tumor which is often encountered in the sublingual glands of women, for which we propose the term “palisading adenocarcinoma.” Although the tumor was biphasic and had a neuroendocrine-like appearance, it lacked convincing immunohistochemical evidence of myoepithelial or neuroendocrine differentiation. Although a subset showed unequivocally invasive growth, this tumor appears to behave in an indolent manner. Moving forward, recognition of palisading adenocarcinoma and its separation from other salivary adenocarcinomas, NOS will facilitate a better understanding of the characteristics of this previously unrecognized tumor.

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Fine‐needle aspiration cytology of palisading adenocarcinoma: The first cytology report of a newly described salivary gland neoplasm

Rivera,

Kuo,

Yeh

et al. 2024

Diagnostic Cytopathology

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Here, we report the first cytology findings of the newly characterized entity, palisading adenocarcinoma of the salivary gland, diagnosed in the sublingual gland of a 61‐year‐old female. The liquid‐based cytology showed a moderately cellular aspirate containing three‐dimensional clusters and trabeculae of tumor cells of various sizes. The cells had dark ovoid nuclei, finely granular chromatin, inconspicuous to punctate nucleoli, and ample cyanophilic cytoplasm with indistinct cell borders. In conventional smears, the cells displayed frequent crush artifacts and anisonucleosis resembling endocrine‐type atypia. The background was clean, devoid of secretions, and contained singly dispersed tumor cells with stripped nuclei. Interestingly, concentrically laminated globules of extracellular matrix surrounded by the tumor cells were identified. Mitotic figures and tumor necrotic debris were absent. The cytologic findings correlated with the histologic findings of the excision specimen. The cytologic differential diagnosis and tumor grading of palisading adenocarcinoma were briefly discussed.

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A Rare Primary Neuroendocrine Tumor (Typical Carcinoid) of the Sublingual Gland

Cited by 4 publications

References 11 publications

Proceedings of the NASHNP Companion Meeting, March 18th, 2018, Vancouver, BC, Canada: Salivary Neuroendocrine Carcinoma—An Overview of a Rare Disease with an Emphasis on Determining Tumor Origin

Proceedings of the NASHNP Companion Meeting, March 18th, 2018, Vancouver, BC, Canada: Salivary Neuroendocrine Carcinoma—An Overview of a Rare Disease with an Emphasis on Determining Tumor Origin

Palisading Adenocarcinoma

Fine‐needle aspiration cytology of palisading adenocarcinoma: The first cytology report of a newly described salivary gland neoplasm

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