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Case Reports
. 2020 Aug 1;13(8):2211-2217.
eCollection 2020.

Secretory carcinoma around Stensen's duct misdiagnosed as salivary duct cyst

Eri Shibata  1 , Kei-Ichi Morita  1   2 , Kou Kayamori  3 , Mayuko Maruiwa  1 , Yasuyuki Michi  4 , Yukiko Sato  5 , Kengo Takeuchi  5   6 , Tohru Ikeda  3 , Hiroyuki Harada  4 , Tetsuya Yoda  1
Affiliations
Case Reports

Secretory carcinoma around Stensen's duct misdiagnosed as salivary duct cyst

Eri Shibata et al. Int J Clin Exp Pathol. .

Abstract

Secretory carcinoma (SC) of the salivary gland was identified in 2010, and it is characterized by a specific ETV6 gene arrangement. The most common primary site for SC is the parotid gland; however, SC around the Stensen's duct is rare. Here we describe a rare case of a SC around the Stensen's duct that was initially misdiagnosed as a salivary duct cyst. A 59-year-old woman presented with a mass in the region of the left parotid papilla. Magnetic resonance imaging (MRI) revealed a well-circumscribed lesion and enhancement with a rim and an inner wall-like part that appeared in the late phase. Based on the initial clinical and imaging findings, a salivary duct cyst of the parotid gland was diagnosed. However, the lesion was histopathologically diagnosed as a SC based on immunohistochemical findings. The tumor cells showed diffuse positive staining for AE1/AE3, vimentin, and mammaglobin and focal positive staining for S-100 protein, SOX-10, and DOG-1. Fluorescence in-situ hybridization revealed ETV6 gene rearrangement in the tumor. In cases of cystic lesions around the Stensen's duct, clinicians should bear in mind that the possibility that they could be minor salivary gland cancers, such as SC.

Keywords: ETV6 gene arrangement; Secretory carcinoma; Stensen’s duct; mammaglobin; salivary duct cyst.

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Conflict of interest statement

None.

Figures

Figure 1
Figure 1
Image and surgical findings. (A) The T1-weighted imaging revealed a lesion with a slightly higher intensity (arrow) than that of the muscle. (B) The T2-weighted, fat-suppressed imaging revealed a high intensity lesion (arrow). (C and D) Dynamic Gd-enhanced MRI revealed enhancement with a rim and an inner wall-like part (arrow) during the late phase (D) but not the early phase (C), and most of the lesion’s inner part was not enhanced. (E) Surgical findings. The mass with a part of buccal mucosa including the parotid papilla was removed and ligated at the normal parotid duct (arrow). (F) Gross view of the resected specimen.
Figure 2
Figure 2
Histologic findings. (A) H&E staining. An anatomical arrangement of tumor tissues. The cystic structures of the tumor tissues, which were partially lined with tumor nests, and the cystic wall involved a large excretory duct (arrowheads). (B-G) Immunohistochemical studies showed that tumor tissues displayed diffuse positive staining for AE1/AE3 (B), vimentin (C) and mammaglobin (D). Staining for S-100 (E), SOX-10 (F), and DOG-1 (G) was focally positive. (H) ETV6 split. For Fluorescence in situ hybridization (FISH), unstained sections (4 μm thick) were subjected to hybridization with the bacterial artificial chromosome (BAC) clone-derived probes for ETV6, which were differentially labeled with FITC (green) (RP11-639O1, RP11-1077I5) and Texas Red (red) (RP11-297N18). The hybridized slides were then stained with DAPI and examined using a BZ-Z710 fluorescence microscope (Keyence, Osaka, Japan).
Figure 3
Figure 3
A. Reverse transcription-polymerase chain reaction analysis of ETV6-NTRK3. The tumor showed the ETV6-NTRK3 fusion transcript of 110 bp (Lane 1). B. Sequencing results. Arrowhead, the fusion point of the ETV6-NTRK3.
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