A case of mammary analogue secretory carcinoma arising in the submandibular region

Ishii, Kazuya; Nakamatsu, Koji; Sato, Koji; Minemura, Chikashi; Kumamaru, Wataru; Yoshikawa, Haruhisa

doi:10.5794/jjoms.61.564

Cited by 3 publications

(1 citation statement)

References 31 publications

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“…The disease concept of secretory carcinoma has been recently established. Currently, the first-line treatment is surgical resection, similar to other salivary gland malignancies [15]. However, the anaplastic lymphoma kinase (ALK) inhibitor crizotinib has been reported to significantly reduce ETV6-NTRK3 gene fusion, and it is potentially effective in the treatment of secretory carcinoma [16].…”

Section: Discussionmentioning

confidence: 99%

A Case of Secretory Carcinoma That Occurred in the Buccal Submucosa

Saito¹,

Kurasawa²,

Moriya³

et al. 2019

IJOHNS

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Secretory carcinoma (SC) is a malignant salivary gland tumor that has been first reported by Skalova et al. in 2010. Histologically, it shows solidity infiltrated with very small cystic cavities and cribriform and papillary features and includes periodic acid-Schiff stain-positive, acid-fast secretions. The cells have oval nuclei, and vacuolated cytoplasm and foamy secretions are seen. Anaplasia is not strong and mitotic figures are rarely seen. These features closely resemble AciCC. Immunohistologically, it is thought to be positive for S-100 protein, vimentin, and mammaglobin and negative for DOG1. The presence of the ETV6-NTRK3 fusion gene is essential in diagnosing secretory carcinoma. In this report, we describe a case of SC in a 52-year-old woman. She was referred to our center because of a mass in left buccal mucosa. A soft and elastic submucosal mass measuring approximately 10 mm × 10 mm in size with a smooth surface was seen in the buccal mucosa in an area corresponding to the left mandibular canine to premolars. The imaging findings revealed that a high-intensity lesion was seen on T2-weighted images. Immunohistochemicalstaing for S-100 protein and vimentin were positive. Furthermore, genetic examination detected the presence of the ETV6-NTRK3 fusion gene. Based on these findings, the definitive diagnosis was secretory carcinoma.

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Section: Discussionmentioning

confidence: 99%

A Case of Secretory Carcinoma That Occurred in the Buccal Submucosa

Saito¹,

Kurasawa²,

Moriya³

et al. 2019

IJOHNS

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Four cases of secretory carcinoma of the salivary gland

et al. 2022

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ラロトレクチニブが著効した唾液腺分泌癌の1例

et al. 2023

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carcinoma is an uncommon neoplasm characterized by the frequent presence of an ETV-NTRK3 gene fusion in addition to distinct histologic features. Secretory carcinoma has been included in the WHO classification of head and neck tumors (4th edition) since 2017. We report herein a case of secretory carcinoma arising in minor salivary glands. The patient was a 68-year-old man who was initially diagnosed in his 30s, by surgery for a tumor of the hard palate, with acinic cell carcinoma arising from the minor salivary glands of the hard palate. He underwent multiple surgeries and radiotherapy for postoperative local recurrence.However, the tumor gradually grew. The local recurrence lesion progressed from the soft palate to the lateral pharyngeal wall and the parapharyngeal space, and multiple lymph node metastases were observed. Multigene cancer panel testing was performed. The ETV6-NTRK3 fusion gene was detected, leading to a diagnosis of secretory carcinoma. The patient was treated with Larotrectinib, a TRK inhibitor, and has maintained a complete response at 1 year and 6 months after the start of treatment. We believe this case exemplifies the use of targeted TRK inhibition as a well-tolerated treatment achieving a rapid and durable response in a secretory carcinoma, for which there is no standard systemic therapy.

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A case of mammary analogue secretory carcinoma arising in the submandibular region

Cited by 3 publications

References 31 publications

A Case of Secretory Carcinoma That Occurred in the Buccal Submucosa

A Case of Secretory Carcinoma That Occurred in the Buccal Submucosa

Four cases of secretory carcinoma of the salivary gland

ラロトレクチニブが著効した唾液腺分泌癌の1例

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