Secretory Carcinoma of Minor Salivary Gland in Buccal Mucosa: A Case Report and Review of the Literature

Paudel, Durga; Nishimura, Michiko; Adhikari, Bhoj Raj; Hiraki, Daichi; Onishi, Aya; Morikawa, Tetsuro; Neopane, Puja; Giri, Sarita; Yoshida, Koki; Sato, Jun; Ono, Masahiro; Kamino, Yoshitaka; Nagayasu, Hiroki

doi:10.1155/2019/2074504

Cited by 9 publications

(9 citation statements)

References 34 publications

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“…As a member of the new classification of salivary gland malignant tumors, it was officially listed in the WHO Head and Neck Tumor Classification (4 th edition) and referred to as secretory carcinoma. SCSGs of other organs have also been reported, such as the minor salivary glands around the sinuses (7), lips (8), and buccal mucosa (9), as well as the thyroid gland (10) and lungs. It often presents as a cystic-solid mass and the boundary is not clear.…”

Section: Historymentioning

confidence: 99%

Two cases report of secretory carcinoma of the salivary gland in the lung: one primary and one metastatic after many years

Wang¹,

Xie²,

Li³

et al. 2022

Gland Surg

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Background: Secretory carcinoma of the salivary gland (SCSG) is a recently discovered salivary gland tumor that occurs mostly in the major salivary glands and occasionally in the skin, cervix, trachea, etc.Secretory carcinoma of the lung is extremely rare. To our knowledge, this is the third report of SCSG arising as a primary pulmonary tumor. The two SCSG cases reported in this paper are unique in that one was primary and the other was metastasized to the lung.Case Description: Case 1 is a primary endobronchial tumor in a 66-year-old man. He went to the doctor complaining of fever, cough and yellow phlegm, and his body weight was significantly reduced by 3 kg. The bronchoscope showed the growth of new organisms in the right upper lobe of the lung.Immunohistochemistry of his biopsy specimen was positive for AE1/AE3, Keratin7 (CK7), S-100, mammaglobin, and pan-TRK, but negative for thyroid transcription factor-1 (TTF-1), napsin-A, synaptophysin (SYN), chromogranin A (CGA), and discovered on GIST-1 (Dog-1), and the MKI-67 (Ki-67) proliferation index was 2%. This case lacked the typical ETV-6 gene rearrangement. After one cycle of chemotherapy, the tumor was significantly reduced, and surgical excision was planned. Case 2 was a metastatic secretory carcinoma with a history of parotid pleomorphic adenoma resection 30 years ago and malignant pleomorphic adenoma resection 16 years ago before the study, respectively. He presented with a complaint of a parotid gland mass. Chest CT examination revealed a mass in the upper lobe of the left lung. The biopsy tissue of him exhibited a typical histological appearance under the microscope.Immunohistochemistry was positive for AE1/AE3, CK7, S-100, and mammaglobin; partially positive for estrogen receptor (ER) and pan-TRK; and negative for TTF-1, Napsin-A, SYN, CGA, P63, P40, and Dog-1. The Ki-67 proliferation index was approximately 3%. Fluorescence in situ hybridization (FISH) revealed ETV-6 gene rearrangement. After the diagnosis of SCSG, the patient underwent resection of the lung mass, and there was no recurrence of the lung after 1 month's follow-up. Conclusions: By examining these two cases, we have a better understanding of the clinicopathological features of secretory carcinoma, which will help to improve the accuracy of pathological diagnosis.

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Section: Historymentioning

confidence: 99%

Two cases report of secretory carcinoma of the salivary gland in the lung: one primary and one metastatic after many years

Wang¹,

Xie²,

Li³

et al. 2022

Gland Surg

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“… 3 , 4 This tumor typically occurs in the major salivary glands; however, SC can also occur in atypical locations arising in minor salivary glands. 5 , 6 The histopathologic diagnosis of SC may be challenging, although it is more commonly recognized since its classification. Initially identified as an aggressive subset of acinic cell carcinomas, it is now recognized to be a more indolent tumor, but with a spectrum of clinical behavior.…”

Section: Introductionmentioning

confidence: 99%

“…In addition, the initial characteristic t(12,15) (p13;q25) translocation resulting in the ETV6‐NTRK3 fusion transcript has been noted in these tumors, and more recently, occurring with some regularity associated with RET fusions 3,4 . This tumor typically occurs in the major salivary glands; however, SC can also occur in atypical locations arising in minor salivary glands 5,6 . The histopathologic diagnosis of SC may be challenging, although it is more commonly recognized since its classification.…”

Section: Introductionmentioning

confidence: 99%

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Diagnostic challenges and successful organ‐preserving therapy in a case of secretory carcinoma of minor salivary glands

et al. 2021

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Background Secretory carcinoma is a more recently described subtype of salivary gland carcinoma that may pose diagnostic challenges and frequently harbors NTRK fusions that may successfully be targeted by TRK inhibitors in advanced disease. Case We present the case of a female patient with secretory carcinoma arising in the base of tongue with persistent disease after debulking surgery and definitive chemoradiation. As an alternative to salvage surgery, which would have resulted in significant impairment of swallowing and speech function, a targeted therapy with the TRK‐inhibitor larotrectinib against an identified ETV6‐NTRK3 fusion product was initiated. Larotrectinib treatment has been well tolerated, resulted in durable complete response and the patient maintains good swallowing and speech function. Conclusion The presented case underscores the importance of the accurate diagnosis of secretory carcinoma. It further highlights the impact of molecular testing as targeted therapies may play an important role in the management of advanced salivary gland cancers.

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“…Approximately 60-70% of the cases were located in the parotid gland, and the total number of cases in the major salivary glands, including the submandibular gland, accounted for approximately 70-80% [6,7]. Overall, there are few reports describing minor salivary gland-derived SCs [8][9][10]. The true frequency of occurrence is unclear because SC is a recently described disease entity, and it is possible that a few SC cases were previously diagnosed as AciCC.…”

Section: Introductionmentioning

confidence: 99%

Diagnosis and Treatment of Secretory Carcinoma of the Oral Cavity: Report of Two Cases and Pooled Analysis of the Recent Literature

Ogawa¹,

Yokoo²,

Yamaguchi³

et al. 2021

Preprint

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Background: Secretory carcinoma (SC) is a malignancy of the salivary glands, which is similar to SC of the breast in terms of its association with neurotrophic tyrosine receptor kinase (NTRK) fusion-positive gene. SC is a recently described salivary gland tumor, and there are few reports describing oral minor salivary gland-derived SC. We reported two cases of SC in the oral cavity and reviewed the literature.Case presentation: The patients included a 65-year-old Japanese woman presented with a mass of the upper lip and an 84-year-old Japanese man presented with a mass on the buccal mucosa, respectively. Diagnosis was based on histomorphological and immunohistochemical findings and identification of a specific translocation of the ETS variant 6-neurotrophic receptor tyrosine kinase 3 (ETV6-NTRK3) gene fusion. Case 1 was finally diagnosed using reverse transcription-polymerase chain reaction using formalin-fixed paraffin-embedded tissue samples, while case 2 was diagnosed using fluorescence in situ hybridization analysis. Conclusion: It is highly likely for many cases of SC to be initially diagnosed as acinic cell carcinoma (AciCC) owing to their similar histological findings. The treatment strategy for minor salivary gland-originated SC is the same as that for AciCC, but SC is often highly malignant and involves a high risk of cervical lymph node metastasis. Making an accurate diagnosis in cooperation with pathologists and confirming the presence of the ETV6-NTRK3 fusion gene using genetic analysis is important.

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Secretory Carcinoma of Minor Salivary Gland in Buccal Mucosa: A Case Report and Review of the Literature

Cited by 9 publications

References 34 publications

Two cases report of secretory carcinoma of the salivary gland in the lung: one primary and one metastatic after many years

Two cases report of secretory carcinoma of the salivary gland in the lung: one primary and one metastatic after many years

Diagnostic challenges and successful organ‐preserving therapy in a case of secretory carcinoma of minor salivary glands

Diagnosis and Treatment of Secretory Carcinoma of the Oral Cavity: Report of Two Cases and Pooled Analysis of the Recent Literature

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