ArticleInternational journal of surgery case reports2026
Primary large-cell neuroendocrine carcinoma of the submandibular gland arising from a long-standing benign tumor: case report.
Article in International journal of surgery case reports, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
Background: Large-cell neuroendocrine carcinoma (LCNEC) is an aggressive malignancy that commonly arises in the lungs. Primary salivary gland involvement is exceptional, presenting a major diagnostic challenge in differentiating it from metastatic pulmonary disease. Case presentation: A 62-year-old man presented with a right submandibular mass that had been stable for 16 years before rapidly expanding. Imaging revealed a massive 11 cm necrotic submandibular tumor with disseminated pulmonary and hepatic metastases. Following partial debulking, pathology revealed a high-grade neuroendocrine tumor (Ki-67 index 85%) arising alongside a benign mesenchymal component. Immunohistochemistry showed diffuse CD56 positivity and TTF-1 negativity. Discussion: While histology mimics pulmonary variants, the dominant pre-existing cervical mass and chronologic evolution strongly suggest a primary salivary origin, likely representing malignant transformation of a benign lesion. Conclusion: This case highlights the rare phenomenon of primary salivary LCNEC developing within a long-standing benign mass. It underscores the importance of including high-grade neuroendocrine carcinoma in the differential diagnosis of rapidly expanding salivary lesions.
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