Primary Small Cell Neuroendocrine Carcinoma of the Sublingual Gland Treated With Definitive Chemoradiotherapy: A Case Report

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ABSTRACT Primary sublingual gland small cell neuroendocrine carcinoma (SCNEC) is extremely rare, with only one prior case reported, and no site‐specific standard of care has been established. We report a case of primary sublingual gland SCNEC treated with definitive concurrent chemoradiotherapy (CRT). A 69‐year‐old man presented with a 21 × 15‐mm right sublingual mass showing fluorodeoxyglucose uptake (SUVmax 9.9) on PET/CT. No nodal or distant disease was found. Biopsy revealed small cell morphology positive for INSM1/synaptophysin and pan‐cytokeratin, with a high Ki‐67 index (> 95%). IMRT delivered 70 Gy in 35 fractions to the primary tumor with elective bilateral nodal irradiation (levels I–V, 46 Gy). Cisplatin (80 mg/m 2 ; day 1) and etoposide (100 mg/m 2 ; days 1–3) were administered every 3 weeks for four cycles. Grade 3 oral mucositis and grade 2 neutropenia resolved. At 24 months post‐CRT, the patient remains disease‐free with preserved daily function. Definitive concurrent CRT appears to be a feasible and effective organ‐preserving treatment option for limited‐stage primary sublingual gland SCNEC, with durable disease control at 24 months in this case.

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