Hyalinizing clear cell carcinoma (HCCC) is a rare neoplasm affecting mainly the minor salivary glands of the oral cavity. We describe an unusual case of HCCC involving the tonsil and its successful management. A 67-year-old Hispanic woman was discovered to have an asymptomatic right tonsillar mass on routine clinic visit that revealed HCCC on biopsy. A right radical tonsillectomy was performed and pathology confirmed HCCC with positive deep surgical margins. She declined the recommended adjuvant radiation therapy. A follow-up CT of the neck with contrast done a year later revealed a suspicious area of enhancement around the prior resection margin with regional cervical lymphadenopathy. Further workup, including biopsy, confirmed local recurrence. She was treated with definitive cisplatin-based chemoradiotherapy, achieving complete response. She remains without recurrence with more than 24 months of follow-up.
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