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Rationale: Many previous studies have investigated the necessity of routine histopathological analysis of tonsillectomy specimen, and most recent studies have suggested that such an analysis is not justified in asymptomatic patients or those with no risk factors for malignancy., Patient Concerns: A 59-year-old man diagnosed with obstructive sleep apnea underwent surgery, including tonsillectomy; a tonsil specimen was sent to the department of pathology., Diagnosis: Although the patient did not exhibit any tonsil-related signs or symptoms, nor did the appearance of the tonsil appear to be pathological, the right tonsil specimen was diagnosed with squamous cell carcinoma, with detection of human papilloma virus 16., Interventions: Chemotherapy and radiotherapy were used to treat the tonsil cancer., Outcomes: No recurrence was observed during the 4-year follow-up., Lessons: In the future, it may be necessary to reinvestigate the necessity of routine histopathological analysis of tonsillectomy specimens in asymptomatic patients, considering the drastically increased rate of detection of human papilloma virus-related oropharyngeal squamous cell carcinomas in these patients.