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Tinnitus as an unusual presentation of Schneiderian papillomatosis.INTRODUCTION: Primary Schneiderian papillomatosis of the middle ear and mastoid cavity is extremely rare. It is frequently associated with intermittent unilateral otorrhoea and mass in the middle ear and mastoid cavity. METHODS: Case presentation, symptoms, diagnostic criteria, management and literature review are discussed. CONCLUSION: Schneiderian papillomatosis is an important differential diagnosis of mass in the middle ear and mastoid cavity, and tinnitus as a presenting symptom has not been reported before. Primary radical treatment is essential in preventing tumour recurrence.