Mycetoma is a neglected tropical disease that causes tremendous suffering and misery to affected patients. In Sudanese rural communities, many patients hide their lesions, especially those located in the perineal, scrotal, inguinal and genital regions, and they regard them as sources of social stigma. We report the case of a 45-year-old male from northern Kordofan State, Sudan, who presented with multiple discharging sinuses in the perineal region. Clinical diagnosis was actinomycetoma and fine-needle aspiration cytology was consistent with Actinomadura pelletieri, confirmed by culture and biochemical analysis. To our knowledge, this is the first case of A. pelletieri with urethral complications and subsequent implications for management.
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