Bowen's disease developing on mucous or mucocutaneous regions is clinically called erythroplasia of Queyrat. We report herein a 56-year-old male with Bowen's disease extending from the penis shaft to the glans penis, and urethral meatus. Physical examination revealed bright red velvety plaques on the prepuce and glans penis and an irregularly pigmented scaly lesion on the dorsum of his penis shaft. Histopathological findings of both lesions were compatible with those of Bowen's disease, supporting the concept that erythroplasia of Queyrat and Bowen's disease should be regarded as one clinicopathologic entity. A partial penectomy was finally performed, because tumor cells were pathologically observed in the mucous epithelium of the urethra. Although several therapeutic modalities exist for Bowen's disease on the external genitalia, treatment options are limited when Bowen's disease extends to the urethral meatus. We discussed the recent therapeutic modalities in genital Bowen's disease.