| Summary: | Abstract Background Atypical melanocytic naevus (AMN) of the urethral meatus is very rare with only a handful of reported cases. It is important to note that the rarity of AMN in the penis may contribute to a delay in diagnosis and treatment, as well as a lack of awareness among healthcare providers. This is the first case report to describe workup and management of AMN of the urethral meatus of a male, making it a valuable contribution to the literature. Case presentation A Caucasian male patient in his fourth decade presented with a several month history of a pigmented lesion on his glans penis. The differential diagnosis was of malignant melanoma but after biopsy and specialist pathological review, atypical melanocytic naevus was diagnosed. A punch biopsy, followed by two wide local excisions, was undertaken for complete excision and evaluation of the lesion. Conclusion Since AMN cannot be clinically distinguished from malignant melanoma, timely histological evaluation of the lesion is essential for workup. In this case, site and visibility of the lesion, expedient biopsy, and tissue analysis resulted in identification and excision of the naevus early enough for penis preserving surgery to be performed.
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