Extramammary Paget disease (EMPD) is a rare cutaneous malignancy, and unresectable or metastatic cases are challenging to treat due to high recurrence rates with existing chemotherapy.1,2 Previous studies on EMPD genetic alterations have mainly reported the involvement of HER2 overexpression and the PI3K/AKT/mTOR pathway.1,3-5 However, due to its rarity, genetic alterations in metastatic cases remain underresearched. This study aimed to investigate genetic alterations as potential therapeutic targets and their clinical implications in EMPD using comprehensive genomic profiling test results.