Background: Several studies reported the presence of the hypopigmentation in Extramammary Paget’s disease (EMPD), however, in-depth analysis regarding its clinical implication is lacking. Objectives: To evaluate the clinical characteristics of EMPD in Korean population and to find out the implication of hypopigmentation on the clinical outcome. Methods: We retrospectively reviewed 142 cases of EMPD diagnosed from a single tertiary hospital from 2005 to 2019. Baseline characteristics of the patients and the presence of hypopigmentation, multifocality, pattern, and anatomical location were analyzed. Also, the number of stages of the Moh’s micrographic surgery (MMS) and recurrence were evaluated in relation to the hypopigmentation. Results: 65.5% (n=93) of the patients showed hypopigmentation, while 31.0% (n=44) revealed no lesion of hypopigmentation. Recurrence was not statistically correlated with the presence of hypopigmentation. The average number of MMS stage was 2.92 stages for the hypopigmentation group, compared to 1.82 stages of the non-hypopigmentation group (95% CI = -1.770 ~ -0.425, p=0.002) The most commonly involved area of the hypopigmentation was the penile shaft, followed by the scrotum, pubis and labia major. Conclusion: Hypopigmented lesions may disguise the definite tumor margin, thus, the status of hypopigmentation must also be considered when deciding the surgical margin. Molecular mechanism of hypopigmentations occurring in EMPD patients’ needs further investigation.