Background:Circumscribed palmar or plantar hypokeratosis defines a benign condition clinically presented as a well-defined depressed area on the skin. It is characterized by a peculiar histopathological picture with the abrupt thinning of the horny layer of the epidermis.Main observations:We present a case of a 17-year-old man, who presented with a one-year history of an occasionally painful lesion on his right sole. The diagnosis of circumscribed plantar hypokeratosis was confirmed by histopathology. The lesion was successfully treated with calcipotriol ointment.Conclusions:Circumscribed palmar or plantar hypokeratosis is a benign condition that typically affects the hands. Different therapeutic approaches have been unsuccessful. In our patient the lesion was at a less frequent plantar location and the lesion responded to topical calcipotriol.