Mal de Meleda is a rare autosomal recessive skin disease which is known as keratoderma palmoplantaris transgradiens. Here we report a case of Mal de Meleda who had skin lesions in the residual limb and pseudoainhum in the thigh after traumatic lower leg amputation. A 71-year-old female was admitted to our tertiary hospital for prosthetic rehabilitation. On the physical examination, thickening of the skin on palms, left sole and residual limb was present. The patient reported that she had these skin lesions since infancy and she realized new skin lesions after amputation in the residual limb. We requested dermatology consultation and she was diagnosed as Mal de Meleda. To our knowledge, this is the first Mal de Meleda case in the literature with new lesions at the residual limb. Although exact pathophysiological mechanisms are not well known in Mal de Meleda, prosthesis use might have accelerated disease process in our patient.