Calciphylaxis is a complication associated with abnormal mineral and bone disorder of chronic kidney disease (CKD-MBD) and the etiology is largely unknown. We encountered a male Caucasian patient with a history of long-term hemodialysis who developed multiple painful skin ulcers on both thighs. The diagnosis was made based on physical findings and skin biopsy, which showed typical findings of calciphylaxis. Together with conventional treatment for CKD-MBD, the patient was given periodic intravenous infusions of sodium tiosulphate, which successfully improved the skin ulcers. Although the incidence is rare, physicians encountering CKD in dialysis practice must be alert to this complication.