Reversible posterior leukoencephalopathy syndrome (RPLS) is one of the important adverse events following organ transplantation, associated with calcineurin inhibitors (CNIs). We describe a case of 54-year-old woman, who was diagnosed with RPLS within weeks after transplantation. Considering the risk of causing fatal rejection by discontinuation of CNIs, the immunosuppressive regimen of the patient was switched from a cyclosporine A-based regimen to a tacrolimus-based regimen. The patient recovered rapidly from RPLS following the switch to tacrolimus. This case demonstrated that not only discontinuation but also a substitution of CNIs would be a valid treatment option for RPLS in transplant recipients.