Kidney transplant recipients (KTR) have a higher risk of developing malignancies compared to the general population, due to the immunosuppressive regimens which can promote the oncogenesis process. The incidence of de novo non-melanoma skin cancer (NMSC) in KTR is greater than in the general population. Basal cell carcinoma (BCC) represents one of the most frequent malignancies in KTR. Sonidegib is a Hedgehog signaling pathway inhibitor approved for the treatment of locally advanced basal-cell carcinoma (LABCC) that following surgery or radiation therapy, or is given to those candidates who are not eligible to surgery or radiation therapy. This paper reports the case of a kidney transplant patient, who developed severe acute kidney injury (AKI) due to rhabdomyolysis (RML) induced by sonidegib therapy which required renal replacement therapy (RRT).