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A 64-year-old man with Kaposi sarcoma in clinical remission after treatment with pegylated liposomal doxorubicin and a history of deceased-donor kidney transplantation 4 years prior presented with a slowly progressive increase in his serum creatinine level, well-controlled hypertension, stable subnephrotic-range proteinuria, and bland urinary sediment. An allograft kidney biopsy demonstrated thrombotic microangiopathy, without clinical or laboratory features of systemic involvement. Based on the timing of drug initiation preceding thrombotic microangiopathy, complete recovery after drug withdrawal, and the absence of other etiologies, it was concluded that pegylated liposomal doxorubicin was the likely cause of kidney-limited thrombotic microangiopathy. When pegylated liposomal doxorubicin was resumed, the patient developed hypertension and kidney allograft dysfunction. A new kidney biopsy was not performed because of the overall risk benefit. The case highlights the importance of recognizing novel etiologies of thrombotic microangiopathy in kidney transplant patients with malignancy. Although Kaposi sarcoma has not been linked to thrombotic microangiopathy, pegylated liposomal doxorubicin has been increasingly associated with drug-induced thrombotic microangiopathy. To our knowledge, this is the first case report that etiologically links pegylated liposomal doxorubicin to kidney-limited thrombotic microangiopathy in a kidney transplant patient.